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Educational late effects among survivors of childhood cancer McMillan, Amy 2007

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E D U C A T I O N A L L A T E E F F E C T S A M O N G S U R V I V O R S O F C H I L D H O O D C A N C E R by A M Y M C M I L L A N B . S c , Mount Al l i son University, 2002 A THESIS S U B M I T T E D I N P A R T I A L F U L F I L L M E N T O F T H E R E Q U I R E M E N T S F O R T H E D E G R E E O F M A S T E R OF A R T S in T H E F A C U L T Y OF G R A D U A T E S T U D I E S (Special Education) T H E U N I V E R S I T Y OF B R I T I S H C O L U M B I A October 2007 © A m y M c M i l l a n , 2007 A B S T R A C T Increased survival rates of childhood cancer have meant a growing population of survivors within the education system. Although research suggests that survivors face educational difficulties, methodological shortcomings and lack of consensus have contributed to difficulties interpreting this literature. Moreover, there exists a paucity of literature objectively measuring survivors' educational outcomes, particularly achievement. In this population-based research, 782 survivors of childhood cancer from the B C cancer registry, and BC school system from 1995-2004, were age and gender-matched with a randomly selected control group of 8386 BC schoolchildren. Objective educational measures including Foundation Skills Assessments (FSAs), Provincial examinations, and special education designations from the B C Ministry of Education were compared between the survivor and control cohorts; potential disease-related risks among survivors were assessed. Survivors were significantly more likely than controls to have special education or physical disability designations and performed significantly more poorly on several FSAs. Notably, once survivors of central nervous system (CNS) tumours and leukemia were excluded from the analysis, there were no significant achievement differences. Survivors younger at diagnosis (<2 years) had higher educational achievement, despite having more hearing and visual impairments than survivors older at diagnosis (>5 years). Childhood cancer survivors appear at increased risk for special education utilization. In particular, survivors of leukemia and CNS tumours may be at increased risk for poor educational achievement and special education designations. It is important that potential adverse educational outcomes and associated risk factors be identified such that surveillance and appropriate interventions be provided to ensure survivors a successful educational experience. ii T A B L E O F C O N T E N T S A B S T R A C T i i T A B L E O F C O N T E N T S i i i L IST O F T A B L E S v A C K N O W L E D G E M E N T S . v i D E D I C A T I O N v i i C O - A U T H O R S H I P S T A T E M E N T v i i i C H A P T E R 1: I N T R O D U C T I O N A N D L I T E R A T U R E R E V I E W 1 1.1 CHILDHOOD CANCER SURVIVAL RATES 1 1.2 LATE EFFECTS 1 1.3 EDUCATIONAL LATE EFFECTS 2 1.3.1 Use of Special Education 3 1.3.2 Grade Retention 3 1.3.3 Educational Attainment 4 1.3.4 Academic Achievement 4 1.4 RISK FACTORS 5 1.4.1 Cancer Diagnosis 5 1.4.2 Treatment Modali ty 6 1.4.3 Therapeutic Dosage Level 7 1.4.4 Age at Diagnosis or Treatment 7 1.4.5 Time Interval since Diagnosis 8 1.4.6 Gender 8 1.4.7 Socioeconomic Status (SES) 9 1.5 LARGE-SCALE NATIONAL COHORT EDUCATIONAL STUDIES 9 1.5.1 Canadian Study 9 1.5.2 Dutch Study .' 11 1.5.3 U . S . Study 12 1.5.4 Danish Study 14 1.5.5 Finnish Studies 15 1.6 METHODOLOGICAL SHORTCOMINGS OF PREVIOUS RESEARCH 16 1.7 GAPS WITHIN THE LITERATURE 17 1.8 OBJECTIVES AND HYPOTHESES 18 1.9 REFERENCES 20 C H A P T E R 2: E D U C A T I O N A L L A T E E F F E C T S A M O N G S U R V I V O R S O F C H I L D H O O D C A N C E R I N B R I T I S H C O L U M B I A .26 2.1 BACKGROUND 26 2.2 MATERIALS AND METHODS 29 2.2.1 Statistical Analysis 32 2.3 RESULTS 32 2.3.1 Sociodemographic and Clinical Characteristics of Survivors and Controls 32 2.3.2 Educational Outcomes of Survivors and Controls 35 2.3.3 Educational Outcomes by Diagnosis 37 2.3.4 Clinical Factors Related to Educational Outcomes among Survivors 39 2.4 CONCLUSIONS 42 2.5 REFERENCES 48 C H A P T E R 3: D I S C U S S I O N 53 3.1 CONCLUSIONS 53 3.2 STRENGTHS AND LIMITATIONS 56 3.3 SIGNIFICANCE 59 3.4 FUTURE RESEARCH .62 3.5 REFERENCES 65 A P P E N D I C E S 68 APPENDIX A - U B C ETHICS APPROVAL 68 I V L I S T O F T A B L E S Table 2.1 Sociodemographic Characteristics of Survivors and Controls 33 Table 2.2 Clinical Characteristics of Survivors 34 Table 2.3 Educational Outcomes of Survivors and Controls 36 Table 2.4 Educational Outcomes of Males and Females 37 Table 2.5 Educational Outcomes by Diagnosis 39 Table 2.6 Clinical Factors Related to Selected Educational Outcomes among Survivors 41 v A C K N O W L E D G E M E N T S I would like to express my sincere thanks to the faculty, staff, and my fellow students of U B C who have supported me throughout my studies. In particular, my supervisor Dr. Linda Siegel is owed immense appreciation for her expertise and helpful guidance. I thank my thesis committee members Mary McBr ide and Dr. Bruno Zumbo for their time and invaluable input in this project. I offer my gratitude to Mar ia Lorenzi for her statistical direction and patience for my never-ending questions. Lastly, my appreciation is also extended to the investigative members of The Childhood/ Adolescent/ Young Adulthood Cancer Survivorship ( C A Y A C S ) research program, of which this project was a part. The C A Y A C S research program is a population-based data linkage study of the long-term outcomes and health utilization of survivors of childhood cancer l iving in the province of British Columbia, Canada and is funded by the Canadian Cancer Society through the National Cancer Institute of Canada, Grant#PPGO 16001. v i l b my husband, (Dan, for His Cove andnever-failing 6eCiefin me, and to my family for their support and encouragement without 'whom the completion of this research project would not have been possible. i vii C O - A U T H O R S H I P S T A T E M E N T The following individuals have contributed to the manuscript preparation: Linda Siegel, Mary McBride , Bruno Zumbo, Maria Lorenzi, John Spinelli, Karen Goddard, Sheila Pritchard, and Paul Rogers. v m C H A P T E R 1: I N T R O D U C T I O N A N D L I T E R A T U R E R E V I E W 1.1 Ch i ldhood Cancer Surv iva l Rates In Canada an average of 1289 children aged birth to 19 years were diagnosed with cancer each year from 1999 to 2003 (Canadian Cancer Society/National Cancer Institute of Canada, 2007). In the Canadian province of British Columbia an average 157 children were diagnosed with cancer each year from 1995-2004 (BC Cancer Agency, 2007). Advances in medical treatment have been made within the past 20 years that have resulted in increased survival rates for children diagnosed with cancer ( M . Smith & Hare, 2004). B y 2003, 5-year survival rates for children (aged 0-14 years) diagnosed with cancer had reached 79.6% (Ries et al., 2007). Thus, as never before, there exists an ever growing population of survivors of childhood cancer who are entering and attending school. Notably, these children outnumber many traditional special education populations within the United States (Peckham, 1991). Within the United Kingdom, approximately 1 in 1000 young adults is a survivor of childhood cancer (Taylor et al., 2004). 1.2 Late Effects Survival is now a tangible possibility for children diagnosed with cancer, but it has not come without some impact upon the children who survive. Two thirds of all survivors of childhood cancer w i l l experience at least one late effect after surviving cancer (National Cancer Policy Board, 2003). A late effect is considered any complication, disability, or adverse outcome resulting from the cancer itself, the treatment of the cancer, or both, that continues or develops more than five years after t 1 The most frequently observed late effects of childhood cancer are neurocognitive in nature (National Cancer Policy Board, 2003). Given this, it is not surprising that there exists an abundance of studies that have investigated the neurocognitive late effects of surviving childhood cancer (V. Anderson, Godber, Smibert, Weiskop, & Ekert, 2000; Bleyer et al., 1990; Butler, H i l l , Steinherz, Meyers, & Finlay, 1994; D . H i l l , Ciesielski, Sethre-Hofstad, Duncan, & Lorenzi, 1997; Kolotas et al., 2001; Rubenstein, Varni , & Katz, 1990; D . Waber, Bernstein, Kammerer, & Tarbell, 1992). Reductions in neuro-cognitive functioning; in particular, decreases in IQ have been reported (Christie, Leiper, Chessells, & Vargha-Khadem, 1995; Fogarty et al., 1988; Moleski , 2000; Robaey et a l , 2000), but it has been noted that IQ may not represent the best measure o f neurocognitive outcome, given that it is not sensitive enough to identify the deficits or areas that may require accommodation or remediation (C. Armstrong, Gyato, Awadalla, Lustig, & Tochner, 2004). Moreover, although a decrease in measures of intelligence, memory, and other central nervous system functions may have been determined by some studies, there is no decisive demonstration that this is associated with a failure to achieve (Allen, Malpas, & Kingston, 1990). 1.3 Educat ional Late Effects Many survivors report that their education has been effected as a result o f their childhood cancer (Langeveld, Stam, Grootenhuis, & Last, 2002). In one study, as many as 67% of survivors stated that their education had suffered due to their cancer (Evans & Radford, 1995), and in another study it was found that 45% of survivors felt that their cancer had adversely impacted their educational achievement to a great or very great extent (Dolgin, Somer, Buchvald, & Zaizov, 1999). 2 1.3.1 Use of Special Education Special education utilization has been well documented among survivors of childhood cancer (F. Armstrong, Blumberg, & Toledano, 1999; Fogarty et a l , 1988; Jenkin, Danjoux, & Greenberg, 1998; Kazak, Christakis, Alderfer, & Coiro, 1994; Kingma, Mooyaart, Kamps, Nieuwenhuizen, & Wilmink, 1993; Mulhern, Wasserman, Friedman, & Fairclough, 1989; Peckham, Meadows, Bartel, & Marrero, 1988; Rubenstein, Varni , & Katz, 1990; D . Waber, Bernstein, Kammerer, & Tarbell, 1992). Reviews report that special education services are often necessary for survivors (D. Anderson et al., 2001) and that increased proportions of survivors require special education (Robaey et al., 2000; Robison et al., 2005) or the use of special education services including tutoring, resource room programs, and special education classes or schools (Stehbens et al., 1991). A review of the literature has indicated that as many as 39-69% of survivors qualify for special education (Coniglio & Blackman, 1995). Studies have found that significantly more survivors than siblings are, or have been, enrolled in special education programs or classes (Kingma, Rammeloo, van Der Does-van den Berg, Rekers-Mombarg, & Postma, 2000; Mi tby et a l , 2003) and learning disability programs (Haupt et a l , 1994). 1.3.2 Grade Retention A n increased rate of grade repetition has been observed among survivors of childhood cancer (I. Moore, Glasser, & Abl in , 1988). Compared to siblings (Haupt et al., 1994) and controls (Buizer, de Sonneville, van den Heuvel-Eibrink, & Veerman, 2006) survivors are more likely to have repeated a grade. It has been suggested that as many as 26% o f survivors w i l l have repeated at least one or more grades during their school career (Coniglio & Blackman, 1995; Mulhern, Wasserman, Friedman, & Fairclough, 1989). 3 1.3.3 Educational Attainment The educational attainment or level of education completed by survivors is another educational outcome that has been reported within the literature (Humpl, Fritsche, Bartels, & Gutjahr, 2001; L i , Winston, & Gimbrere, 1984; Syndikus, Tait, Ashley, & Jannoun, 1994). Survivors have been found less likely to graduate high school when compared with siblings (Kelaghan et al., 1988; Nagarajan et al., 2003). A s well , some research suggests that those survivors who do graduate from high school are less likely to enter college or continue on to secondary education than siblings (Evans & Radford, 1995; Kelaghan et al., 1988) and to have lower levels of secondary education than their siblings (Kingma, Rammeloo, van Der Does-van den Berg, Rekers-Mombarg, & Postma, 2000; Langeveld, Stam, Grootenhuis, & Last, 2002). 1.3.4 Academic Achievement Academic difficulties and poor or low academic achievement have been frequently reported within reviews of the literature (D. Anderson et al., 2001; Brown & Madan-Swain, 1993; Cousens, 1997; Patenaude & Kupst, 2005; Robaey et al., 2000; Schwartz, 1999). The most commonly observed measures of academic achievement among studies of survivors are the Wide Range Achievement Test ( W R A T ) and anecdotal reports of school performance (V. Anderson, Godber, Smibert, & Ekert, 1997; V . Anderson, Smibert, Ekert, & Godber, 1994; V . Anderson, Godber, Smibert, Weiskop, & Ekert, 2000; F. Armstrong & Mulhern, 1999; Copeland et al., 1985; Moehle, Berg, Ch'ien, & Lancaster, 1983; Smibert, Anderson, Godber, & Ekert, 1996). Parents of survivors have reported that their children perform more poorly on everyday cognitive and academic tasks (F. Armstrong & Mulhern, 1999) and have poorer school performances than children without cancer (V. Anderson, Smibert, Ekert, & Godber, 1994). A t school, teachers have suggested that survivors express more learning difficulties (Jannoun & Chessells, 1987) and 4 make slower academic progress compared to other students (Raymond-Speden, Tripp, Lawrence, & Holdaway, 2000). W R A T reading, spelling, and arithmetic scores o f survivors have been found to be lower than expected within the general population (Seaver et al., 1994; D . Waber et al., 1990). Furthermore, it has commonly been noted that the greatest or more common deficit exists in arithmetic or mathematic skills (Coniglio & Blackman, 1995; Duffner, 2004; Gamis & Nesbit, 1991; Kaemingk, Carey, Moore, Herzer, & Flutter, 2004; Moleski , 2000; B . Moore, 2005; Mulhern & Palmer, 2003; Peckham, 1991; Peckham, Meadows, Bartel, & Marrero, 1988; Stehbens et al., 1991; Whitt, Wells, Lauria, Wilhelm, & M c M i l l a n , 1984). 1.4 R i sk Factors A number of disease-related, personal, and socioeconomic factors are associated with an increased risk of adverse educational late effects among survivors of childhood cancer. These confounding variables include: the cancer diagnosis, treatment modality, therapeutic dose, age at diagnosis or treatment, time elapsed since diagnosis, gender and socioeconomic status (SES). 1.4.1 Cancer Diagnosis Specific diagnoses including leukemia; in particular, acute lymphoblastic leukemia ( A L L ) , Hodgkin's lymphoma, central nervous system (CNS) tumours, and neuroblastoma have been associated with an increased risk of educational late effects (Barrera, Shaw, Speechley, Maunsell, & Pogany, 2005; Langeveld et al., 2003; Mi tby et al., 2003). A L L is the most frequently diagnosed cancer in childhood followed by diagnoses of C N S tumours (National Cancer Pol icy Board, 2003). Between 1999 and 2003 A L L was diagnosed in 1197 children and C N S tumours in 1068 children in Canada (Canadian Cancer Society/National Cancer Institute of Canada, 2007). A L L is a disease of the blood, and while it is diagnosed in all ages of children, the highest rate of incidence occurs among those aged two to six years (National Cancer Policy 5 Board, 2003) and peaks dramatically at ages two to three years ( M . Smith, Ries, Gurney, & Ross, 1999). The diagnosis of C N S tumours, the majority being brain tumours, is most prevalent between infancy and age seven (National Cancer Policy Board, 2003) . The National Cancer Institute reported that U . S . 5-year survival rates from 1996 to 2003 for A L L and C N S tumours diagnosed in children aged 0-14 years were 8 7 . 2 % and 7 4 . 1 % respectively (Ries et al., 2007). This suggests that the children with the most common diagnoses, and thus representative of the largest population of survivors, are also the most likely to experience adverse educational late effects and be present within our school systems due to the relatively high survival rates. 1.4.2 Treatment Modality Treatment involving cranial or craniospinal radiation therapy (CRT) and/or intrathecal methotrexate (IT M T X ) has consistently been associated with an increased risk of neurocognitive deficits and poorer educational outcomes (Copeland, Dowell , Fletcher, Bordeaux et al., 1988; Kingma et a l , 2001 ; Mi tby et al., 2003 ; Precourt et al., 2002). Survivors treated with C R T are commonly found to have lower W R A T scores than controls or survivors treated with chemotherapy only (V. Anderson, Godber, Smibert, & Ekert, 1997; V . Anderson, Smibert, Ekert, & Godber, 1994; V . Anderson, Godber, Smibert, Weiskop, & Ekert, 2000 ; Copeland et al., 1985; Raymond-Speden, Tripp, Lawrence, & Holdaway, 2000 ; Smibert, Anderson, Godber, & Ekert, 1996). A s well , survivors who received radiation were found to have lower IQ scores and were more often identified as "learning disabled" in comparison with survivors who received no radiation (von der Weid, 2001). Survivors treated with a combination of C R T and IT M T X have displayed deficits in neuropsychological functioning and poorer performance on school achievement scores (Butler, H i l l , Steinherz, Meyers, & Finlay, 1994; J . H i l l et al., 1998). 6 1.4.3 Therapeutic Dosage Level Not only is the treatment modality a risk factor to be considered, but also the dosage at which the treatment is received. Higher therapeutic doses of C R T and M T X have been correlated with poorer academic achievement and cognitive performance (V. Anderson, Godber, Smibert, & Ekert, 1997; Buizer, de Sonneville, van den Heuvel-Eibrink, & Veerman, 2006; Kingma, Mooyaart, Kamps, Nieuwenhuizen, & Wilmink, 1993). Specifically, survivors treated with 2400cGy (centigrade) of radiation had lower W R A T scores than those treated with only 1800cGy (Halberg et al., 1992; I. Moore, Kramer, Wara, Halberg, & A b l i n , 1991; Smibert, Anderson, Godber, & Ekert, 1996) and lower IQ scores (Halberg et al., 1992; Kingma, Mooyaart, Kamps, Nieuwenhuizen, & Wilmink, 1993). Survivors who received a higher dose of M T X had poorer School Performance Index (SPI) scores for subtests including: total score, mathematics, and language skills than those who received a low dose of M T X (Buizer, de Sonneville, van den Heuvel-Eibrink, & Veerman, 2006). 1.4.4 Age at Diagnosis or Treatment Children who are diagnosed at a younger age and/or receive treatment at a younger age are more likely to have cognitive deficits, lower academic achievement, and poorer educational outcomes (Kingma, Mooyaart, Kamps, Nieuwenhuizen, & Wilmink, 1993; Kingma, Rammeloo, van Der Does-van den Berg, Rekers-Mombarg, & Postma, 2000). A greater need for special education, poorer W R A T scores and executive function tasks, lower school levels, and lower IQ scores have all been found to correlate with younger age at diagnosis (V. Anderson, Godber, Smibert, & Ekert, 1997; Copeland et al., 1985; Kingma, Mooyaart, Kamps, Nieuwenhuizen, & Wilmink, 1993; Kingma, Rammeloo, van Der Does-van den Berg, Rekers-Mombarg, & Postma, 2000; Seaver et al., 1994). Those who are less than five years of age at diagnosis or treatment 7 (especially C R T ) appear to be particularly at risk (Jannoun & Chessells, 1987; Smibert, Anderson, Godber, & Ekert, 1996). Moore and colleagues (I. Moore, Kramer, Wara, Halberg, & Abl in , 1991) found that children treated with 2400cGy of radiation before 48 months of age had lower IQ and W R A T scores than those treated after they were 48 months old. 1.4.5 Time Interval since Diagnosis Reduced cognitive and academic functioning has been associated with longer time intervals since diagnosis or treatment (V. Anderson, Godber, Smibert, & Ekert, 1997; Moehle, Berg, Ch'ien, & Lancaster, 1983; Rubenstein, Varni , & Katz, 1990). IQ scores of survivors have shown a decrease from evaluation at remission or diagnosis to evaluation four or five years later (Ochs et al., 1991; Rubenstein, Varni , & Katz, 1990). A s well , significant declines in W R A T reading, spelling, and arithmetic scores have been observed as time off therapy increased (Moehle, Berg, Ch'ien, & Lancaster, 1983; Mulhern, Fairclough, & Ochs, 1991). 1.4.6 Gender Female survivors of childhood cancer appear more at risk for poorer educational outcomes than males (Eiser, 1991; Haupt et al., 1994; Langeveld et al., 2003). Females are less likely to enter college, and those who do complete one year of college are less l ikely to complete four years compared to males (Kelaghan et al., 1988). Waber and colleagues (D. Waber, Bernstein, Kammerer, & Tarbell, 1992) found that females were more impaired than males according to a severity rating of 0-5 where 0 represented no services needed and 5 denoted the requirement of a fulltime special education placement. Differences in both Verbal IQ (VIQ) and Performance IQ (PIQ) scores have also been found between males and females. While males did not differ from normative scores regarding any IQ scores (Full Scale, Verbal, or Performance), females had significantly lower PIQ scores than normative data (Brown et al., 1998). In another 8 study, females more commonly displayed a significant decline (>15 points) in V I Q than their male counterparts (Mulhern, Fairclough, & Ochs, 1991). 1.4.7Socioeconomic Status (SES) Lower socioeconomic status has been associated with cognitive performance and educational outcomes among survivors (Butler, H i l l , Steinherz, Meyers, & Finlay, 1994; Mulhern & Palmer, 2003). In a review of the literature, it was noted that survivors from families of higher SES had better performance on measures of IQ and other neurocognitive abilities than survivors of families with lower SES (Mulhern & Palmer, 2003). Lower SES has been correlated with more impaired test scores on cognitive (IQ) and academic achievement ( W R A T ) measures (Butler, H i l l , Steinherz, Meyers, & Finlay, 1994). In addition, socioeconomic factors have been considered more powerful correlates of neuropsychological performance among survivors than treatment modality, age at diagnosis, or gender (Whitt, Wells, Lauria, Wilhelm, & M c M i l l a n , 1984). 1.5 Large-Scale Nat ional Cohor t Educat ional Studies 1.5.1 Canadian Study A recent publication (Barrera, Shaw, Speechley, Maunsell, & Pogany, 2005) represents one of the few studies to have explored the educational achievements o f survivors of childhood cancer using a national population-based cohort. Children who were diagnosed with cancer between 1981 and 1990, surviving more than five years after diagnosis, and still school age (less than 17 years old) at the time of study were identified from pediatric oncology centres and provincial cancer registries across Canada. O f the 1162 eligible survivors, parents or guardians of 800 survivors completed a questionnaire concerning the school experiences of their child. A control group of 1633 age and gender-matched individuals without a history of cancer were 9 randomly selected from the general population; of which, 923 parents or guardians completed the same questionnaire as the parents of survivors. Items on the questionnaire addressing educational outcomes were selected from the Chi ld Behaviour Checklist ( C B C L ) and included such questions as: "How would you rank your child's current school performance in the following subjects: English, French, History/Social Science, Mathematics, Science?" and "Has your child ever been in any of the following programs: learning disabled, special education?". Ratings of course performance were categorized as an above-average, average, below-average, or failing grade. Results indicated that survivors were significantly more l ikely than controls to have poorer educational outcomes. This was determined by the survivors' higher rates of attendance in learning disabled (19% vs. 7%) and special education programs (20% vs. 8%), more often repeating or failing a grade (21% vs. 9%), and more commonly experiencing academic or other school problems (46% vs. 23%) compared to controls. Furthermore, below-average or failing grades were more often obtained by survivors than controls in all areas of study, with the greatest differences noted in mathematics (29% vs. 10%), followed by English (26% vs. 11%), and then science (20% vs. 6%). These results remained significant after controlling for possible confounding variables including: gender, current health problems, stressful events, self-esteem, parental education, and year of study. Parents o f all cancer groups were more l ikely than controls to report poorer educational outcomes with the poorest educational outcomes reported by parents of survivors of C N S tumours, leukemia, and neuroblastoma. In addition, parents of survivors of C N S tumours were most l ikely to report failing grades in math and English compared to controls. Data was also extracted from the medical records of the survivor participants so as to investigate possible treatment-related risks. Those who were treated with C R T were more likely 10 to have reports of repeating a grade, participating in a learning disabled or special education program, and experiencing academic or other school problems compared to survivors who received treatment that did not involve C R T or IT M T X . Survivors whose treatment consisted of both C R T and IT M T X were also more likely to have been a member of a learning disabled class and experienced academic or other school problems than those who received neither treatment. Survivors who had at least two physical health problems were also more likely to report educational difficulties. Survivors who report higher self-esteem and those whose parents had higher postsecondary education were less likely to have academic or school problems. No significant differences in educational outcomes were found according to gender, age at study (6-12 years vs. 13-16 years), or age at diagnosis (<2 years vs. 2-4 years vs. >5 years). Barrera and colleagues (Barrera, Shaw, Speechley, Maunsell, & Pogany, 2005) are the first to examine the educational performance, as measured by school achievement, of survivors of childhood cancer and compare them to the general population in a national, large-scale, population-based study. This research adds to the current knowledge regarding the educational late effects of surviving childhood cancer by exploring a concern that had yet to be addressed, that is, how well are these individuals doing at school? Furthermore, this question was considered by investigating the actual academic achievement as measured by the parental rating of the grades attained in the subject areas of English, French, history/social science, math, and science (i.e., failed, below-average, average, above-average grade). 1.5.2 Dutch Study Langeveld and colleagues (Langeveld et al., 2003) gathered information regarding the highest level of education completed and enrolment in learning disabled programs among a sample of survivors of childhood cancer attending a long-term follow-up clinic established in the 11 Netherlands. Survivors of the long-term follow-up clinic had successfully completed cancer treatment at least five years earlier. Further inclusion criteria included a cancer diagnosis prior to 19 years of age and at least 16 years of age at the time of study. Comparisons were made with a control group of 1092 individuals with no history of cancer. Results of the author-lead, self-report questionnaire indicated poorer educational achievement of survivors of childhood cancer; specifically, significantly more survivors than controls were unable to participate in regular elementary or secondary school and had to be enrolled in learning disabled programs. Female survivors were less likely to complete high school or attain an advanced graduate degree than male survivors (30% vs. 39%). Survivors of C N S tumours had lower levels of education (i.e., less than high school) compared to survivors of leukemia or non-Hodgkin's lymphoma who were treated without C R T (80% vs. 59%) and survivors of solid tumours (80% vs. 59%). Lower educational levels were also achieved by survivors of leukemia and non-Hodgkin's lymphoma who were treated with C R T than survivors of leukemia and non-Hodgkin's lymphoma who were treated without C R T (78% vs. 59%) and survivors of solid tumours (78% vs. 59%). Furthermore, significant differences were found in the level of education attained with regards to C R T dosage. Survivors who did not receive C R T were less l ikely to have attained lower educational levels than those who received less than 2500cGy of C R T (59% vs. 82%) and those who received 2600cGy o f C R T or more (59% vs. 74%). 1.5.3 U.S. Study Mitby and colleagues (Mitby et al., 2003), similar to the Canadian study (Barrera, Shaw, Speechley, Maunsell, & Pogany, 2005), employed a questionnaire to investigate the use of special education services and the level of education attained by cancer survivors. From Childhood Cancer Survivor Study (CCSS) centres across Canada and the United States 20 276 5-12 year survivors of cancer, diagnosed before 21 years of age between 1970 and 1986 were identified as potential participants. O f those eligible, 12 431 completed questionnaires along with 3528 siblings from a potential 5800 who had been acknowledged. Questionnaire items pertaining to education included the highest grade or level of schooling completed and the type of high school diploma received: standard, as compared to general educational development credit (GED) which is an alternative method of completing high school through an equivalency test (Mitby et al., 2003). Participation in special education or learning disabled services was also examined, and for those who indicated that they had received these services the reason for these services and the grade levels in which they were received were sought. Results indicated that survivors of cancer were overall more likely to be enrolled in special education than their siblings (23% vs. 8%), with the greatest differences observed between females and same-gender siblings than males and same-gender siblings (e.g., female survivors o f leukemia diagnosed at 0-5 years were 7.6 times more likely vs. males with the same diagnosis and age group who were 2.9 times more likely). Survivors diagnosed before the age o f six years were most likely to have received special education, especially survivors of C N S tumours (OR: 18.8; 95% CI: 15.01-23.49), leukemia (OR: 4.4, 95% CI: 3.75-5.16) and Hodgkin's lymphoma (OR: 4.4, 95% CI: 2.64-7.24). Special education use was also significantly different by treatment era (overall rates were 17%, 22%, and 25% for those diagnosed during 1970-1975, 1976-1980, and 1981-1986 respectively) and by all three treatment categories: IT M T X only (OR: 1.3, 95% CI: 1.09-1.78), C R T only (OR; 7.2, 95% CI: 6.14-8.39), and C R T plus IT M T X (OR: 2.6, 95% CI: 2.30-2.95). The risk of using special education also increased significantly as the dose of C R T increased. Survivors, compared with siblings, received special education at an increased rate due to school missed; most notably, survivors of bone tumours (OR: 7.4, 95% CI: 3.95-13.92) and soft tissue 13 sarcoma (OR: 6.2, 95% CI: 3.34-11.33). Those treated with C R T alone or C R T plus IT M T X most often reported low test scores and problems learning and concentrating as the reasons for receiving special education. Those diagnosed with leukemia, C N S tumours, non-Hodgkin's lymphoma, kidney cancer, and neuroblastoma were mostly likely to report the reason for special education as low test scores (53.4%, 50.8%, 40.0%, 48.5%, and 56.5% respectively). Wi th respect to the duration of special education services rendered, those who received high-dose C R T required services for a significantly longer time (mean of 5.7 years) than survivors who never had C R T (mean of 4.7 years). Survivors of leukemia, C N S tumours, non-Hodgkin's lymphoma, and neuroblastoma were significantly less likely than siblings to complete high school; whereas there was no significant difference between the other diagnosis groups and siblings. A similar pattern was identified regarding college completion. No significant results were found for treatment modality and high school completion, but survivors who participated in special education and received C R T were 1.9 times more likely not to complete college than siblings. Lastly, results suggested that children diagnosed with neuroblastoma were at risk for educational deficits, a finding that had not been previously documented, but has since been supported by the Canadian study (Barrera, Shaw, Speechley, Maunsell, & Pogany, 2005). 1.5.4 Danish Study In Denmark 2384 children who had been diagnosed with cancer before the age of 20 between 1960 and 1996 were identified from the Danish Cancer Register (Koch, Kejs, Engholm, Johansen, & Schmiegelow, 2004). Survivor participants were born within 1960 to 1980 to allow adequate follow-up time, and were at least 13 years of age at the start of follow-up. A random sample of 53 143 age and gender matched controls were selected from the Register of Population Statistics. Information pertaining to educational attainment was collected from education records 14 obtained from Statistics Denmark. It was discovered that overall, similar proportions of survivors of childhood cancer and controls were reaching some level of higher education (17% vs. 18%). However, while male survivors attained levels of higher education equally well as, i f not slightly better than male controls (RR: 1.13, 95% CI: 0.97-1.32), female survivors had a reduced chance (RR: 0.75, 95% CI: 0.62-0.90). Survivors of non-CNS tumours attained similar higher education levels overall compared to controls (34% vs. 34% for males and 18% vs. 21% for females). On the other hand, survivors of C N S tumours had reduced chances o f attaining education at most levels compared with controls (e.g., attaining higher education overall, R R : 0.77, 95% CI: 0.55-1.07 for males and R R : 0.55, CI: 0.37-0.82 for females). Overall, a significant trend was found for attaining youth education (upper secondary school) with increasing age at diagnosis (non-C N S tumours, R R per year: 1.02, 95% CI: 1.01-1.04; C N S tumours, R R per year: 1.06, 95% CI: 1.03-1.10), but not for higher education. Educational outcomes did not appear to be influenced by treatment era or time since diagnosis. Survivors of C N S tumours were more likely to finish vocational training i f their parents had higher education than lower education levels (RR: 2.12, 95% CI: 1.36-3.31), yet male survivors of C N S tumours whose parents had higher education were less likely to attain higher education (RR: 0.37, 95% CI: 0.18-0.78). 7.5.5 Finnish Studies Finnish population-based studies investigated 300 individuals with brain tumours (Lahteenmaki et al., 2007) and 371 individuals diagnosed with leukemia (Harila-Saari et al., 2007) and compared them with population controls matched by age, gender and region of residence. These are the only studies to have utilized record linkage and data of standardized reporting to investigate scholastic achievement. Both studies identified patients born between 1974 and 1986 who were diagnosed before 16 years of age and alive on their 16 t h birthdays. The 15 age 16 was used as this is the common age of individuals in grade 9, the year for which data was collected. Demographic and clinical data were collected from the Finnish Cancer Registry and scholastic achievement data based upon grade 9 (which is the last year o f comprehensive schooling) school reports collected since 1990 by Statistics Finland. National annual testing of core grade 9 school subjects provides uniform grading. Findings indicated that those with brain tumours had lower overall averages and specific course averages than controls irrespective of treatment, but that females treated with cranial radiation were most at risk for poor scholastic achievement (Lahteenmaki et al., 2007). Individuals diagnosed with leukemia and treated with cranial radiation had both lower overall and lower course averages; of those treated with chemotherapy, only females treated before 7 years of age were at risk for lower school marks than their controls (Harila-Saari et al., 2007). Interestingly, both studies noted greatest discrepancies in marks for foreign language course; both studies suggest this represents impairment in verbal performance. 1.6 Methodological Shortcomings of Previous Research Findings regarding long-term educational outcomes among survivors of childhood cancer have not been consistent (Dolgin, Somer, Buchvald, & Zaizov, 1999; Haupt et al., 1994; Hays et al., 1992); this lack of consensus maybe due to methodological shortcomings (Moleski, 2000). There exists a general lack of consideration of confounding variables that may affect school performance; specifically, SES (Bader-Meunier, Tchernia, & Dommergues, 1996) and physical health or physical impairments (F. Daniel Armstrong & Horn, 1995). A s well , studies have been criticized for employing inappropriate comparison'groups or no comparison group (Madan-Swain & Brown, 1991; Moleski , 2000) and for the use of small sample sizes (Patenaude & Kupst, 2005). Moreover, objective outcome measures are necessary to investigate educational 16 achievement (Brown & Madan-Swain, 1993). Recent studies have commonly employed self-reported or proxy-reported data in exploring educational outcomes among survivors (Barrera, Shaw, Speechley, Maunsell, & Pogany, 2005; Langeveld et al., 2003; Mi tby et al., 2003). It is crucial that any possible areas of academic deficit be identified that may obstruct school success which may be addressed by early identification and appropriate educational intervention (Peckham, 1991). 1.7 Gaps wi th in the Literature Moreover, while researchers have considered the school experiences of survivors of childhood cancer with respect to special education utilization, level of education attained, grade retention, and school achievement based on anecdotal reports or academic achievement tests, namely the W R A T , rarely has the actual educational achievement within school or school performance been examined. Only two studies (Harila-Saari et al., 2007; Lahteenmaki et al., 2007) to date employ school achievement data of standardized reporting and these studies investigated only two specific cancer diagnoses: leukemia and brain tumours. A s well , published work has often focused on IQ rather than actual learning difficulties, yet it is these learning difficulties that have been reported to cause the most angst for teachers and parents (Eiser, 1998) and likely the survivors themselves. Furthermore, numerous studies have considered the immediate or acute effects of surviving childhood cancer on education (Brown et al., 1992; Copeland, Dowel l , Fletcher, Sullivan et al., 1988; Eiser, 1980; Precourt et al., 2002), but there exists a paucity of research concerning the educational late effects (Eiser, 1998). "Although a growing number of studies have documented the considerable impact of cancer diagnosis and treatment in quality of life in short-term survivors, less attention has focused on quality of life in long-term young adult survivors, partly because the rise in survival rates is relatively 17 recent"(Langeveld et al., 2002, p. 580). Awareness and understanding of the distinct learning needs of survivors of childhood cancer, as an at risk population, must be addressed and raised among educators. In doing so, early identification of learning difficulties can be made so as to effectively plan special education interventions (Robison, 2005) and preventions that w i l l improve the educational experiences and quality o f life for survivors of childhood cancer. 1.8 Objectives and Hypotheses This research project endeavours to explore the educational outcomes, specifically academic achievement and special education utilization, of survivors of childhood cancer and draw comparisons with the general population. A s well , disease-related effects and other possible modifiers including demographic, socioeconomic, and school system variables w i l l be investigated. Specifically, this project intends to: 1. Describe the level of educational achievement in a cohort of 5-year survivors of childhood cancer compared to a control group representative of the general population. Hypothesis: Survivors of childhood cancer w i l l have poorer educational achievement than population controls. 2. Describe the use of special education services among the survivor cohort compared to a control group representative of the general population. Hypothesis: Survivors of childhood cancer w i l l have increased special education utilization compared to population controls. 3. Examine the relationship between potentially modifying risk factors and educational outcomes (including achievement and special education) among survivors. 18 Hypothesis: Survivors of leukemia and C N S tumours, female gender, those treated with radiation or cranial radiation, and those younger in age at diagnosis w i l l be at increased risk for poor educational outcomes compared to other survivors. 19 1.9 References Allen , A . , Malpas, J. S., & Kingston, J. E . (1990). Educational achievements o f survivors of childhood cancer. Pediatr Hematol Oncol, 7(4), 339-345. Anderson, D . M . , Rennie, K . M . , Ziegler, R. S., Neglia, J. P., Robison, L . R. , & Gurney, J. G . (2001). Medical and neurocognitive late effects among survivors of childhood central nervous system tumors. Cancer, 92(10), 2709-2719. Anderson, V . , Godber, T., Smibert, E . , & Ekert, H . (1997). Neurobehavioural sequelae following cranial irradiation and chemotherapy in children: an analysis of risk factors. Pediatr Rehabil, 1(2), 63-76. Anderson, V . , Smibert, E . , Ekert, H . , & Godber, T. (1994). Intellectual, educational, and > behavioural sequelae after cranial irradiation and chemotherapy. Arch Dis Child, 70(6), 476-483. Anderson, V . A . , Godber, T., Smibert, E . , Weiskop, S., & Ekert, H . (2000). Cognitive and academic outcome following cranial irradiation and chemotherapy in children: a longitudinal study. Br J Cancer, 82(2), 255-262. Armstrong, C. L . , Gyato, K . , Awadalla, A . W. , Lustig, R., & Tochner, Z . A . (2004). A critical review of the clinical effects of therapeutic irradiation damage to the brain: the roots of controversy. Neuropsychol Rev, 14(1), 65-86. Armstrong, F. D . , Blumberg, M . J., & Toledano, S. R. (1999). Neurobehavioral issues in childhood cancer. School Psychology Review, 28(2), 194. Armstrong, F. D . , & Horn, M . (1995). Educational issues in childhood cancer. School Psychology Quarterly, 10(A), 292-304. Armstrong, F. D . , & Mulhern, R. K . (1999). Acute lymphoblastic leukemia and brain tumors. In R. T. Brown (Ed.), Cognitive Aspects of Chronic Illness in Children (pp. 1-14). New York: The Guilford Press. Bader-Meunier, B . , Tchernia, G. , & Dommergues, J. P. (1996). [Neuro-cognitive sequelae during acute lymphoblastic leukemia in children]. Arch Pediatr, 3(8), 745-748. Barrera, M . , Shaw, A . K . , Speechley, K . N . , Maunsell, E . , & Pogany, L . (2005). Educational and social late effects of childhood cancer and related clinical, personal, and familial characteristics. Cancer, 104(8), 1751-1760. B C Cancer Agency. (2007). Childhood Incidence 1995-2004. Retrieved June 12 2007, from http://wvvw.bccancer.bc.ca/HPI/CancerStatistics/FF/childhood.htm Bleyer, W . A . , Fallavollita, J., Robison, L . , Balsom, W. , Meadows, A . , Heyn, R., et al. (1990). Influence of age, sex, and concurrent intrathecal methotrexate therapy on intellectual function after cranial irradiation during childhood: a report from the Children's Cancer Study Group. Pediatr Hematol Oncol, 7(A), 329-338. Brown, R. T., & Madan-Swain, A . (1993). Cognitive, neuropsychological, and academic sequelae in children with leukemia. J Learn Disabil, 26(2), 74-90. Brown, R. T., Madan-Swain, A . , Pais, R., Lambert, R. G. , Baldwin, K . , Casey, R., et al. (1992). Cognitive status of children treated with central nervous system prophylactic chemotherapy for acute lymphocytic leukemia. Arch Clin Neuropsychol, 7(6), 481-497. Brown, R. T., Madan-Swain, A . , Walco, G . A . , Cherrick, I., levers, C. E . , Conte, P. M . , et al. (1998). Cognitive and academic late effects among children previously treated for acute 20 lymphocytic leukemia receiving chemotherapy as C N S prophylaxis. JPediatr Psychol, 23(5), 333-340. Buizer, A . I., de Sonneville, L . M . J., van den Heuvel-Eibrink, M . M . , & Veerman, A . J. P. (2006). Behavioral and educational limitations after chemotherapy for childhood acute lymphoblastic leukemia or Wilms tumor. Cancer, 106(9), 2067-2075. Butler, R. W. , H i l l , J. M . , Steinherz, P. G . , Meyers, P. A . , & Finlay, J. L . (1994). Neuropsychologic effects of cranial irradiation, intrathecal methotrexate, and systemic methotrexate in childhood cancer. J Clin Oncol, 12(12), 2621-2629. Canadian Cancer Society/National Cancer Institute of Canada. (2007). Canadian Cancer Statistics 2007. Toronto, Canada. Christie, D . , Leiper, A . D . , Chessells, J. M . , & Vargha-Khadem, F. (1995). Intellectual performance after presymptomatic cranial radiotherapy for leukaemia: effects of age and sex. Arch Dis Child, 73(2), 136-140. Coniglio, S. J., & Blackman, J. A . (1995). Developmental outcome of childhood leukemia. Topics in Early Childhood Special Education, 15(1), 19. Copeland, D . R., Dowell , R. E . , Jr., Fletcher, J. M . , Bordeaux, J. D . , Sullivan, M . P., Jaffe, N . , et al. (1988). Neuropsychological effects of childhood cancer treatment. J Child Neurol, 3(1), 53-62. Copeland, D . R., Dowell , R. E . , Jr., Fletcher, J. M . , Sullivan, M . P., Jaffe, N . , Cangir, A . , et al. (1988). Neuropsychological test performance of pediatric cancer patients at diagnosis and one year later. J Pediatr Psychol, 13(2), 183-196. Copeland, D . R., Fletcher, J. M . , Pfefferbaum-Levine, B . , Jaffe, N . , Ried, H . , & Maor, M . (1985). Neuropsychological sequelae of childhood cancer in long-term survivors. Pediatrics, 75(4), 745-753. Cousens, P. (1997). Specific learning problems among children treated for acute lymphoblastic leukemia. International Journal of Pediatric Hematology and Oncology, 4(4), 353-362. Dolgin, M . J., Somer, E . , Buchvald, E . , & Zaizov, R. (1999). Quality o f life in adult survivors of childhood cancer. Soc Work Health Care, 28(4), 31-43. Duffner, P. K . (2004). Long-term effects of radiation therapy on cognitive and endocrine function in children with leukemia and brain tumors. Neurologist, 10(6), 293-310. Eiser, C. (1980). How leukaemia affects a child's schooling. Br J Soc Clin Psychol, i ° ( P t 4), 365-368. Eiser, C. (1991). Cognitive deficits in children treated for leukaemia. Arch Dis Child, 66(1), 164-168. Eiser, C. (1998). Practitioner review: long-term consequences o f childhood cancer. J Child Psychol Psychiatry, 39(5), 621-633. Evans, S. E . , & Radford, M . (1995). Current lifestyle of young adults treated for cancer in childhood. Arch Dis Child, 72(5), 423-426. Fogarty, K . , Volonino, V . , Caul, J., Rongey, J., Whitman, B . , O'Connor, D . , et al. (1988). Acute leukemia. Learning disabilities following C N S irradiation. Clin Pediatr (Phila), 27(11), 524-528. Gamis, A . S., & Nesbit, M . E . (1991). Neuropsychologic (cognitive) disabilities in long-term survivors of childhood cancer. Pediatrician, 18(1), 11-19. Halberg, F. E . , Kramer, J. H . , Moore, I. M . , Wara, W . M . , Matthay, K . K . , & Ab l in , A . R. (1992). Prophylactic cranial irradiation dose effects on late cognitive function in children treated for acute lymphoblastic leukemia. Int JRadiat Oncol Biol Phys, 22(1), 13-16. 21 Harila-Saari, A . H . , Lahteenmaki, P. M . , Pukkala, E . , Kyyronen, P., Lanning, M . , & Sankila, R. (2007). Scholastic Achievements of Childhood Leukemia Patients: A Nationwide, Register-Based Study. J Clin Oncol, 25(23), 3518-3524. Haupt, R., Fears, T. R., Robison, L . L . , M i l l s , J. L . , Nicholson, H . S., Zeltzer, L . K . , et al. (1994). Educational attainment in long-term survivors of childhood acute lymphoblastic leukemia. Jama, 272(18), 1427-1432. Hays, D . M . , Landsverk, J., Sallan, S. E . , Hewett, K . D . , Patenaude, A . F. , Schoonover, D . , et al. (1992). Educational, occupational, and insurance status of childhood cancer survivors in their fourth and fifth decades of life. Journal Of Clinical Oncology: Official Journal Of The American Society Of Clinical Oncology, 10(9), 1397-1406. H i l l , D . E . , Ciesielski, K . T., Sethre-Hofstad, L . , Duncan, M . H . , & Lorenzi, M . (1997). Visual and verbal short-term memory deficits in childhood leukemia survivors after intrathecal chemotherapy. J Pediatr Psychol, 22(6), 861-870. H i l l , J. M . , Kornblith, A . B . , Jones, D . , Freeman, A . , Holland, J. F. , Glicksman, A . S., et al. (1998). A comparative study of the long term psychosocial functioning of childhood acute lymphoblastic leukemia survivors treated by intrathecal methotrexate with or without cranial radiation. Cancer, 82(1), 208-218. Humpl, T., Fritsche, M . , Bartels, U . , & Gutjahr, P. (2001). Survivors of childhood cancer for more than twenty years. Acta Oncol, 40(1), 44-49. Jannoun, L . , & Chessells, J. M . (1987). Long-term psychological effects of childhood leukemia and its treatment. Pediatr Hematol Oncol, 4(4), 293-308. Jenkin, D . , Danjoux, C , & Greenberg, M . (1998). Subsequent quality of life for children irradiated for a brain tumor before age four years. Med Pediatr Oncol, 31(6), 506-511. Kaemingk, K . L . , Carey, M . E . , Moore, I. M . , Herzer, M . , & Hutter, J. J. (2004). Math Weaknesses in Survivors of Acute Lymphoblastic Leukemia Compared to Healthy Children. Child Neuropsychology, 10(1), 14-23. Kazak, A . E . , Christakis, D . , Alderfer, M . , & Coiro, M . J. (1994). Young adolescent cancer survivors and their parents: Adjustment, learning problems, and gender. Journal of Family Psychology, 5(1), 74-84. Kelaghan, J., Myers, M . H . , Mulv ih i l l , J. J., Byrne, J., Connelly, R. R., Austin, D . F. , et al. (1988). Educational achievement of long-term survivors of childhood and adolescent cancer. Med Pediatr Oncol, 16(5), 320-326. Kingma, A . , Mooyaart, E . L . , Kamps, W . A . , Nieuwenhuizen, P., & Wilmink, J. T. (1993). Magnetic resonance imaging of the brain and neuropsychological evaluation in children treated for acute lymphoblastic leukemia at a young age. Am J Pediatr Hematol Oncol, 15(2), 231-238. Kingma, A . , Rammeloo, L . A . , van Der Does-van den Berg, A . , Rekers-Mombarg, L . , & Postma, A . (2000). Academic career after treatment for acute lymphoblastic leukaemia. Arch Dis Child, 82(5), 353-357. Kingma, A . , van Dommelen, R. I., Mooyaart, E . L . , Wilmink, J. T., Deelman, B . G . , & Kamps, W . A . (2001). Slight cognitive impairment and magnetic resonance imaging abnormalities but normal school levels in children treated for acute lymphoblastic leukemia with chemotherapy only. J Pediatr, 139(3), 413-420. Koch , S. V . , Kejs, A . M . T., Engholm, G. , Johansen, C , & Schmiegelow, K . (2004). Educational attainment among survivors of childhood cancer: a population-based cohort study in Denmark. British Journal of Cancer, 91(5), 923-928. 22 Kolotas, C , Daniel, M . , Demetriou, L . , Martin, T., Kurek, R., Tonus, C , et al. (2001). Long-term effects on the intelligence of children treated for acute lymphoblastic leukemia. Cancer Invest, 19(6), 581-587. Lahteenmaki, P. M . , Harila-Saari, A . , Pukkala, E . I., Kyyronen, P., Salmi, T. T., & Sankila, R. (2007). Scholastic achievements of children with brain tumors at the end of comprehensive education: A nationwide, register-based study. Neurology, 69(3), 296-305. Langeveld, N . E . , Stam, H . , Grootenhuis, M . A . , & Last, B . F. (2002). Quality of life in young adult survivors of childhood cancer. Supportive Care In Cancer: Official Journal Of The Multinational Association Of Supportive Care In Cancer, 10(8), 579-600. Langeveld, N . E . , Ubbink, M . C , Last, B . F. , Grootenhuis, M . A . , Voute, P. A . , & De Haan, R. J. (2003). Educational achievement, employment and l iving situation in long-term young adult survivors of childhood cancer in the Netherlands. Psychooncology, 12(3), 213-225. L i , F. P., Winston, K . R., & Gimbrere, K . (1984). Follow-up of children with brain tumors. Cancer, 54(\), 135-138. Madan-Swain, A . , & Brown, R. T. (1991). Cognitive and psychosocial sequelae for children with acute lymphocytic leukemia and their families. Clinical Psychology Review, 11(3), 267-294. Mitby, P. A . , Robison, L . L . , Whitton, J. A . , Zevon, M . A . , Gibbs, I. C , Tersak, J. M . , et al. (2003). Utilization of special education services and educational attainment among long-term survivors of childhood cancer: a report from the Childhood Cancer Survivor Study. Cancer, 97(A), 1115-1126. Moehle, K . A . , Berg, R. A . , Ch'ien, L . T., & Lancaster, W . (1983). Language-related skills in children with acute lymphocytic leukemia. JDev Behav Pediatr, 4(4), 257-261. Moleski , M . (2000). Neuropsychological, Neuroanatomical, and Neurophysiological Consequences of C N S Chemotherapy for Acute Lymphoblastic Leukemia. Archives of Clinical Neuropsychology, 15(1), 603-630. Moore, B . D . , 3rd. (2005). Neurocognitive outcomes in survivors of childhood cancer. J Pediatr Psychol, 30(1), 51-63. Moore, I. M . , Glasser, M . E . , & Ab l in , A . R. (1988). The late psychosocial consequences of childhood cancer. J Pediatr Nurs, 3(3), 150-158. Moore, I. M . , Kramer, J. H . , Wara, W. , Halberg, F. , & Ab l in , A . R. (1991). Cognitive function in children with leukemia. Effect of radiation dose and time since irradiation. Cancer, 68(9), 1913-1917. Mulhern, R. K . , Fairclough, D . , & Ochs, J. (1991). A prospective comparison of neuropsychologic performance of children surviving leukemia who received 18-Gy, 24-Gy, or no cranial irradiation. J Clin Oncol, 9(8), 1348-1356. Mulhern, R. K . , & Palmer, S. L . (2003). Neurocognitive late effects in pediatric cancer. Curr Probl Cancer, 27(4), 177-197. Mulhern, R. K . , Wasserman, A . L . , Friedman, A . G. , & Fairclough, D . (1989). Social competence and behavioral adjustment of children who are long-term survivors of cancer. Pediatrics, 83(1), 18-25. Nagarajan, R., Neglia, J. P., Clohisy, D . R., Yasui , Y . , Greenberg, M . , Hudson, M . , et al. (2003). Education, employment, insurance, and marital status among 694 survivors of pediatric lower extremity bone tumors: a report from the childhood cancer survivor study. Cancer, 97(10), 2554-2564. 23 National Cancer Pol icy Board (Ed.). (2003). Childhood Cancer Survivorship: Improving Care and Quality of Life. Washington, D C : National Academic Press. Ochs, J., Mulhern, R., Fairclough, D . , Parvey, L . , Whitaker, J., Ch'ien, L . , et al. (1991). Comparison of neuropsychologic functioning and clinical indicators of neurotoxicity in long-term survivors of childhood leukemia given cranial radiation or parenteral methotrexate: a prospective study. J Clin Oncol, 9(1),, 145-151. Patenaude, A . F. , & Kupst, M . J. (2005). Psychosocial functioning in pediatric cancer. J Pediatr Psychol, 30(1), 9-27. Peckham, V . C. (1991). Educational deficits in survivors of childhood cancer. Pediatrician, 18(1), 25-31. Peckham, V . C , Meadows, A . T., Bartel, N . , & Marrero, O. (1988). Educational late effects in long-term survivors of childhood acute lymphocytic leukemia. Pediatrics, 81(1), 127-133. Precourt, S., Robaey, P., Lamothe, I., Lassonde, M . , Sauerwein, H . C , & Moghrabi, A . (2002). Verbal cognitive functioning and learning in girls treated for acute lymphoblastic leukemia by chemotherapy with or without cranial irradiation. Dev Neuropsychol, 21(2), 173-195. Raymond-Speden, E . , Tripp, G. , Lawrence, B . , & Holdaway, D . (2000). Intellectual, neuropsychological, and academic functioning in long-term survivors of leukemia. J Pediatr Psychol, 25(2), 59-68. Ries, L . A . G . , Melbert, D . , Krapcho, M . , Mariotto, A . , Mi l le r , B . A . , Feuer, E . J., et al. (Eds.). (2007). SEER Cancer Statistics Review, 1975-2004. Bethesda, M D : National Cancer Institute. Robaey, P., Dobkin, P., Leclerc, J . - M . , Cyr, F. , Sauerwein, C , & Theoret, Y . (2000). A comprehensive model of the development of mental handicap in children treated for acute lymphoblastic leukaemia: A synthesis of the literature. International Journal of Behavioral Development, 24(\), 44-58. Robison, L . L . (2005). The Childhood Cancer Survivor Study: a resource for research of long-term outcomes among adult survivors of childhood cancer. Minn Med, 88(4), 45-49. Robison, L . L . , Green, D . M . , Hudson, M . , Meadows, A . T., Mertens, A . C , Packer, R. J., et al. (2005). Long-term outcomes of adult survivors of childhood cancer. Cancer, 104(11 Suppl), 2557-2564. Rubenstein, C. L . , Varni , J. W. , & Katz, E . R. (1990). Cognitive functioning in long-term survivors of childhood leukemia: a prospective analysis. J Dev Behav Pediatr, 11(6), 301-305. Schwartz, C. L . (1999). Long-term survivors of childhood cancer: the late effects of therapy. Oncologist, 4(1), 45-54. Seaver, E . , Geyer, R., Sulzbacher, S., Warner, M . , Batzel, L . , Milstein, J., et al. (1994). Psychosocial adjustment in long-term survivors o f childhood medulloblastoma and ependymoma treated with craniospinal irradiation. Pediatr Neurosurg, 20(4), 248-253. Smibert, E . , Anderson, V . , Godber, T., & Ekert, H . (1996). Risk factors for intellectual and educational sequelae of cranial irradiation in childhood acute lymphoblastic leukaemia. BrJ Cancer, 73(6), 825-830. Smith, M . , & Hare, M . L . (2004). A n overview of progress in childhood cancer survival. J Pediatr Oncol Nurs, 21(3), 160-164. 24 Smith, M . A . , Ries, L . A . G . , Gurney, J. G . , & Ross, J. A . (1999). Leukemia. In L . A . G . Ries, M . A . Smith, J. G . Gurney, M . Linet, T. Tamra, J. L . Young & G . R. Bunin (Eds.), Cancer Incidence and Survival among Children and Adolescents: United States SEER Program 1975-1995 (pp. 17-34). Bethesda, M D : National Cancer Institute, S E E R Program. Stehbens, J. A . , Kaleita, T. A . , N o l l , R. B . , MacLean, W . E . , Jr., O'Brien, R. T., Waskerwitz, M . J., et al. (1991). C N S prophylaxis of childhood leukemia: what are the long-term neurological, neuropsychological, and behavioral effects? Neuropsychol Rev, 2(2), 147-177. Syndikus, I., Tait, D . , Ashley, S., & Jannoun, L . (1994). Long-term follow-up of young children with brain tumors after irradiation. Int JRadiat Oncol Biol Phys, 30(4), 781-787. Taylor, A . , Hawkins, M . , Griffiths, A . , Davies, H . , Douglas, C , Jenney, M . , et al. (2004). Long-term follow-up of survivors of childhood cancer in the U K . Pediatr Blood Cancer, 42(2), 161-168. von der Weid, N . (2001). Late effects in long-term survivors of A L L in childhood: experiences from the S P O G late effects study. Swiss Med Wkly, 737(13-14), 180-187. Waber, D . P., Bernstein, J. H . , Kammerer, B . L . , & Tarbell, N . J. (1992). Neuropsychological diagnostic profiles of children who received C N S treatment for acute lymphoblastic leukemia: The systemic approach to assessment. Developmental Neuropsychology, 8(1), 1-28. Waber, D . P., Urion, D . K . , Tarbell, N . J., Niemeyer, C , Gelber, R. , & Sallan, S. E . (1990). Late effects of central nervous system treatment of acute lymphoblastic leukemia in childhood are sex-dependent. Dev Med Child Neurol, 32(3), 238-248. Whitt, J. K . , Wells, R. J., Lauria, M . M . , Wilhelm, C. L . , & M c M i l l a n , C. W . (1984). Cranial radiation in childhood acute lymphocytic leukemia. Neuropsychologic sequelae. Am J Dis Child, 138(8), 730-736. 25 C H A P T E R 2: E D U C A T I O N A L L A T E E F F E C T S A M O N G S U R V I V O R S O F C H I L D H O O D C A N C E R IN B R I T I S H C O L U M B I A 1 2.1 Background Advances in medical treatment have been made within the past 20 years that have resulted in dramatically increased survival rates for children diagnosed with cancer (Smith & Hare, 2004). The five-year survival rate of childhood cancer has increased substantially since 1970 (Canadian Cancer Society/National Cancer Institute of Canada, 2007), recently reaching almost 80% (Ries et al., 2007). This has led to a growing population of children within our education systems that did not previously exist, and of whom relatively little is known regarding long term educational achievement. Within the United States, survivors of childhood cancer outnumber traditional special education populations such as those with hearing impairments, deafness, visual impairments, and severe intellectual disabilities (Peckham, 1991). Only two studies (Harila-Saari et al., 2007; Lahteenmaki et al., 2007), recently published, have investigated educational achievement using standardized data o f school marks. Although there is a paucity of literature regarding objective educational achievements of survivors of childhood cancer, educational difficulties have been reported by survivors, parents and teachers (Dolgin, Somer, Buchvald, & Zaizov, 1999; Jannoun & Chessells, 1987; Raymond-Speden, Tripp, Lawrence, & Holdaway, 2000). A s well , it has been found that survivors of childhood cancer attain lower levels of education (Koch, Kejs, Engholm, Johansen, & Schmiegelow, 2004; Link et al., 2006; Nagarajan et al., 2003; Zebrack et al., 2007), have an increased use of special education services compared to both peers and siblings (Barrera, Shaw, Speechley, Maunsell, & Pogany, 2005; Kingma, Rammeloo, van Der Does-van den Berg, Rekers-Mombarg, & Postma, 1 A version of this chapter will be submitted for publication. 26 2000; Mi tby et al., 2003; Robison et al., 2005), and have a greater likelihood of grade repetition (Barrera, Shaw, Speechley, Maunsell, & Pogany, 2005; Buizer, de Sonneville, van den Heuvel-Eibrink, & Veerman, 2006). There is no lack of evidence within the literature of adverse neurocognitive late effects (Campbell et a l , 2007; Dickerman, 2007; Mulhern & Palmer, 2003; National Cancer Policy Board, 2003) or long-term neuropsychological deficits (Link et al., 2006) among survivors of childhood cancer which may inhibit educational performance; specifically, lower intelligence quotient (IQ) scores compared to normative or sibling data (Christie, Leiper, Chessells, & Vargha-Khadem, 1995; Moleski , 2000). Several clinical factors are associated with an increased risk of adverse educational outcomes. These include younger age at diagnosis and/or treatment (V. Anderson, Godber, Smibert, & Ekert, 1997; Jannoun & Chessells, 1987; Kingma, Rammeloo, van Der Does-van den Berg, Rekers-Mombarg, & Postma, 2000; Seaver et al., 1994), treatment involving cranial or craniospinal radiation therapy (CRT) and/or intrathecal methotrexate (IT M T X ) (V. Anderson, Godber, Smibert, & Ekert, 1997; V . Anderson, Godber, Smibert, Weiskop, & Ekert, 2000; Dolgin, Somer, Buchvald, & Zaizov, 1999; H i l l et al., 1998; Kingma et al., 2001; Mi tby et al., 2003; Smibert, Anderson, Godber, & Ekert, 1996), higher therapeutic doses of C R T or IT M T X (V. Anderson, Godber, Smibert, & Ekert, 1997; Buizer, de Sonneville, van den Heuvel-Eibrink, & Veerman, 2006; Halberg et al., 1992; Kingma, Mooyaart, Kamps, Nieuwenhuizen, & Wilmink, 1993; I. Moore, Kramer, Wara, Halberg, & Ab l in , 1991; Smibert, Anderson, Godber, & Ekert, 1996), and a diagnosis of leukemia (in particular, acute lymphoblastic leukemia: A L L ) , Hodgkin's lymphoma, central nervous system (CNS) tumours, or neuroblastoma (Barrera, Shaw, Speechley, Maunsell, & Pogany, 2005; Langeveld et al., 2003; Mi tby et al., 2003). Other demographic, socioeconomic and temporal variables have been associated with an increased risk 27 of poorer educational outcomes among survivors of childhood cancer such as female gender (Brown et al., 1998; Eiser, 1991; Haupt et al., 1994; Kelaghan et al., 1988; Langeveld et al., 2003; Mulhern, Fairclough, & Ochs, 1991; Waber, Bernstein, Kammerer, & Tarbell, 1992), lower socioeconomic status (SES)(Butler, H i l l , Steinherz, Meyers, & Finlay, 1994; Mulhern & Palmer, 2003; Whitt, Wells, Lauria, Wilhelm, & M c M i l l a n , 1984), and longer time interval since diagnosis (V. Anderson, Godber, Smibert, & Ekert, 1997; Moehle, Berg, Ch'ien, & Lancaster, 1983; Mulhern, Fairclough, & Ochs, 1991; Rubenstein, Varni , & Katz, 1990). Unfortunately, past research has lacked consensus regarding the long-term educational outcomes of survivors of childhood cancer (Dolgin, Somer, Buchvald, & Zaizov, 1999; Haupt et al., 1994). Inconsistencies within the literature may be due in part to methodological shortcomings (Moleski, 2000). The methodological shortcomings of prior research, as mentioned earlier, include a lack of control for potential confounding variables, small sample sizes, inappropriate or no control group(s), and subjective educational measures must be addressed. This study addresses the need for objective measures of long term school achievement among survivors of childhood cancer while considering the methodological limitations of previous research in this field. New data is provided on the overall educational achievement among survivors of childhood cancer and the disease-related impact on educational outcomes. These findings w i l l further the understanding o f how various clinical variables such as: diagnosis group, age at diagnosis, and treatment modality may be associated with long term educational performance. B y identifying those survivors who may be at risk for poorer educational outcomes, educational interventions can be developed, including preventative and rehabilitative programs, that w i l l improve the educational experiences and learning opportunities for the growing 28 population of children who have survived cancer. Although evidence exists that survivors of childhood cancer are at risk for learning problems, very little connection has been made, or work done, regarding possible intervention or rehabilitation opportunities for these children (Eiser, 2004). In addition, it is hoped that this study will elicit and inform future research that will more profoundly explore the educational achievements of these individuals and continue to address their unique and specific educational needs. 2.2 Mater ia ls and Methods The study cohort included all individuals with a primary diagnosis of cancer included in the International Classification of Childhood Cancer (ICCC) (Kramarova & Stiller, 1996) between 1975 and 1995 and resident in the province of British Columbia (BC) at the time of diagnosis, identified from the BC Cancer Registry. Individuals were considered survivors of childhood cancer i f they were diagnosed with a primary cancer before 15 years of age and had survived five years or more since diagnosis. Our original cohort included only those individuals who were identified within the BC Kindergarten to Grade 12 (secondary school graduation year) school system between the years 1990 and 2004 (including the 2004/2005 school year), but due to a lack of educational data (i.e., some key outcome measures) for the earlier years, we restricted our survivor cohort (and control group) to those found in the system between the 1995/1996 and 2004/2005 school year. Only data occurring post-survivorship (at least 5 years since diagnosis), with the inclusion of the school year in which survivorship was attained, was included. A comparison group was provided by the BC Ministry of Education (MOE). This cohort initially consisted of randomly selected age and gender frequency matched individuals enrolled within the BC school system between the years 1990 and 2004, but as stated above, was 29 eventually restricted to those in the system between 1995 and 2004. The B C M O E requires that records be kept of all students enrolled in the B C K-12 school system through Edudata Canada, a repository of M O E datasets for research use, based at the University of B C . Educational data included: a) standardized scores and percentages of Foundation Skills Assessments (FSAs) written in Grades 4, 7, and 10 in the subject areas of reading, writing and numeracy; b) Grade 12 provincial examination scores in English, Mathematics (Math), Biology, History, and Communications; and c) special education utilization, including particular special education designations such as: learning disability, gifted, deaf/blind, hearing impairment, visual impairment, and physical disability/chronic health impairment. F S A s are province-wide, standardized tests employed by B C ' s M O E to assess annually student performance with respect to basic academic skills. F S A s are scored on a 5-point scale defined as: 1) exceeds expectations, 2) borderline between meets and exceeds expectations, 3) meets expectations, 4) borderline between meets and does not meet expectations, and 5) does not meet expectations. Achievement on F S A s was investigated using a cut-point of meets expectations. F S A data was available for Grades 4 and 7 for the 1999/2000 to 2004/2005 school years and for Grade 10 from 1999/2000 to 2002/2003 school years. The Grade 12 provincial exams are also a standardized measure for which percent scores are given. A letter grade mark of C, which is equal to 60%, was employed as an achievement cut-point for the Grade 12 provincial exams; a mark of C is considered satisfactory performance with respect to standard learning objectives ( B C Ministry of Education, 2006). F S A participation was calculated as having F S A test data (i.e., possessing a scaled-score) and being in the appropriate grade for the corresponding F S A (i.e., in Grade 4 and wrote a Grade 4 F S A ) . For Grade 12 courses, participation was determined as those ever having a school mark (score assigned for the course by the school) or 30 provincial exam mark and had reached Grade 12 or graduated. Special education designations refer to the special education services/program the student requires for a disability of an intellectual, physical, sensory, emotional or behavioural nature, a learning disability or special gifts or talents (Policy Document: Special Education, 2006). F S A and Grade 12 provincial exam measures were considered educational achievement, while educational outcome included both educational achievement (i.e., F S A and Grade 12 provincial exams) and special education data. Only data occurring post-survivorship (at least 5 years since diagnosis), with the inclusion of the school year in which survivorship was attained, was included. Sociodemographic measures (neighborhood SES quintile and rural/urban status) were derived by Edudata who employed Statistics Canada postal code conversion software (Wilkins, 2002) which linked the individual's postal code to the appropriate census enumeration area. In cases where an individual had more than one postal code, the earliest available postal code (i.e., closest to the 5-year survivor date) was used. Clinical data for the survivor cohort was provided by chart abstraction from individual health records to supply specific details pertaining to the diagnosis and treatment. This data included: a) diagnosis group (classified according to the I C C C ) , b) age at diagnosis and c) treatment for primary cancer diagnoses. At the time of this study, treatment data was available for leukemia, lymphoma, and C N S cancer diagnoses and treatment modality. Treatment was categorized as: 1) ever having received chemotherapy, 2) ever having received radiation, and 3) ever having received C R T ; these categories are not mutually exclusive, thus it is possible for a survivor to have received more than one type o f treatment. Age at diagnosis was classified into three categories: <2 years, 2 to <5 years, and >5 years. 31 2.2.1 Statistical Analysis Demographic and socioeconomic characteristics of both the survivor and control cohorts and clinical characteristics of the survivors were investigated through crosstabulations and chi-square test. Participation rates of survivors as a whole and specific diagnosis groups compared with controls for the F S A s and Grade 12 courses were computed using independent-samples t-tests. Logistic regression analyses were employed to compare the educational outcomes of the survivor cohort with the control group and the relationship between potential modifiers (gender, diagnosis group, age at diagnosis, and treatment modality) and educational outcomes among the survivor cohort. Odds ratios (OR), adjusted odds ratios (OR adj), 95% confidence intervals (95%CI), andp values are reported. A l l statistical analyses were calculated using the Statistical Package for the Social Science (SPSS, Inc., 2001). 2.3 Results 2.3.1 Sociodemographic and Clinical Characteristics of Survivors and Controls Sociodemographic characteristics were, in general, comparable between survivors and controls (Table 1). More than 50% of both survivors and controls were male and both groups had reached similar levels of education within the K-12 system. A greater proportion of survivors than controls had physical disabilities (19.2% vs. 1.1%, pO.OOl ) . A s expected, leukemia was the most common diagnosis among survivors, followed by C N S tumours (Table 2). Survivors were a mean age of 4.6 years at diagnosis. Among those survivors where treatment data was available (diagnoses of leukemia, lymphoma, and C N S cancer) chemotherapy (75.8%) was the most common treatment received, followed by radiation (30.3%). 32 Table 2.1 Sociodemographic Characteristics of Survivors and Controls Survivors Controls Characteristic No. % No. % P value Gender Male 431 55.1 4529 54.0 Female 351 44.9 3857 46.0 Highest Grade Home School 2 0.3 16 0.2 Elementary 1 0.1 11 0.1 Kindergarten 0 0 19 0.2 Grade 1 2 0.3 32 0.4 Grade 2 2 0.3 31 0.4 Grade 3 3 0.4 51 0.6 Grade 4 11 1.4 117 1.4 Grade 5 19 2.4 197 2.3 Grade 6 25 3.2 283 3.4 Grade 7 25 3.2 248 3.0 Grade 8 66 8.4 635 7.6 Grade 9' 40 5.1 420 5.0 Grade 10 49 6.3 591 7.0 Grade 11 76 9.7 905 10.8 Grade 12 461 59.0 4830 57.6 Physical Disabili ty 3 Yes 150 19.2 94 1.1 O .001 N o 632 80.8 8263 98.9 SES Quintile 3 1 (lowest) 102 14.2 1358 17.8 2 154 21.5 1433 18.7 3 142 19.8 1569 20.5 4 156 21.8 1608 21.0 5 (highest) 162 22.6 1676 21.9 Residence Status3 Rural 126 17.5 1426 18.6 0.003 Small Community 116 16.1 1050 13.7 Large Community 129 17.9 1080 14.1 Metropolitan 348 48.4 4126 53.7 Data were missing from Edudata on physical disability for 29 controls. SES quintile was missing for 66 survivors and 742 controls, and urban/rural status for 63 survivors and 704 controls as SES and urban/rural status were derived from postal codes which in some cases were missing in Edudata and in other cases were not able to be linked by the Statistics Canada software. Table 2.2 Clinical Characteristics of Survivors Survivors (n=782) Characteristic No. % Diagnosis Leukemia Lymphoma C N S Tumours Neuroblastoma Other Age at Diagnosis <2yrs 2 - <5yrs >5 yrs Treatment3 Chemotherapy Radiation C R T CRT: cranial radiation therapy "Treatment data was only available for leukemia, lymphoma, and CNS tumours (n=476) and referred only to treatment for primary cancers; categories were not ^nutuall^xclu^ive^^^^^^^^^^^^^^^^^^^^^ 270 58 166 48 240 34.5 7.4 21.2 6.1 30.7 225 279 278 28.8 35.7 35.5 361 144 136 75.8 30.3 28.6 2.3.2 Educational Outcomes of Survivors and Controls In general, controls performed at higher levels than survivors on all F S A s ; specifically, controls were significantly more likely than survivors to meet or exceed expectations on Numeracy Grades 4 and 7, and Reading Grades 7 and 10. There were no significant differences in achievement between the controls and survivors on any o f the Grade 12 provincial examinations (Table 3). There were no significant differences in F S A participation rates between survivors and controls; survivor participation rates were at least 95.3%. A significantly higher percentage of controls than survivors were enrolled in English 12, Math 12, and History 12 (75.9% vs. 66.6%, p<0.001; 37.6% vs. 25.4%, p<0.001; and 20.0% vs. 16.1%, p=0.028 respectively). With respect to special education, survivors had significantly increased special education designations than controls. In particular, survivors were significantly more l ikely than controls to have a physical disability, visual disability, and/or hearing disability designation (Table 3). There were no significant differences regarding grade repetition (ever having repeated a school grade), learning disability or gifted designation between the two cohorts. Male and female survivors compared with male and female controls differed on some educational achievement measures and special education designations. Although male survivors were significantly less likely than male controls to meet or exceed expectations on F S A Numeracy Grades 4 and 7 and Reading Grade 7, female survivors were significantly less likely than female controls to meet or exceed expectations on F S A Numeracy Grades 7 and 10, Reading Grade 10, and Writing Grade 7, and to have a mark of at least 60% on the ' Communications 12 provincial exam. Regarding special education, both male and female survivors compared with their control counterpart were significantly more at risk to have special 35 education, physical disability, hearing disability and visual disability designations. In addition, female survivors were also significantly more likely to have a learning disability than female controls whereas male survivors were no more likely than male controls to have a learning disability (Table 4). There were significantly lass female controls with a learning disability than male controls (OR, 0.298, p<0.001); in contrast, there was no significant difference in the number with a learning disability designation between female and male survivors (OR, 0.563, p=0.149). Table 2.3 Educat ional Outcomes of Survivors and Controls Controls (n=8386) Survivors (n=782) Outcome No. % No. % ORjdi" 95% CI p value 5-Pt FSA meets expectations11 Numeracy Gr 4 836 68.2 72 53.3 0.51 0.4-0.8 0.001 Numeracy Gr 7 1255 66.1 107 53.8 0.76 0.4-0.8 0.001 Numeracy Gr 10 1024 52.6 89 46.8 0.79 0.6-1.1 Reading Gr 4 799 65.2 77 57.0 0.74 0.5-1.1 Reading Gr 7 1233 64.9 107 53.8 0.63 0.5-0.9 0.003 Reading Gr 10 1029 52.8 85 44.7 0.73 0.5-1.0 0.043 Writing Gr 4 1006 82.1 102 75.6 0.72 0.5-1.1 Writing Gr 7 1419 74.7 135 67.8 0.73 0.5-1.0 Writing Gr 10 1212 62.2 117 61.6 0.97 0.7-1.3 Gr 12 Provincial Exam C (60%) or abovec English 2825 77.1 239 77.9 1.12 0.8-1.5 Math 1296 71.3 77 65.8 0.81 0.5-1.3 History 695 71.9 56 75.7 1.20 0.7-2.1 Biology 929 61.1 85 61.6 0.97 0.7-1.4 Communications 472 74.7 57 69.5 0.87 0.5-1.5 Special education 1176 14.1 254 32.5 3.05 2.6-3.6 <0.001 Physical disability 94 1.1 150 19.2 21.47 16-28 <0.001 Visual disability 31 0.4 43 5.5 16.18 10-26 <0.001 Hearing disability 24 0.3 21 2.7 9.69 5.4-18 O.001 95% CI: 95% confidence interval; ORadj: adjusted OR No.: number of individuals who meet the outcome criteria. aAdjusting for: gender, urban/rural status, SES, hearing disability and visual disability; except special education, physical disability, hearing disability, and visual disability outcomes which were not adjusted for visual or hearing disability. bCompared with does not meet expectations and those who did not write. cCompared with a percent mark below a letter grade of C (60%) and those who did not write. 36 Table 2.4 Educat ional Outcomes of Males and Females Male Male Female Controls* Survivors Controls* Female (n=4529) (n=431) Survivors (n=3857) (n=351) Outcome No. % No. % p value No. % No. % ORadi' 3 p value 5-Point FSA score meets expectationsb Numeracy Grade 4 450 69.0 33 47.1 0.41 0.001 386 67.4 39 60.0 0.68 Numeracy Grade 7 675 66.7 60 54.5 0.60 0.016 580 65.4 47 52.8 0.55 0.013 Numeracy Grade 10 555 52.8 54 52.9 0.97 469 52.3 35 39.8 0.63 0.048 Reading Grade 4 413 63.3 35 50.0 0.64 386 67.4 42 64.6 0.84 Reading Grade 7 617 61.0 53 48.2 0.62 0.024 616 69.4 54 60.7 0.66 Reading Grade 10 494 47.0 47 46.1 0.93 535 59.6 38 43.2 0.56 0.013 Writing Grade 4 511 78.4 50 71.4 0.74 495 86.4 52 80.0 0.69 Writing Grade 7 695 68.7 70 63.6 0.85 724 81.6 65 73.0 0.57 0.034 Writing Grade 10 582 55.4 61 59.8 1.13 630 70.2 56 63.6 0.75 Gr 12 Provincial Exam C (60%) or abovec English 1277 71.6 115 70.6 0.94 1548 82.3 124 86.1 1.53 Math 672 68.7 41 62.1 0.72 624 74.3 36 70.6 0.97 History 355 72.6 37 77.1 1.29 340 71.1 19 73.1 1.07 Biology 311 58.5 34 59.6 0.96 618 62.5 35 63.0 0.98 Communications 317 73.2 38 73.1 1.25 155 77.9 19 63.3 0.41 0.045 Special education 715 15.8 147 34.1 2.89 O.001 461 12.0 107 30.5 3.32 O.001 Learning disability 179 4.0 20 4.6 1.22 48 1.2 10 2.8 2.42 0.013 Physical disability 57 1.3 81 18.8 18.69 <0.001 37 1.0 69 19.7 26.53 <0.001 Visual disability 17 0.4 21 4.9 14.20 O.001 14 0.4 22 6.3 19.13 O.001 Hearing disability 12 0.3 10 2.3 9.11 <0.001 12 0.3 11 3.1 10.35 <0.001 *Reference group OR: odds ratio; 95% CI: 95% confidence interval; OR a d j : adjusted OR No. Enrolled: number of individuals ever in the corresponding grade for the FSA's and ever having a school score or a provincial exam score in the corresponding course for the grade 12 outcomes No.: number of individuals who meet the outcome criteria "Adjusting for: gender, urban/rural status, SES, hearing disability and visual disability; except special education, physical disability, visual disability, and hearing disability outcomes which were not adjusted for hearing or visual disability bCompared with does not meet expectations and those who did not write. cCompared with a percent mark below a letter grade of C (60%) and those who did not write. 2.3.3 Educational Outcomes by Diagnosis Two diagnosis groups appeared most at risk for poor educational outcomes: C N S tumours and leukemia. Survivors of C N S tumours compared with controls were significantly less likely to meet or exceed expectations on all F S A s : Numeracy Grades 4, 7 and 10; Reading Grades 4, 7 and 10; and Writing Grades 4, 7 andlO and significantly more likely to have a Communications Grade 12 provincial exam mark below 60%. Survivors of C N S tumours had 37 significantly more special education, physical disability, visual disability, and hearing disability designations than controls. Survivors of leukemia were significantly less l ikely to meet or exceed expectations than controls on F S A Numeracy Grades 4 and 7 and Reading Grade 7, and had significantly increased special education, learning disability, physical disability and visual disability designations compared with controls. Once survivors of C N S tumours and leukemia were excluded from the analysis of survivors compared with controls, educational achievement measures (i.e., F S A s and Grade 12 provincial exams) no longer exhibited any significant differences between survivors and controls, while special education designations which were previously significant remained so (Table 5). A s well , with the exclusion of survivors of both C N S tumours and leukemia, only Math 12 continued to have a significantly higher enrolment rate among controls than survivors (37.6% vs. 29.9%, p=0.019) while all other grade 12 course enrolment rates remained insignificantly different between survivors and controls. Survivors of neuroblastoma compared with controls showed no significant differences on any educational achievement measure, but were significantly more likely to have special education (OR, 2.3, p=0.010) and physical disability (OR, 9.6, p<0.001) designations than controls. There were no significant differences between survivors o f lymphoma and controls on any educational achievement measures or special education designation. Other diagnosis groups were numerically too small to perform meaningful comparisons. 38 Table 2.5 Educat ional Outcomes by Diagnosis Survivors excluding CNS Controls* tumours and Leukemia (n=8386) (n=346) Leukemias (n=270) CNS Tumours (n=166) P P P Outcome No. % No. % OR a d i a value No. % ORadi" value No. % ORad," value 5-pt FSA meets expectations'5 Numeracy Gr 4 836 68.2 41 59.4 0.69 26 53.1 0.48 0.016 5 29.4 0.16 0.001 Numeracy Gr 7 1255 66.1 53 58.9 0.70 41 54.7 0.60 0.033 13 38.2 0.29 0.001 Numeracy Gr 10 1024 52.6 50 58.8 1.33 30 43.5 0.67 9 25.0 0.30 0.002 Reading Gr 4 799 65.2 45 65.2 1.14 27 55.1 0.64 5 29.4 0.20 0.003 Reading Gr 7 1233 64.9 54 60.0 0.79 40 53.3 0.61 0.042 13 38.2 0.34 0.003 Reading Gr 10 1029 52.8 42 49.4 0.94 32 46.4 0.74 11 30.6 0.37 0.008 Writing Gr 4 1006 82.1 53 76.8 0.79 40 81.6 1.00 9 52.9 0.20 0.002 Writing Gr 7 1419 74.7 65 72.2 0.87 52 69.3 0.76 18 52.9 0.39 0.010 Writing Gr 10 1212 62.2 54 63.5 1.14 47 68.1 1.25 16 44.4 0.41 0.012 Gr 12 Prov. Exam C (60%) or above' English 2825 77.1 115 79.3 1.33 83 76.9 0.97 41 75.9 1.04 Math 1296 71.3 38 63.3 0.72 28 71.8 0.96 11 61.1 0.90 History 695 71.9 25 80.6 1.83 26 76.5 1.21 5 55.6 0.46 Biology 929 61.1 38 61.3 1.08 32 61.5 0.98 15 62.5 0.69 Communications 472 74.7 25 73.5 1.05 20 83.3 1.81 12 50.0 0.38 0.037 Special education 1176 14.1 87 25.1 2.09 <0.001 89 33.0 3.05 <0.001 78 47.0 6.13 <0.001 Learning disability 227 2.7 12 3.5 1.25 15 5.6 2.11 0.007 3 1.8 0.70 Physical disability 94 1.1 48 13.9 14.43 <0.001 40 14.8 15.50 <0.001 62 37.3 57.65 O.001 Visual disability 31 0.4 22 6.4 19.06 <0.001 4 1.5 4.07 0.009 17 10.2 32.79 <0.001 Hearing disability 24 0.3 14 4.0 15.24 <0.001 0 0 - 7 4.2 16.05 <0.001 *Reference group 0Radj: adjusted OR No.: number of individuals who meet the outcome criteria. "Adjusting for: gender, urban/rural status, SES, hearing disability and visual disability; except special education, physical disability, visual disability, and hearing disability outcomes which were not adjusted for hearing or visual disability. bCompared with does not meet expectations and those who did not write. 'Compared with a percent mark below a letter grade of C (60%) and those who did not write. 2.3.4 Clinical Factors Related to Educational Outcomes among Survivors A n investigation of the treatment modality for the primary cancer indicated that survivors who received radiation were significantly less likely than survivors who received no radiation to meet expectations on F S A Numeracy Grades 7 and 10 and Writing Grade 7 and to have a Communications 12 provincial exam letter grade of at least C (60%). As/wel l , survivors who received radiation were more likely to have physical and visual disabilities than those survivors never having had radiation. The findings were similar among the survivors who received C R T compared to those who had no C R T . Survivors who ever received chemotherapy compared with survivors never receiving chemotherapy were significantly more l ikely to meet expectations on 39 F S A Writing Grade 10 and less likely to have a physical disability (Table 6). With respect to the age at diagnosis, survivors who were less than 2 years of age at diagnosis were significantly more likely to meet expectations on F S A Numeracy Grade 7 and Writing Grade 7, despite having significantly more visual and hearing disabilities than those who were 5 years o f age or older at diagnosis (Table 6). 40 Table 2.6 Clinical Factors Related to Selected Educational Outcomes among Survivors FSA Numeracy FSA Numeracy FSA Writing FSA Writing Communications Physical Visual Hearing Gr 7 meets Gr 10 meets Gr 7 meets Gr 10 meets GrUExamofC Disability Disability Disability expectations'1 expectations'* expectations'1 expectations'* (60%) or above0 Designation Designation Designation Factors ORadi" p value O R a d i a P value ORadi" p value ORadi" P value ORadi" P value ORadi" p value ORadi" p value ORadi" P value Treatment No Radiationd 1.00 Radiation No CRT1 0,26 1.00 0.027 0.33 0.040 0.29 0.035 0.68 0.01 0.011 2.29 0.001 3.79 0.006 -CRT 0.26 0.027 0.20 0.008 0.29 0.035 0.56 0.01 0.011 2.50 <0.001 3.52 0.009 -No Chemotherapy1 Chemotherapy 1.00 1.38 1.56 0.98 2.51 0.050 3.68 0.56 0.021 0.56 0.80 Age at Diagnosis <2 years'1 2 to <5 years 1.00 0.63 0.66 0.49 0.049 1.14 0.52 1.02 0.80 0.72 >5 years 0.30 0.013 0.85 0.35 0.033 0.80 0.97 0.85 0.28 0.008 0.17 0.023 ORadj: adjusted OR aAdjusting for: gender, urban/rural status, SES, hearing disability and visual disability; except special education, physical disability, visual disability, and hearing disability outcomes which were not adjusted for hearing or visual disability. bCompared with does not meet expectations and those who did not write. 'Compared with a percent mark below a letter grade of C (60%) and those who did not write. dReference group. 2.4 Conclusions This is the first Canadian, population-based cohort study to examine the educational late effects of survivors of childhood cancer using standardized data. Based upon data collected by the B C Ministry of Education on F S A and provincial exams, survivors were at increased risk for poor educational achievement compared to population controls, although this excess risk was generally confined to survivors of C N S tumours and leukemia. A s well , consistent with earlier research (Kingma, Rammeloo, van der Does-van den Berg, Rekers-Mombarg, & Postma, 2000; Langeveld et al., 2003; Mi tby et al., 2003; Robison et al., 2005), survivors were more likely than population controls to require special education services. In particular, survivors were at increased risk to have a physical disability, visual disability, and/or hearing disability. Unlike some studies (Buizer, de Sonneville, van den Heuvel-Eibrink, & Veerman, 2006; Haupt et al., 1994; I. Moore, Glasser, & Ab l in , 1988), survivors were no more l ikely than population controls to have repeated a grade in school. Consistent with the literature (Barrera, Shaw, Speechley, Maunsell , & Pogany, 2005; Haupt et al., 1994; Koch , Kejs, Engholm, Johansen, & Schmiegelow, 2004; Mi tby et al., 2003; Peckham, Meadows, Bartel, & Marrero, 1988) survivors of C N S tumours and survivors of leukemia appeared most at risk for poor educational outcomes, as demonstrated by an increased likelihood of poor achievement and greater special education utilization compared with population controls. Survivors of C N S tumours were also more l ikely to have physical disability designations, including increased visual and/or hearing disabilities than population controls. Visual and hearing disabilities have previously been observed among survivors of C N S tumours (Aarsen et al., 2006; F. D . Armstrong & Mulhern, 1999; Hoppe-Hirsch et a l , 1995; Jenkin, Danjoux, & Greenberg, 1998; Packer et a l , 2003). Similar to Mulhern and colleagues (Mulhern, 42 Wasserman, Friedman, & Fairclough, 1989), survivors of leukemia were more likely to have visual disabilities than population controls and consistent with several studies (Haupt et al., 1994; Madan-Swain & Brown, 1991; Stehbens et al., 1991), survivors of leukemia also had significantly more learning disability designations than the control group. Unlike Barrera et al. (Barrera, Shaw, Speechley, Maunsell, & Pogany, 2005), survivors of neuroblastoma were not more likely to have academic school problems than the comparison population based upon achievement on F S A or provincial exams; however, in agreement with Mi tby and colleagues (Mitby et al., 2003), survivors of neuroblastoma were at increased risk for special education compared to population controls. Notably, survivors of childhood cancer other than survivors of both leukemia and C N S tumours displayed no performance differences in comparison with the general population controls on any educational achievement measure (FSA or provincial exam). This finding suggests that cancer survivors, excluding those of leukemia and C N S tumours, are achieving similarly within school to their peers. In addition, when comparing the remaining survivor group - C N S tumours and leukemia excluded - with the comparison population there were no significant differences in F S A participation rates or Grade 12 course enrolment rates, except in Math 12. Both survivors of C N S tumours and leukemia displayed significantly reduced enrolment in Math 12 compared to population controls. It is possible that the decreased enrolment in Math 12 may reflect a decision not to partake in this course due to a deficit in mathematic skills; deficits in this area and poor arithmetic achievement have been commonly reported among survivors of childhood cancer (Coniglio & Blackman, 1995; Duffner, 2004; Gamis & Nesbit, 1991; Kaemingk, Carey, Moore, Herzer, & Hutter, 2004; Moleski , 2000; B . Moore, 2005; Mulhern & Palmer, 2003; Peckham, 1991; Peckham, Meadows, Battel, & 43 Marrero, 1988; Stehberis et a l , 1991; Whitt, Wells, Lauria, Wilhelm, & M c M i l l a n , 1984). Among survivors for whom treatment data was available, survivors whose treatment involved radiation, or more specifically cranial radiation, had poorer achievement and more physical disabilities, including increased visual disability designations, than those survivors who did not receive these treatments. Radiation, especially cranial radiation, is frequently noted as a risk factor for poor educational outcomes among survivors of childhood cancer (V. Anderson, Godber, Smibert, & Ekert, 1997; V . Anderson, Smibert, Ekert, & Godber, 1994; V . Anderson, Godber, Smibert, Weiskop, & Ekert, 2000; Copeland et al., 1985; H i l l et al., 1998; Smibert, Anderson, Godber, & Ekert, 1996) and visual impairments are often reported among those who receive radiation treatment (F. Armstrong & Mulhern, 1999; Jenkin, Danjoux, & Greenberg, 1998; Mulhern, Wasserman, Friedman, & Fairclough, 1989; Ober, Beaverson, & Abramson, 2004; Syndikus, Tait, Ashley, & Jannoun, 1994). Contrary to previous findings (V. Anderson, Godber, Smibert, & Ekert, 1997; Copeland et al., 1985; Jannoun & Chessells, 1987; Kingma, Mooyaart, Kamps, Nieuwenhuizen, & Wilmink, 1993; Kingma, Rammeloo, van der Does-van den Berg, Rekers-Mombarg, & Postma, 2000; I. Moore, Kramer, Wara, Halberg, & Abl in , 1991; Seaver et a l , 1994; Smibert, Anderson, Godber, & Ekert, 1996), survivors who were younger in age at diagnosis (<2 years) were less at risk for poor educational achievement than survivors who were older in age at diagnosis (>5 years). Furthermore, this was despite survivors younger in age at diagnosis being more likely to have hearing and/or visual disabilities than survivors older in age at diagnosis. It is possible that the accommodations provided for these disabilities may have helped compensate with respect to achievement. 44 Gender differences within the survivor group were similar to past research (Brown et al., 1998; Eiser, 1991; Haupt et al., 1994; Kelaghan et al., 1988; Langeveld et al., 2003; Mulhern, Fairclough, & Ochs, 1991; Waber, Bernstein, Kammerer, & Tarbell, 1992); females were more at risk for poorer educational outcomes than males with respect to both achievement and special education designations. Although both female and male survivors were more likely to have special education designations and physical, hearing and/or visual disabilities than their control counterparts, female survivors also had increased learning disability designations than female controls whereas there were no differences in learning disability designations between male survivors and controls. There were no significant differences between male and female survivors with respect to learning disability designations and within controls, males were significantly more likely than females to have a learning disability. Interestingly, female gender also appears to be a risk factor among disorders such as autism and attention deficit hyperactivity disorder ( A D H D ) ; while these disorders are more prevalent among males, females with these disorders are more severely impaired (DuPaul et a l , 2006; Erne, 1992; Madan-Swain & Brown, 1991; Rucklidge & Tannock, 2001), including more impaired intellectual functioning (Gaub & Carlson, 1997; Gershon, 2002; Volkmar & Szatmari, 1993). This study addresses past methodological shortcomings, particularly in using standardized educational data to investigate long-term educational achievement. Commonly employed questionnaire-based survivor cohort studies (Barrera, Shaw, Speechley, Maunsell, & Pogany, 2005; Kadan-Lottick et al., 2002; Langeveld et al., 2003; Taylor et al., 2004) that use self- or proxy-reported data are less reliable than primary sources of outcome data, and limited to outcome information that is identified prior to the study and what can be reasonably and reliably recalled. Only two recent studies (Harila-Saari et al., 2007; Lahteenmaki et al., 2007) have 45 employed outcome data with standardized reporting to investigate educational achievement and these studies involved only leukemia and brain tumours. A s well , the use of a population-based cohort ascertains information on a complete survivor group and relieves concerns of small sample sizes.(Patenaude & Kupst, 2005) Earlier studies have typically been small institution-based case series or cooperative group studies focused on specific diagnostic groups with short follow-up. In addition to addressing methodological issues of earlier research, this study fills a gap in the literature on late effects of survivors of childhood cancer by examining standardized educational achievement from an objective data source. Although previous studies have investigated educational attainment, special education utilization, and reported educational difficulties, until now, there have been no reports on how survivors, as a population, are actually performing in school long-term. It has been consistently suggested that survivors experience adverse neurocognitive late effects (Campbell et al., 2007; Dickerman, 2007; Mulhern & Palmer, 2003; National Cancer Pol icy Board, 2003; Spencer, 2006), but this has yet to be clearly linked with poor achievement in school. Knowing that survivors of leukemia and C N S tumours appeared at risk for poor educational achievement, while the remainder of survivors were essentially achieving similarly to their peers, has important implications for educators. This remaining group o f survivors did exhibit an increased use of special education programs, suggesting that surveillance may be necessary to address possible special education needs, but intense and time consuming assessment may not be required. Survivors of leukemia and C N S tumours, given their susceptibility as a high risk group for both poor achievement and special education utilization, should be more thoroughly monitored and assessed to ensure that the necessary interventions are 46 developed and implemented. In addition, regular monitoring of progress over time (F. Armstrong, Blumberg, & Toledano, 1999; F. Armstrong & Horn, 1995) and early identification (Coniglio & Blackman, 1995; Robison et al., 2005; Spencer, 2006) are essential in providing appropriate special education services or approaches to learning. Educational practitioners need to be aware of potential educational difficulties and associated risk factors, particularly who is most at risk, among survivors of childhood cancer so as to meet the educational needs of this population as their numbers continue to increase (Dickerman, 2007). Furthermore, dissemination of information between medical and school personnel is fundamental (Coniglio & Blackman, 1995) towards improving the educational experiences of survivors. 47 2.5 References Aarsen, F. K . , Paquier, P. F. , Reddingius, R. E . , Streng, I. C , Arts, W . M . , Evera-Preesman, M . , et al. (2006). Functional outcome after low-grade astrocytoma treatment in childhood. Cancer, 106(2), 396-402. Anderson, V . , Godber, T., Smibert, E . , & Ekert, H . (1997). Neurobehavioural sequelae following cranial irradiation and chemotherapy in children: an analysis of risk factors. Pediatr Rehabil, 1(2), 63-76. Anderson, V . , Smibert, E . , Ekert, H . , & Godber, T. (1994). Intellectual, educational, and behavioural sequelae after cranial irradiation and chemotherapy. Arch Dis Child, 70(6), 476-483. Anderson, V . A . , Godber, T., Smibert, E . , Weiskop, S., & Ekert, H . (2000). Cognitive and academic outcome following cranial irradiation and chemotherapy in children: a longitudinal study. Br J Cancer, 82(2), 255-262. Armstrong, F. D . , Blumberg, M . J., & Toledano, S. R. (1999). Neurobehavioral issues in childhood cancer. School Psychology Review, 28(2), 194. Armstrong, F. D . , & Horn, M . (1995). Educational issues in childhood cancer. School Psychology Quarterly, 10(A), 292-304. Armstrong, F . D . , & Mulhern, R. K . (1999). Acute lymphoblastic leukemia and brain tumors. In R. T. Brown (Ed.), Cognitive Aspects of Chronic Illness in Children (pp. 1-14). New York: The Guilford Press. Barrera, M . , Shaw, A . K . , Speechley, K . N . , Maunsell, E . , & Pogany, L . (2005). Educational and social late effects of childhood cancer and related clinical, personal, and familial characteristics. Cancer, 104(8), 1751-1760. B C Ministry of Education, G . a. L . U . (2006). Provincial Letter Grades Order. Retrieved July, 13, 2007, from http://www.bced.gov.bc.ca/policy/policies/student_reporting.htm Brown, R. T., Madan-Swain, A . , Walco, G . A . , Cherrick, I., levers, C. E . , Conte, P. M . , et al. (1998). Cognitive and academic late effects among children previously treated for acute lymphocytic leukemia receiving chemotherapy as C N S prophylaxis. J Pediatr Psychol, 23(5), 333-340. Buizer, A . I., de Sonneville, L . M . J., van den Heuvel-Eibrink, M . M . , & Veerman, A . J. P. (2006) . Behavioral and educational limitations after chemotherapy for childhood acute lymphoblastic leukemia or Wilms tumor. Cancer, 106(9), 2067-2075. Butler, R. W. , H i l l , J. M . , Steinherz, P. G. , Meyers, P. A . , & Finlay, J. L . (1994). Neuropsychologic effects of cranial irradiation, intrathecal methotrexate, and systemic methotrexate in childhood cancer. J Clin Oncol, 12(12), 2621-2629. Campbell, L . K . , Scaduto, M . , Sharp, W. , Dufton, L . , V a n Slyke, D . , Whitlock, J. A . , et al. (2007) . A meta-analysis of the neurocognitive sequelae of treatment for childhood acute lymphocytic leukemia. Pediatr Blood Cancer, 49(1), 65-73. Canadian Cancer Society/National Cancer Institute of Canada. (2007). Canadian Cancer Statistics 2007. Toronto, Canada. Christie, D . , Leiper, A . D . , Chessells, J. M . , & Vargha-Khadem, F. (1995). Intellectual performance after presymptomatic cranial radiotherapy for leukaemia: effects of age and sex. Arch Dis Child, 73(2), 136-140. 48 Coniglio, S. J . , & Blackman, J. A . (1995). Developmental outcome of childhood leukemia. Topics in Early Childhood Special Education, 75(1), 19. Copeland, D. R., Fletcher, J. M . , Pfefferbaum-Levine, B. , Jaffe, N . , Ried, H . , & Maor, M . (1985). Neuropsychological sequelae of childhood cancer in long-term survivors. Pediatrics, 75(4), 745-753. Dickerman, J. D . (2007). The Late Effects of Childhood Cancer Therapy. Pediatrics, 119(3), 554-568. Dolgin, M . J. , Somer, E . , Buchvald, E . , & Zaizov, R. (1999). Quality of life in adult survivors of childhood cancer. Soc Work Health Care, 28(4), 31-43. Duffher, P. K . (2004). Long-term effects of radiation therapy on cognitive and endocrine function in children with leukemia and brain tumors. Neurologist, 10(6), 293-310. DuPaul, G . J. , Jitendra, A . K . , Tresco, K . E . , Vile Junod, R. E . , Volpe, R. J. , & Lutz, J. G . (2006) . Children With Attention Deficit Hyperactivity Disorder: Are There Gender Differences in School Functioning? School Psychology Review, 35(2), 292-308. Eiser, C . (1991). Cognitive deficits in children treated for leukaemia. Arch Dis Child, 66(1), 164-168. Eiser, C . (2004). Neurocognitive sequelae of childhood cancers and their treatment: a comment on Mulhern and Butler. Pediatric Rehabilitation, 7(1), 15-16. Erne, R. F. (1992). Selective Females Affliction in the Developmental Disorders of Childhood: A Literature Review. Journal of Clinical Child Psychology, 21(4), 354. Gamis, A . S., & Nesbit, M . E . (1991). Neuropsychologic (cognitive) disabilities in long-term survivors of childhood cancer. Pediatrician, 18(1), 11-19. Gaub, M . , & Carlson, C . L . (1997). Gender differences in A D H D : A meta-analysis and critical review. Journal of the American Academy of Child & Adolescent Psychiatry, 36(8), 1036. Gershon, J. (2002). A meta-analytic review of gender differences in A D H D . Journal of Attention Disorders, 5(3), 143-154. Halberg, F. E . , Kramer, J. H . , Moore, I. M . , Wara, W. M . , Matthay, K . K . , & Ablin, A . R. (1992). Prophylactic cranial irradiation dose effects on late cognitive function in children treated for acute lymphoblastic leukemia. IntJRadiat Oncol Biol Phys, 22(1), 13-16. Harila-Saari, A . H . , Lahteenmaki, P. M . , Pukkala, E . , Kyyronen, P., Lanning, M . , & Sankila, R. (2007) . Scholastic Achievements of Childhood Leukemia Patients: A Nationwide, Register-Based Study. J Clin Oncol, 25(23), 3518-3524. Haupt, R., Fears, T. R., Robison, L . L . , Mills, J. L . , Nicholson, H . S., Zeltzer, L . K . , et al. (1994). Educational attainment in long-term survivors of childhood acute lymphoblastic leukemia. Jama, 272(18), 1427-1432. Hil l , J. M . , Kornblith, A . B. , Jones, D. , Freeman, A . , Holland, J. F . , Glicksman, A . S., et al. (1998). A comparative study of the long term psychosocial functioning of childhood acute lymphoblastic leukemia survivors treated by intrathecal methotrexate with or without cranial radiation. Cancer, 82(1), 208-218. Hoppe-Hirsch, E . , Brunet, L . , Laroussinie, F. , Cinalli, G . , Pierre-Kahn, A . , Renier, D. , et al. (1995). Intellectual outcome in children with malignant tumors of the posterior fossa: influence of the field of irradiation and quality of surgery. Child's Nervous System, 11(6), 340-345. Jannoun, L . , & Chessells, J. M . (1987). Long-term psychological effects of childhood leukemia and its treatment. Pediatr Hematol Oncol, 4(4), 293-308. 49 Jenkin, D . , Danjoux, C , & Greenberg, M . (1998). Subsequent quality of life for children irradiated for a brain tumor before age four years. Med Pediatr Oncol, 31(6), 506-511. Kadan-Lottick, N . S., Robison, L . L . , Gurney, J. G . , Neglia, J. P., Yasui , Y . , Hayashi, R., et al. (2002) . Childhood Cancer Survivors' Knowledge About Their Past Diagnosis and Treatment: Childhood Cancer Survivor Study. JAMA, 257(14), 1832-1839. Kaemingk, K . L . , Carey, M . E . , Moore, I. M . , Herzer, M . , & Hutter, J. J. (2004). Math Weaknesses in Survivors of Acute Lymphoblastic Leukemia Compared to Healthy Children. Child Neuropsychology, 10(\), 14-23. Kelaghan, J., Myers, M . H . , M u l v i h i l l , J. J., Byrne, J., Connelly, R. R. , Austin, D . F. , et al. (1988). Educational achievement of long-term survivors of childhood and adolescent cancer. Med Pediatr Oncol, 16(5), 320-326. Kingma, A . , Mooyaart, E . L . , Kamps, W . A . , Nieuwenhuizen, P., & Wilmink, J. T. (1993). Magnetic resonance imaging of the brain and neuropsychological evaluation in children treated for acute lymphoblastic leukemia at a young age. Am J Pediatr Hematol Oncol, 15(2), 231-238. Kingma, A . , Rammeloo, L . A . , van Der Does-van den Berg, A . , Rekers-Mombarg, L . , & Postma, A . (2000). Academic career after treatment for acute lymphoblastic leukaemia. Arch Dis Child, 82(5), 353-357. Kingma, A . , van Dommelen, R. I., Mooyaart, E . L . , Wilmink, J. T., Deelman, B . G . , & Kamps, W . A . (2001). Slight cognitive impairment and magnetic resonance imaging abnormalities but normal school levels in children treated for acute lymphoblastic leukemia with chemotherapy only. J Pediatr, 139(3), 413-420. Koch , S. V . , Kejs, A . M . T., Engholm, G. , Johansen, C , & Schmiegelow, K . (2004). Educational attainment among survivors of childhood cancer: a population-based cohort study in Denmark. British Journal of Cancer, 91(5), 923-928. Kramarova, E . , & Stiller, C. A . (1996). The international classification o f childhood cancer. Int J Cancer, 68(6), 759-765. Lahteenmaki, P. M . , Harila-Saari, A . , Pukkala, E . I., Kyyronen, P., Salmi, T. T., & Sankila, R. (2007). Scholastic achievements of children with brain tumors at the end of comprehensive education: A nationwide, register-based study. Neurology, 69(3), 296-305. Langeveld, N . E . , Ubbink, M . C , Last, B . F., Grootenhuis, M . A . , Voute, P. A . , & De Haan, R. J. (2003) . Educational achievement, employment and l iving situation in long-term young adult survivors of childhood cancer in the Netherlands. Psychooncology, 12(3), 213-225. Link, K . , Moe l l , C , Osterberg, K . , Persson, R., Orbaek, P., Garwicz, S., et al. (2006). Adult survivors of childhood acute lymphoblastic leukaemia with G H deficiency have normal self-rated quality of life but impaired neuropsychological performance 20 years after cranial irradiation. Clin Endocrinol (Oxf), 65(5), 617-625. Madan-Swain, A . , & Brown, R. T. (1991). Cognitive and psychosocial sequelae for children with acute lymphocytic leukemia and their families. Clinical Psychology Review, 11(3), 267-294. Mitby, P. A . , Robison, L . L . , Whitton, J. A . , Zevon, M . A . , Gibbs, I. C , Tersak, J. M . , et al. (2003). Utilization of special education services and.educational attainment among long-term survivors of childhood cancer: a report from the Childhood Cancer Survivor Study. Cancer, 97(4), 1115-1126. 50 Moehle, K . A . , Berg, R. A . , Ch'ien, L . T., & Lancaster, W . (1983). Language-related skills in children with acute lymphocytic leukemia. J Dev Behav Pediatr, 4(4), 257-261. Moleski , M . (2000). Neuropsychological, Neuroanatomical, and Neurophysiological Consequences of C N S Chemotherapy for Acute Lymphoblastic Leukemia. Archives of Clinical Neuropsychology, 15(1), 603-630. Moore, B . D . , 3rd. (2005). Neurocognitive outcomes in survivors of childhood cancer. J Pediatr Psychol, 30(1), 51-63. Moore, I. M . , Glasser, M . E . , & Ab l in , A . R. (1988). The late psychosocial consequences of childhood cancer. J Pediatr Nurs, 3(3), 150-158. Moore, I. M . , Kramer, J. H . , Wara, W. , Halberg, F. , & Ab l in , A . R. (1991). Cognitive function in children with leukemia. Effect o f radiation dose and time since irradiation. Cancer, 68(9), 1913-1917. Mulhern, R. K . , Fairclough, D . , & Ochs, J. (1991). A prospective comparison of neuropsychologic performance of children surviving leukemia who received 18-Gy, 24-Gy, or no cranial irradiation. J Clin Oncol, 9(8), 1348-1356. Mulhern, R. K . , & Palmer, S. L . (2003). Neurocognitive late effects in pediatric cancer. Curr Probl Cancer, 27(4), 177-197. Mulhern, R. K . , Wasserman, A . L . , Friedman, A . G. , & Fairclough, D . (1989). Social competence and behavioral adjustment of children who are long-term survivors of cancer. Pediatrics, 83(1), 18-25. Nagarajan, R., Neglia, J. P., Clohisy, D . R., Yasui , Y . , Greenberg, M . , Hudson, M . , et al. (2003). Education, employment, insurance, and marital status among 694 survivors of pediatric lower extremity bone tumors: a report from the childhood cancer survivor study. Cancer, 97(10), 2554-2564. National Cancer Policy Board (Ed.). (2003). Childhood Cancer Survivorship: Improving Care and Quality of Life. Washington, D C : National Academic Press. Ober, M . , Beaverson, K . , & Abramson, D . (2004). Ocular complications. In W . H . Wallace & D . M . Green (Eds.), Late Effects of Childhood Cancer (pp. 37-48). New York: Oxford University Press. Packer, R. J., Gurney, J. G . , Punyko, J. A . , Donaldson, S. S., Inskip, P. D . , Stovall, M . , et al. (2003). Long-Term Neurologic and Neurosensory Sequelae in Adult Survivors of a Childhood Brain Tumor: Childhood Cancer Survivor Study. J Clin Oncol, 21(11), 3255-3261. Patenaude, A . F. , & Kupst, M . J. (2005). Psychosocial functioning in pediatric cancer. J Pediatr Psychol, 30(\), 9-27. Peckham, V . C. (1991). Educational deficits in survivors of childhood cancer. Pediatrician, 18(1), 25-31. Peckham, V . C , Meadows, A . T., Bartel, N . , & Marrero, O. (1988). Educational late effects in long-term survivors of childhood acute lymphocytic leukemia. Pediatrics, 81(1), 127-133. Policy Document: Special Education. (2006). Retrieved, from http://www.bced.gov.bc.ca/policy/policies/special_ed.htm. Raymond-Speden, E . , Tripp, G . , Lawrence, B . , & Holdaway, D . (2000). Intellectual, neuropsychological, and academic functioning in long-term survivors of leukemia. J Pediatr Psychol, 25(2), 59-68. 51 Ries, L . A . G . , Melbert, D . , Krapcho, M . , Mariotto, A . , Mi l le r , B . A . , Feuer, E . J., et al. (Eds.). (2007). SEER Cancer Statistics Review, 1975-2004. Bethesda, M D : National Cancer Institute. Robison, L . L . , Green, D . M . , Hudson, M . , Meadows, A . T., Mertens, A . C , Packer, R. J., et al. (2005). Long-term outcomes of adult survivors of childhood cancer. Cancer, 104(l\ Suppl), 2557-2564. Rubenstein, C. L . , Varni , J. W. , & Katz, E . R. (1990). Cognitive functioning in long-term survivors of childhood leukemia: a prospective analysis. JDev Behav Pediatr, 11(6), 301-305. Rucklidge, J. J., & Tannock, R. (2001). Psychiatric, psychosocial, and cognitive functioning of female adolescents with A D H D . Journal of the American Academy of Child & Adolescent Psychiatry, 40(5), 530-540. Seaver, E . , Geyer, R., Sulzbacher, S., Warner, M . , Batzel, L . , Milstein, J., et al. (1994). Psychosocial adjustment in long-term survivors of childhood medulloblastoma and ependymoma treated with craniospinal irradiation. Pediatr Neurosurg, 20(4), 248-253. Smibert, E . , Anderson, V . , Godber, T., & Ekert, H . (1996). Risk factors for intellectual and educational sequelae of cranial irradiation in childhood acute lymphoblastic leukaemia. Br J Cancer, 73(6), 825-830. Smith, M . , & Hare, M . L . (2004). A n overview of progress in childhood cancer survival. J Pediatr Oncol Nurs, 21(3), 160-164. Spencer, J. (2006). The role of cognitive remediation in childhood cancer survivors experiencing neurocognitive late effects. Journal of Pediatric Oncology Nursing, 23(6), 321-325. Stehbens, J. A . , Kaleita, T. A . , N o l l , R. B . , MacLean, W . E . , Jr., O'Brien, R. T., Waskerwitz, M . J., et al. (1991). C N S prophylaxis of childhood leukemia: what are the long-term neurological, neuropsychological, and behavioral effects? Neuropsychol Rev, 2(2), 147-177. Syndikus, I., Tait, D . , Ashley, S., & Jannoun, L . (1994). Long-term follow-up of young children with brain tumors after irradiation. Int JRadiat Oncol Biol Phys, 30(4), 781-787. Taylor, A . , Hawkins, M . , Griffiths, A . , Davies, H . , Douglas, C , Jenney, M . , et al. (2004). Long-term follow-up of survivors of childhood cancer in the U K . Pediatr Blood Cancer, 42(2), 161-168. Volkmar, F. R., & Szatmari, P. (1993). Sex differences in pervasive developmental disorders. Journal of Autism & Developmental Disorders, 23(4), 579-591. Waber, D . P., Bernstein, J. H . , Kammerer, B . L . , & Tarbell, N . J. (1992). Neuropsychological diagnostic profiles of children who received C N S treatment for acute lymphoblastic leukemia: The systemic approach to assessment. Developmental Neuropsychology, 8(1), 1-28. Whitt, J. K . , Wells, R. J., Lauria, M . M . , Wilhelm, C. L . , & M c M i l l a n , C. W . (1984). Cranial radiation in childhood acute lymphocytic leukemia. Neuropsychologic sequelae. Am J Dis Child, 138(8), 730-736. Wilkins, R. (2002). PCCF+ version 3 J user's guide. Automated geographic coding based on the Statistics Canada postal code conversion files (Vol . 61). Ottawa, Canada: Health Analysis and Measurement Group, Statistics Canada. Zebrack, B . J., Zevon, M . A . , Turk, N . , Nagarajan, R., Whitton, J., Robison, L . L . , et al. (2007). Psychological distress in long-term survivors of solid tumors diagnosed in childhood: a report from the childhood cancer survivor study. Pediatric Blood & Cancer, 49(1), 47-51. 52 C H A P T E R 3: D I S C U S S I O N 3.1 Conclusions Survivors of childhood cancer as a whole may not necessarily be at risk for poor educational outcomes with respect to school achievement. However, survivors of C N S tumours, followed by survivors of leukemia, were found most at risk for both poor achievement (according to provincial exams and FSAs) and increased special education utilization (overall and with respect to several specific special education designations). Once survivors of these diagnoses were excluded from analyses, remaining survivors performed similarly on all F S A s and provincial exams to population controls. These remaining survivors were still more likely to have a special education designation; specifically, to have a physical disability, a hearing disability, or a visual disability compared to population controls and as such survivors of childhood cancer as a whole may require some unique consideration within the education system. These might include surveillance or extra vigilance and the provision of accommodations on the part of educators and school personnel. A s was noted earlier, survivors of childhood C N S tumours and leukemia are particularly at risk for poor educational outcomes. Survivors of C N S tumours are clearly the most at risk group for poor achievement, performing more poorly on all F S A s and the Communications 12 provincial exam than population controls. Survivors of leukemia, on the other hand, performed more poorly on three of the nine F S A s than the population control group, but similarly on all five provincial exams investigated. With respect to special education, both survivors of C N S tumours and leukemia were more likely, overall, to have a special education designation, and specifically, a general physical disability or a visual disability than the population control group. Survivors of C N S tumours were also more likely to have a hearing disability and survivors of leukemia a 53 learning disability compared to controls. Survivors of neuroblastoma were the only other diagnosis group to show any specific at risk tendencies/being more likely to have a special education designation or physical disability than population controls. Survivors of CNS tumours (most obviously) and survivors of leukemia are most in need of early educational intervention given their propensity towards both poor achievement and increased special education. Survivors of neuroblastoma still require additional support from educators due their likelihood of increased special education designations. Several other at risk groups emerged among survivors that may also require educational interventions; these include female survivors and survivors who received radiation or CRT. First, female survivors may be more likely to have poor achievement as they performed more poorly than their control counterparts on more FSA and provincial exams than males survivors compared to their control counterparts. As well, female survivors were more likely to have a learning disability than female controls, while there was no difference between male survivors and male controls with respect to learning disabilities. Secondly, although treatment data was limited, among survivors of leukemia, lymphoma, and CNS tumours it was found that those treated with radiation or CRT were more at risk for poor achievement and physical and visual disabilities than those not receiving radiation or CRT. Age at diagnosis was also explored; survivors younger in age at diagnosis (<2 years) were less likely than survivors older in age at diagnosis (5 years or older) to have poor educational achievement (although the difference existed on only two outcome measures), but more likely to have hearing and visual disabilities. It may be more informative to examine the effects of age at treatment rather than age at diagnosis as some studies have suggested that an interaction between age and radiation exists in that individuals who are younger in age at treatment are more vulnerable to the effects of radiation, 54 particularly C R T , and as such are more likely to experience educational deficits (Moore, Kramer, Wara, Halberg, & Ab l in , 1991; Smibert, Anderson, Godber, & Ekert, 1996); literature reviews have reported similarly (F. D . Armstrong & Mulhern, 1999; Eiser, 1998; Mulhern & Palmer, 2003). In general these findings are consistent with the current survivorship literature on long-term educational outcomes. Survivors of C N S cancer and survivors of leukemia are commonly identified as likely to experience educational difficulties (Barrera, Shaw, Speechley, Maunsell, & Pogany, 2005; Langeveld et a l , 2003; Mitby et al., 2003). However, survivors of Hodgkin's and non-Hodgkin's lymphoma were not found at increased risk for poor educational outcome as was reported previously (Mitby et al., 2003) and nor were survivors of neuroblastoma more likely to have poor scholastic achievement (Barrera, Shaw, Speechley, Maunsell, & Pogany, 2005), although they were more vulnerable towards special education designations than population controls. Also in agreement with past findings, treatment including radiation or C R T was related to increased educational deficits among survivors (Kingma et al., 2001; Mi tby et al., 2003; Precourt et al., 2002; von der Weid, 2001). Female gender among survivors as a risk modifier for increased likelihood of poor educational outcomes (Eiser, 1991; Haupt et al., 1994; Langeveld et al., 2003) was also supported by this research. A n d lastly, conversely to prior results, younger age at diagnosis (<2 years) was not associated with poorer educational achievement (Anderson, Godber, Smibert, & Ekert, 1997; Copeland et al., 1985; Jannoun & Chessells, 1987; Kingma, Mooyaart, Kamps, Nieuwenhuizen, & Wilmink, 1993; Kingma, Rammeloo, van Der Does-van den Berg, Rekers-Mombarg, & Postma, 2000; Moore, Kramer, Wara, Halberg, & Ab l in , 1991; Seaver et al., 1994; Smibert, Anderson, Godber, & Ekert, 1996). 55 3.2 Strengths and Limitat ions Potential limitations of this study include the heterogeneity o f the survivor group, small sample sizes within survivor subgroups, the comparison group used, and the consideration of possible confounding variables. Every effort was made to address or make adjustments for these potential shortcomings and one of the major strengths of this project is that it addresses methodological criticisms of previous research. Furthermore, this project has considerable value in its use of a population-based survivor cohort and use of objective education data that was collected and reported in a standardized manner. The heterogeneous nature of the survivor cohort, given that this was a population-based cohort study, was unavoidable; no one survivor w i l l have the exact same experience as another. In an attempt to attend to this concern, in addition to examining the survivor cohort in its entirety, analyses of specific diagnosis groups and clinical-related modifiers (e.g., treatment modality and age at diagnosis) were considered in relation to educational outcome. This assists in improving the homogeneity of the study cohort by limiting the group to only specific diagnosis or diagnoses groups, those who received particular treatment regimens, and certain age at diagnosis groups. However, the impact of the cancer itself and its treatment may vary even within a group who have the same diagnosis and treatment (F. Daniel Armstrong & Horn, 1995). In creating more homogeneous groups, through the study of particular subgroups within the survivor cohort (e.g., specific diagnosis groups and treatment modality), sample size was at times compromised. Some of the specific cancer diagnoses and potential modifiers intended for study elicited relatively small subgroups which limited, and in some cases, even eliminated probable statistical analyses. Small sample sizes have commonly been noted as a methodological shortcoming among studies of survivors of cancer (Brown & Madan-Swain, 1993; Patenaude & 56 Kupst, 2005). Conversely, the fact that this was a population-based cohort was a significant strength as the survivor cohort was more than ample in numbers and findings are representative of a complete populace of survivors for a geographical region (an entire Canadian province). Earlier studies have often been small institution-based case series or cooperative group studies focused on specific diagnostic groups limited in both length and nature of follow-up. Some would consider the sole use of a population control group for comparison purposes inadequate; however, the use of healthy population controls matched by gender, age, and SES have been noted as more appropriate controls than normative data (Campbell et al., 2007). Some studies have employed or suggested that sibling control groups (Buizer, de Sonneville, van den Heuvel-Eibrink, & Veerman, 2006; Haupt et al., 1994; Mi tby et al., 2003), samples of individuals with a chronic illness (Raymond-Speden, Tripp, Lawrence, & Holdaway, 2000), or even groups of other survivors of cancer with different diagnoses or treatments (Madan-Swain & Brown, 1991) provide the best comparison groups. There does not appear to be consensus within the literature as to what entails the most appropriate comparison group for survivors of childhood cancer research. The omission of a control group is obviously o f concern, and in some cases, past studies have lacked a control group all together (Brown et al., 1998; Fogarty et al., 1988; Humpl, Fritsche, Bartels, & Gutjahr, 2001; Rubenstein, Varni , & Katz, 1990; Syndikus, Tait, Ashley, & Jannoun, 1994). With respect to the present project, the identification of a sibling control group had been intended for comparison purposes; unfortunately, this was not possible as approval from the B C Ministry of Education was not granted for the release o f sibling education data at the time of the initial data request. This research did however employ gender- and age-matched population controls, as well as performed analyses within the survivor cohort comparing different treatment modality groups and age at diagnosis groups; thus, in effect, comparing 57 different survivor groups with each other. Although many possible confounding variables have been taken into consideration, school missed is one factor that could not be directly controlled for given that school records providing this information could not be acquired. A criticism of previous studies is their lack of control for school attendance (Brown & Madan-Swain, 1993). Although it should be noted that school attendance may be of concern during treatment, attendance does improve after therapy completion (Eiser & Vance, 2002; Jannoun & Chessells, 1987) and thus would not appear to be a problem for 5-year survivors, as is the case in this research project. A s an indicator of school attendance, the full-time course enrolment status of both cohorts was explored and found not to differ between survivors and population controls (not reported). Wi th respect to other potential confounding factors raised within the literature such as SES (Bader-Meunier, Tchernia, & Dommergues, 1996) and health or physical impairments, (F. Daniel Armstrong & Horn, 1995) these were all considered in the analysis model of all educational outcomes (achievement measures and special education designations). One of the most important aspects of this research was the type of data and data collection method employed. To date, very few studies have investigated school performance through actual school marks (Harila-Saari et al., 2007; Lahteenmaki et al., 2007) and the use of self- or proxy-reported data has been common among recent survivor population studies in this field (Barrera, Shaw, Speechley, Maunsell, & Pogany, 2005; Langeveld et al., 2003; Mi tby et al., 2003). Self- or proxy-reported data relies on an individual's ability to recall accurately past information and can often be biased. The use of objective data that has been collected and reported in a standardized format, as is the case in this project, provides higher quality and substantially more accurate data and thus more credible results upon which to base 58 interpretations and recommendations. Unfortunately, there were several limitations of the data which reduced potential analyses or posed possible shortcomings within this project. First, clinical data with respect to treatment was only available, at the time of analysis, for diagnoses of leukemia, lymphoma, and C N S tumours. Fortunately, these diagnosis groups were the predominant cancer diagnoses within the survivor cohort, but interpretation of the effects of treatment should still only consider those cancer diagnoses for which treatment information was available. A s well , only basic treatment modality (chemotherapy, radiation, C R T , and surgery) data existed and more specific information pertaining to radiation dosage and chemotherapy drug and dosage were unavailable at the time of study. Although treatment data was incomplete valuable preliminary results were still established. Moreover, many studies lack treatment data entirely, such as the Danish population study (Koch, Kejs, Engholm, Johansen, & Schmiegelow, 2004). Lastly, the potential exists that these findings may not generalize to other populations of survivors of childhood cancer. Although a major strength of research is the fact that it is a population-based cohort, not a sample group, and thus better representative of the study population, it is still the population of a specific geographical region within a specific country. Other geographical regions, due to any number of potential differences within the population such as their cancer treatment protocols, education system, and so on, may therefore find alternative outcomes among survivors o f childhood cancer. 3.3 Significance This research provides long-term objective data on overall and disease-related educational outcomes of survivors of childhood cancer. This is the first Canadian population cohort study to investigate educational late effects by means of standardized educational data 59 including school marks. Only two recently published studies (Harila-Saari.et al., 2007; Lahteenmaki et al., 2007) have utilized standardized school marks to investigate the educational achievements of a population of individuals diagnosed with cancer. What these two studies lack is a complete survivor population as one of these studies focused solely on childhood leukemia (Harila-Saari et al., 2007) and the other on childhood brain tumours (Lahteenmaki et al., 2007). The only other Canadian population-based research of survivors' educational outcomes employed proxy-reported data (Barrera, Shaw, Speechley, Maunsell, & Pogany, 2005). A s was noted earlier, employing actual school data that is collected and reported in a standardized fashion improves the quality of data, particularly in comparison with self- or proxy-reported data which has been previously utilized within other population studies (Langeveld et al., 2003; Mi tby et al., 2003). This project is clearly unique, and therefore fills a gap within the literature, in its use of standardized education achievement (school marks) data through administrative records to investigate the long-term educational outcomes of a population o f survivors of childhood cancer. A s well , as was mentioned within the previous section discussing the limitations and strengths, this research addresses the methodological shortcomings of previous work in this field of study; taking into consideration the criticisms of prior studies and improving upon earlier work. The distinctiveness of this research in its methodologies (survivor population and data utilized) and ability to attend to previous concerns within this area of the survivorship literature provide significant impact. The findings o f this project w i l l add to our understanding of the effects of diagnosis on the long-term educational achievement and special education utilization of survivors of childhood cancer. These findings clearly indicate which diagnosis groups are most at risk for poor educational achievement and special education designations and thus suggests where 60 resources may most be needed with respect to monitoring and intervention within the education system. A significant and optimistic finding to come out of this research was that while survivors of childhood leukemia and C N S tumours are the most at risk diagnosis groups for poor educational outcomes (including both exam/test achievement and special education designations), excluding these survivors from analyses indicates that the remainder of survivors are achieving similarly (according to standardized test/exam marks) to population controls. Moreover, this result is one not commonly reported, in fact, just the opposite was recently suggested in a survivor population within Canada (Barrera, Shaw, Speechley, Maunsell, & Pogany, 2005). The results of this research support the current understanding o f how cancer treatment impacts long-term educational outcomes. Although treatment data was only available for survivors of leukemia, lymphoma, and C N S tumours (approximately half the study population), findings are in keeping with previous studies. Survivors who receive radiation therapy or C R T are more at risk for poor educational outcomes (both achievement and special education) than those not receiving these therapies. In addition, findings regarding gender are in keeping with current survivorship literature; female survivors of childhood cancer are more at risk for poor educational outcomes (achievement and special education, especially learning disabilities) than male survivors. These effects of treatment and gender among survivors indicate that these are also potential at risk groups within the education system who may therefore require appropriate monitoring and intervention on behalf of educators. It is hoped that this work w i l l elicit an increased awareness among educators that survivors of childhood cancer are a potential at risk population within the school system who may require monitoring and intervention. Moreover, in providing information that increases the 61 understanding of which subgroups of survivors may be at increased risk of poor educational outcomes informs educational practice in aiding educators in the decision making process of who might be more in need of monitoring and intervention, and thus where to focus resources (e.g., time and money). These findings w i l l help guide the development o f appropriate educational interventions, including preventive and rehabilitative programs within schools. Overall, this project is intended to assist in providing successful educational experiences for survivors and improve the quality of life of survivors. 3.4 Future Research It is important to note that the number of survivors of childhood cancer w i l l only continue to increase (Dickerman, 2007) and thus so w i l l the number of survivors within our education systems. A continuation of research regarding the long term educational outcomes and experiences of survivors of childhood cancer is necessary to support current understandings, but also to continue to investigate the clinically-related effects; particularly as time passes and treatment regimens change due to medical improvements. A s well , to better understand why it is that certain diagnosis groups are more at risk for poor educational outcomes than others; is it the diagnosis itself, the treatment received, or a combination of both? Clearly, more work is needed with respect to more detailed treatment-related influences on long-term educational outcome such as mode of chemotherapy (intravenous, intrathecal, etc.), chemotherapy drug, dose of chemotherapy, and radiation or C R T dose. Obviously, the more knowledge that exists pertaining to which survivors are possibly more likely to experience poor educational outcomes, the more informative this is to educational practice and decision-making. Education personnel w i l l be capable o f more readily allocating resources and designing interventions that meet both the needs of educators and most importantly, survivors of childhood cancer. 62 With respect to appropriate educational intervention, further research is much needed identify where and what intervention is required. Presently, there exists a void as to which areas of learning within school, outside of standardized educational assessments, may pose the greatest difficulties for survivors and what type of intervention(s) may most meet their individual needs in providing success at school. Intervention design is challenging when little is known regarding which scholastic areas are likely to present problems among survivors (and certainly these may differ among survivor subgroups according diagnosis, treatment and so forth) and which particular interventions may most address these deficits within this population. The study of educational intervention effectiveness among survivors is even more lacking than that of the specific learning deficits. There exists almost no research on which intervention methods are best suited for these at risk individuals. Knowledge dissemination and transfer is another area where additional research would have a profound impact in the field of educational outcomes of survivors. More is needed to be done to increase the awareness among educators that survivors, notably particular subgroups of survivors, are potentially at risk for poor educational achievement and increased special education utilization. Educators have many students to be concerned with and may have few resources at hand (including their time) to already deal with the demanding responsibilities at hand; thus, the more information that can be provided them that informs decision-making and practice w i l l only assist both educators and survivors. Communication between medical and educational personnel is key, but first it is necessary to answer questions such as how this might be facilitated? Whether directly or indirectly, it is necessary for educators to be given specific information about the students they work with who are survivors o f childhood cancer. It is all well to have educators aware o f potential at risk groups among survivors, but this serves little 63 purpose i f they do not know i f their student falls into any of these subgroups. Whose responsibility is it to provide information to the schools? It may not be practical or feasible (or even legal) for medical practitioners to have direct contact with educators regarding specific cases, so are parents expected to relay and explain all medical information to their child's school or teacher? What methods and processes are in place to educate parents and educators? One study found that teachers provided with a web-based cancer education program demonstrated significant growth in their knowledge of cancer from pre- to post-test and most (87.8%) indicated that they would recommend the training to a colleague (Dubowy et al., 2006); unfortunately, few similar studies exist. Survivors themselves are not always able to provide accurate information with respect to their diagnosis or treatment (Kadan-Lottick et al., 2002) and of substantial concern is the fact that some survivors and their families even claim they were never made aware of potential late effects or the need for follow-up (National Cancer Pol icy Board, 2003). The goal of future survivorship research in the field of educational late effects should be how do we best ameliorate the educational experiences of survivors? This is a demanding goal, but one that is essential to improving the quality of life o f survivors. 64 ( 3.5 References Anderson, V . , Godber, T., Smibert, E . , & Ekert, H . (1997). Neurobehavioural sequelae following cranial irradiation and chemotherapy in children: an analysis of risk factors. Pediatr Rehabil, 1(2), 63-76. Armstrong, F. D . , & Horn, M . (1995). Educational issues in childhood cancer. School Psychology Quarterly, 10(4), 292-304. Armstrong, F. D . , & Mulhern, R. K . (1999). Acute lymphoblastic leukemia and brain tumors. In R. T. Brown (Ed.), Cognitive Aspects of Chronic Illness in Children (pp. 1-14). New York: The Guilford Press. Bader-Meunier, B . , Tchernia, G. , & Dommergues, J. P.. (1996). [Neuro-cognitive sequelae during acute lymphoblastic leukemia in children]. Arch Pediatr, 3(8), 745-748. Barrera, M . , Shaw, A . K . , Speechley, K . N . , Maunsell, E . , & Pogany, L . (2005). Educational and social late effects of childhood cancer and related clinical, personal, and familial characteristics. Cancer, 104(8), 1751-1760. Brown, R. T., & Madan-Swain, A . (1993). Cognitive, neuropsychological, and academic sequelae in children with leukemia. J Learn Disabil, 26(2), 74-90. Brown, R. T., Madan-Swain, A . , Walco, G . A . , Cherrick, I., levers, C. E . , Conte, P. M . , et al. (1998). Cognitive and academic late effects among children previously treated for acute lymphocytic leukemia receiving chemotherapy as C N S prophylaxis. J Pediatr Psychol, 23(5), 333-340. Buizer, A . I., de Sonneville, L . M . J., van den Heuvel-Eibrink, M . M . , & Veerman, A . J. P. (2006) . Behavioral and educational limitations after chemotherapy for childhood acute lymphoblastic leukemia or Wilms tumor. Cancer, 106(9), 2067-2075. Campbell, L . K . , Scaduto, M . , Sharp, W. , Dufton, L . , Van Slyke, D . , Whitlock, J. A . , et al. (2007) . A meta-analysis of the neurocognitive sequelae of treatment for childhood acute lymphocytic leukemia. Pediatr Blood Cancer, 49(\), 65-73. Copeland, D . R., Fletcher, J. M . , Pfefferbaum-Levine, B . , Jaffe, N . , Ried, H . , & Maor, M . (1985). Neuropsychological sequelae of childhood cancer in long-term survivors. Pediatrics, 75(4), 745-753. Dickerman, J. D . (2007). The Late Effects of Childhood Cancer Therapy. Pediatrics, 119(3), 554-568. Dubowy, R. L . , Rieger, B . P., Songer, N . S., Kleinmann, A . E . , Lewandowski, L . J., Rogers, C. L . , et al. (2006). Teaching teachers about childhood cancer: the effects of a web-based training program. Journal Of Pediatric Hematology/Oncology: Official Journal Of The American Society Of Pediatric Hematology/Oncology, 25(11), 729-733. Eiser, C. (1991). Cognitive deficits in children treated for leukaemia. Arch Dis Child, 66(1), 164-168. Eiser, C. (1998). Practitioner review: long-term consequences of childhood cancer. J Child Psychol Psychiatry, 39(5), 621-633. Eiser, C , & Vance, Y . H . (2002). Implications of cancer for school attendance and behavior. Med Pediatr Oncol, 38(5), 317-319. Fogarty, K . , Volonino, V . , Caul, J., Rongey, J., Whitman, B . , O'Connor, D . , et al. (1988). Acute leukemia. Learning disabilities following C N S irradiation. Clin Pediatr (Phila), 27(11), 524-528. 65 Harila-Saari, A . H . , Lahteenmaki, P. M . , Pukkala, E . , Kyyronen, P., Lanning, M . , & Sankila, R. (2007). Scholastic Achievements of Childhood Leukemia Patients: A Nationwide, Register-Based Study. J Clin Oncol, 25(23), 3518-3524. Haupt, R., Fears, T. R., Robison, L . L . , M i l l s , J. L . , Nicholson, H . S., Zeltzer, L . K . , et al. (1994). Educational attainment in long-term survivors of childhood acute lymphoblastic leukemia. Jama, 272(18), 1427-1432. Humpl, T., Fritsche, M . , Bartels, U . , & Gutjahr, P. (2001). Survivors of childhood cancer for more than twenty years. Acta Oncol, 40(1), 44-49. Jannoun, L . , & Chessells, J. M . (1987). Long-term psychological effects of childhood leukemia and its treatment. Pediatr Hematol Oncol, 4(A), 293-308. Kadan-Lottick, N . S., Robison, L . L . , Gurney, J. G . , Neglia, J. P., Yasui , Y . , Hayashi, R., et al. (2002) . Childhood Cancer Survivors' Knowledge About Their Past Diagnosis and Treatment: Childhood Cancer Survivor Study. JAMA, 257(14), 1832-1839. Kingma, A . , Mooyaart, E . L . , Kamps, W . A . , Nieuwenhuizen, P., & Wilmink, J. T. (1993). Magnetic resonance imaging of the brain and neuropsychological evaluation in children treated for acute lymphoblastic leukemia at a young age. Am J Pediatr Hematol Oncol, 15(2), 231-238. Kingma, A . , Rammeloo, L . A . , van Der Does-van den Berg, A . , Rekers-Mombarg, L . , & Postma, A . (2000). Academic career after treatment for acute lymphoblastic leukaemia. Arch Dis Child, 82(5), 353-357. Kingma, A . , van Dommelen, R. I., Mooyaart, E . L . , Wilmink, J. T., Deelman, B . G . , & Kamps, W . A . (2001). Slight cognitive impairment and magnetic resonance imaging abnormalities but normal school levels in children treated for acute lymphoblastic leukemia with chemotherapy only. J Pediatr, 139(3), 413-420. Koch , S. V . , Kejs, A . M . T., Engholm, G. , Johansen, C , & Schmiegelow, K . (2004). Educational attainment among survivors of childhood cancer: a population-based cohort study in Denmark. British Journal of Cancer, 91(5), 923-928. Lahteenmaki, P. M . , Harila-Saari, A . , Pukkala, E . I., Kyyronen, P., Salmi, T. T., & Sankila, R. (2007). Scholastic achievements of children with brain tumors at the end of comprehensive education: A nationwide, register-based study. Neurology, 69(3), 296-305. Langeveld, N . E . , Ubbink, M . C , Last, B . F. , Grootenhuis, M . A . , Voute, P. A . , & De Haan, R. J. (2003) . Educational achievement, employment and l iving situation in long-term young adult survivors of childhood cancer in the Netherlands. Psychooncology, 12(3), 213-225. Madan-Swain, A . , & Brown, R. T. (1991). Cognitive and psychosocial sequelae for children with acute lymphocytic leukemia and their families. Clinical Psychology Review, 11(3), 267-294. Mitby, P. A . , Robison, L . L . , Whitton, J. A . , Zevon, M . A . , Gibbs, I. C , Tersak, J. M . , et al. (2003). Utilization of special education services and educational attainment among long-term survivors of childhood cancer: a report from the Childhood Cancer Survivor Study. Cancer, 97(A), 1115-1126. Moore, I. M . , Kramer, J. H . , Wara, W. , Halberg, F. , & Ab l in , A . R. (1991). Cognitive function in children with leukemia. Effect o f radiation dose and time since irradiation. Cancer, 68(9), 1913-1917. Mulhern, R. K . , & Palmer, S. L . (2003). Neurocognitive late effects in pediatric cancer. Curr Probl Cancer, 27(A), 177-197. 66 National Cancer Pol icy Board (Ed.). (2003). Childhood Cancer Survivorship: Improving Care and Quality of Life. Washington, D C : National Academic Press. Patenaude, A . F. , & Kupst, M . J. (2005). Psychosocial functioning in pediatric cancer. J Pediatr Psychol, 30(1), 9-27. Precourt, S., Robaey, P., Lamothe, I., Lassonde, M . , Sauerwein, H . C , & Moghrabi, A . (2002). Verbal cognitive functioning and learning in girls treated for acute lymphoblastic leukemia by chemotherapy with or without cranial irradiation. Dev Neuropsychol, 21(2), 173-195. Raymond-Speden, E . , Tripp, G . , Lawrence, B . , & Holdaway, D . (2000). Intellectual, neuropsychological, and academic functioning in long-term survivors of leukemia. J Pediatr Psychol, 25(2), 59-68. Rubenstein, C. L . , Varni , J. W. , & Katz, E . R. (1990). Cognitive functioning in long-term survivors of childhood leukemia: a prospective analysis. J Dev Behav Pediatr, 11(6), 301-305. Seaver, E . , Geyer, R., Sulzbacher, S., Warner, M . , Batzel, L . , Milstein, J., et al. (1994). Psychosocial adjustment in long-term survivors of childhood medulloblastoma and ependymoma treated with craniospinal irradiation. Pediatr Neurosurg, 20(4), 248-253. Smibert, E . , Anderson, V . , Godber, T., & Ekert, H . (1996). Risk factors for intellectual and educational sequelae of cranial irradiation in childhood acute lymphoblastic leukaemia. Br J Cancer, 73(6), 825-830. Syndikus, I., Tait, D . , Ashley, S., & Jannoun, L . (1994). Long-term follow-up of young children with brain tumors after irradiation. Int JRadiat Oncol Biol Phys, 30(4), 781 -787. von der Weid, N . (2001). Late effects in long-term survivors of A L L in childhood: experiences from the S P O G late effects study. Swiss Med Wkly, 737(13-14), 180-187. 67 

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