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Effects of Electrical Stimulation,  Exercise Training & Motor Skills  Training on Strength for Children  with Meningomyelocele: A Systematic Review Liese Dagenais ‐ Erin Lahay ‐ Kailey Stueck ‐ Erin White – Lindsay Williams Supervisor ‐ Susan Harris Acknowledgements Susan Harris Supervisor and mentor Charlotte Beck Reference Librarian Outline • Introduction • Purpose • Methods • Results • Discussion  Introduction Spina bifida • Congenital abnormality • Incomplete closure of the spinal     column1 • ~1/1000 births annually2 • 94% classified as meningomyelocele Spina bifida Meningomyelocele • Results in nerve damage below                      level of lesion1 • Common impairments1 – Muscle weakness  – Muscle paralysis – Sensory impairments – Bowel & bladder                      dysfunction Interventions • Multidisciplinary approach to  treatment is ideal • Physical therapy plays a key role1 • Interventions include1 – Muscle strengthening – Positioning – Improving postural control – Increasing independence  & mobility1 Existing Research • Limited evidence regarding efficacy  of physical therapy interventions for  children with meningomyelocele • Systematic review: Mazur JM & Kyle S,  20043 – Inconsistent results – No conclusive findings – Further research necessary Cerebral Palsy Research • Current & high quality articles  available • Systematic review: Dodd KJ et al.,  20024 – 23 relevant articles – 11 high quality Purpose To provide an overview of the existing  research regarding the effects of  electrical stimulation, exercise training  and motor skills training on muscle  strengthening for children with  meningomyelocele Methods Literature Search •Electronic databases Limits: English language •Pubmed •Embase •Cinahl •Medline •PEDro •CENTRAL  •DARE •CIRRIE •Cochrane database •Hand Search  Reference lists of selected articles Spina Bifida MeningomyeloceleSpinal Dysraphism Physical therapy Physical therapy modalitiesRehabilitation ExerciseElectrical stimulation Functional mobility Muscle strength Gait   /   Ambulation Search Terms Inclusion & Exclusion Criteria • Inclusion – Participants diagnosed with  meningomyelocele – Participants 21 years of age and under – Study involves physical therapy intervention   • Exclusions – Book chapters  – Duplicate publications – Focus on surgical techniques, orthoses, bowel  & bladder function, cognition or scoliosis • Resulting 11 studies extremely     heterogeneous  • Therefore… STRENGTH  was used as an    outcome measure Inclusion & Exclusion Criteria PICO • Population: participants 21 years of age        and under with a diagnosis of  meningomyelocele • Intervention: Electrical stimulation,  exercise training & motor skills training • Comparison: N/A •Outcome: Strength Studies included in Review  (N =6) Failed to meet full text inclusion/ exclusion criteria  (N=9) Retrieved for Evaluation  (N = 20) Obtained by Hand Searching  (N = 2) After Screening Abstracts  (N = 18) Failed to meet abstract inclusion/ exclusion criteria  (N=120) After Screening Titles  (N = 138) Failed to meet title inclusion/ exclusion criteria  (N=160) Did not use strength as an outcome measure  (N=5) After Screening Full Text  (N = 11) Potentially Relevant Citations  (N = 298) Full Text Exclusions • 5 did not use ‘strength’ as outcome  measure • 3 duplicates  • 2 did not have a relevant physical therapy  intervention • 1 focused on scoliosis • 1 did not have a diagnosis of  meningomyelocele • 1 book chapter • 1 participants did not meet age  requirement Included Studies 6  Studies 2  Electrical  Stimulation 3 Exercise  Training 1  Motor Skills  Training AACPDM Levels of Evidence4 Level Intervention (Group) Studies I Systematic Review of randomized controlled trials (RCT’s) Large RCT (with narrow confidence intervals) (n>100)  II Smaller RCT’s (with wider confidence intervals) (n<100) Systematic Reviews of cohort studies “Outcomes research” (very large ecologic studies) III Cohort studies (must have concurrent control group) Systematic Reviews of Case Control Studies IV Case series Cohort study without concurrent control group (e.g. with historical  control group) Case‐control Study V Expert Opinion  Case Study or report Bench research  Expert opinion based on theory or physiologic research Common sense/anecdotes Level of Agreement = 100% AACPDM Quality Assessment4 1. Was inclusion criteria well described & followed? 2. Were interventions well described & was there  adherence?  3. Were measures clearly described, valid & reliable? 4. Were there blind assessments? 5. Were appropriate statistical evaluations conducted &  reported? 6. Were dropouts reported and less than 20%?  7. Were appropriate methods for controlling  confounding variables & limiting potential biases  used?  Level II studies Case Study Quality Assessment5 Level IV & V studies 1. Was the purpose clearly stated? 2. Was the hypothesis clearly stated? 3. Were participants described in sufficient detail? 4. Were interventions & treatment settings described  in sufficient detail? 5. Were measures clearly described, valid & reliable? 6. Was the effect size clinically important? 7. Were limitations of the study identified &  discussed? Study Quality Study Level 1 2 3 4 5 6 7 Total AACPDM Quality Assessment4 Karmel‐Ross et al.6 II √ √ √ √ 4 Andrade et al.8 II √ √ √ √ √ 5 Case Study Quality Assessment5 O’Connell &  Barnhart9 IV √ √ √ √ √ 5 Mazliah et al.7 IV √ √ √ √ √ √ 6 Rapport & Bailey10 V √ √ √ √ √ 5 Manella & Varni11 V √ √ √ √ 4 Level of Agreement = 83%Strong = score of 6‐7 Moderate = score of 4‐5  Weak = score of ≤ 3 Data Extraction & Analysis • Standardized data extraction form • Summary tables • Descriptive synthesis method of data  analysis – Evaluating participants, interventions,  outcomes – Determine if findings can be generalized – Limitations investigated Results Electrical Stimulation Authors Therapy  Interventions Karmel‐Ross et  al.6 PRE TEST ‐ POST  TEST CONTROL Level II Quality 4/7 Applied to  quadriceps  femoris unilaterally ‐30 minute  sessions 6x/wk  for 8 wks Applied to  quadriceps  femoris bilaterally ‐1‐2 hour sessions  daily for 6  months Mazliah et al.7 ONE GROUP PRE  TEST – POST  TEST Level IV  Quality 6/7 Control  Intervention Sample Contralateral limb N=5 (2 males; 3  females) ‐Children with  Spina Bifida; lesion  at level L2‐3 ‐5‐21 years N/A N=3  ‐ Children with  lumbar MM  ‐Knee flexion  contractures >15 ° ‐ 9‐12 years Electrical Stimulation: Results Study Outcome of  Interest Measure Result Karmel Ross et  al.6 Quadriceps  femoris muscle  strength Maximum isometric  voluntary knee  extension torque 2/5 statistically  significant  improvement Mazliah et al.7 Quadriceps  femoris muscle  strength Isometric Torque  measurements  2/3 improved Exercise Training Authors Therapy  Intervention Control  Intervention Andrade et al.8 PRE TEST ‐ POST  TEST CONTROL Level II Quality 5/7 Children not  attending  exercise  program N/A N/A O’Connell &  Barnhart9 ONE GROUP PRE  TEST – POST TEST Level IV Quality 5/7 UE strengthening ‐30 min 3x/wk for 9  wks N=6  ‐ MM (N=3); lesion  below T8  ‐ Children diagnosed  with CP (N=3)  ‐ 4‐16 years Rapport &  Bailey10 SINGLE SUBJECT Level V Quality 5/7 Fine/gross motor  ‐Clinic: 90 min  1x/wk for 6 wks ‐Home: 30 min/day  5x/wk for 56 wks N=1 (male) ‐ Child with MM  ‐ 8.5 years  Sample Aerobic and UE  strengthening ‐1 hr/wk for 10 wks N=13 (7 males; 6  females) ‐ MM; lesion below T6 ‐ 8‐13 years Exercise Training: Results Study Outcome of  Interest Measure Result Andrade  et al.8 Strength Isometric tests using  hand held  dynamometer Ss (p<0.01) O’Connell  &  Barnhart9 Wheelchair  propulsion 6 repetition maximum  Ss (p= .018‐ .031) Rapport &  Bailey10 UE fine motor UE gross motor OSCO Pinchmeter Dynamometer grip  strength All Improved  All Improved Motor Skills Training Authors Therapy Intervention Manella &  Varni11 SINGLE  SUBJECT Level V Quality 4/7 Functional activities  & behavioural  therapy ‐Clinic: 30 min 4 wks ‐Home: 30 min daily ‐Follow up:  1x/month for 5  months N/A N=1 (female) ‐Child with MM @ L3 ‐ 5 years Control  Intervention Sample Motor Skills Training: Results Study Outcome of  Interest Measure Result Manella &  Varni11 Independence  in motor skills Quadriceps manual  muscle test  Improved grade 3  to grade 4  bilaterally Discussion This systematic review concludes there is a positive trend towards increasing muscle strength using electrical stimulation, exercise training and motor skills training for children with meningomyelocele. Electrical Stimulation Karmel‐Ross et al.6 Mazliah et al.7 •Level II •Functional activities  during e‐stim •Improvements in gait •Health & instructional issues •Level IV •Longer e‐stim application  & study duration  •Sedentary during e‐stim •No improvements in gait •Possible low adherence or  ineffective application Exercise Training Andrade et al.8 O’Connell &  Barnhart9 Rapport &  Bailey10 •Level II •10 week program •Resistance  training •Level IV •9 week program •Resistance  training •Level V •56 week program •Functional  activities Motor Skills Training Manella & Varni12 •Task specific training •Behavioral therapy Limitations • Limited search to English  • Limited search to published articles • Did not extensively search grey  literature • Descriptive synthesis vs.  meta‐analysis • Small sample sizes • Dated studies • Lack of literature and low levels of  evidence Strengths of Review • Investigations of LE & UE • Relating strength gains to function • Extensive literature search • Monthly updates • 5 reviewers to limit bias • High inter‐rater reliability  • Spina bifida expert  Future Research • Effects of electrical stimulation on  ambulation ability6 & knee flexion                contractures7 • Effects of compliance,  motivation, education  & parental involvement  • Optimal treatment  protocols & duration Conclusion Although this review supports               aspects of physical therapy  interventions, the literature is out of  date and scarce.   In order to ensure  physical therapy is  delivered in an  effective and efficient  manner, further  research is necessary http://www.youtube.com/watch?v=EnCx64 tyJHY&mode=related&search= References (1) Campbell S, Vander Linden D, Palisano R. Physical therapy for children. 3rd ed. Philadelphia: Saunders Elsevier Inc; 2006. (2) National Dissemination Center for Children with Disabilities.  Spina Bifida Fact Sheet; 2004. [Cited 2007 May 18]. Available from: http://www.nichcy.org/pubs/factshe/ fs12txt.htm. (3) Mazur JM & Kyle S. Efficacy of bracing the lower limbs and ambulation training in children with myelomeningocele. Developmental Medicine & Child Neurology. 2004;46(5):352-356. (4) Dodd KJ, Taylor NF, Damiano DL. A systematic review of the effectiveness of strength-training programs for people with cerebral palsy. Arch Phys Med Rehabil. 2002 Aug;83(8):1157-64. (5) O’Donnel M., Darrah J, Adams R, Butler C, Roxborough L, Damiano D. AACPDM methodology to Develop Systematic Reviews of Treatment Interventions; 2004 [Cited 2007 April 17]. Available from: http://www.aacpdm.org/resources/systematicReviews Methodology.pdf. (6) Dean E, Arscott S, Desaulles P, Hughes K, Kotzo S, Preto R. The effect of manual therapy on range of motion following lateral ankle sprains: a systematic review. 2006. (7) Center for reviews and dissemination. CRD Report 4 (2nd edition); March 2001 [Cited 1007 April 17]. Available from: http://www.york.ac.uk/inst/crd/report4.htm. (8) Karmel-Ross K, Cooperman DR, Van Doren CL. The effect of electrical stimulation on quadriceps femoris muscle torque in children with spina bifida. Physical Therapy. 1992 Oct;72(10):723-730. (9) Mazliah J, Naumann S, White C, et al. Electrostimulation as a means of decreasing knee flexion contractures in children with spina bifida. Proceedings of the 6th annual conference on Rehabilitation Engineering 1983:63-65. (10) Andrade CK, Kramer J, Garber M, Longmuir P. Changes in self-concept, cardiovascular endurance and muscular strength of children with spina bifida aged 8 to 13 years in response to a 10-week physical-activity programme: a pilot study. Child Care Health Development 1991;17(3):183-196. (11) O'Connell DG, Barnhart R. Improvement in wheelchair propulsion in pediatric wheelchair users through resistance training: A pilot study. Archives of Physical Medicine & Rehabilitation 1995;76(4):368-372. (12) Rapport MD, Bailey JS. Behavioral physical therapy and spina bifida: a case study. J.Pediatr.Psychol. 1985 Mar;10(1):87-96. (13) Manella K, Varni J. Behavioral treatment of ambulatory function in a child with myelomeningocele: a case report. Physical Therapy 1984;64(10):1536- 1539. (14) Levels of evidence and grades of recommendation. [Cited 2007 July 18]. Available from: http://www.minervation.com/cebm2/docs/levels.html. (15) Falk B, Tenenbaum G. The effectiveness of resistance training in children. A meta-analysis. Sports Med. 1996;22:176–186. 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