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Screening for congenital dislocation of the hip in infants in British Columbia Fulton Fraser, Jane 1982

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S C R E E N I N G F O R C O N G E N I T A L D I S L O C A T I O N O F T H E H I P I N I N F A N T S I N B R I T I S H C O L U M B I A b y J A N E J F U L T O N F R A S E R B . H . E c , T h e U n i v e r s i t y o f B r i t i s h C o l u m b i a , 1969 A T H E S I S S U B M I T T E D I N P A R T I A L F U L F I 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 O F S C I E N C E i n T H E F A C U L T Y O F G R A D U A T E S T U D I E S ( D e p a r t m e n t o f H e a l t h C a r e a n d E p i d e m i o l o g y ) We a c c e p t t h i s t h e s i s a s c o n f o r m i n g t o t h e r e q u i r e d s t a n d a r d T H E U N I V E R S I T Y O F B R I T I S H C O L U M B I A M a r c h , 1 9 8 2 ( c ) J a n e F u l t o n F r a s e r , 1982 In presenting t h i s thesis i n p a r t i a l f u l f i l m e n t of the requirements for an advanced degree at the University of B r i t i s h Columbia, I agree that the Library s h a l l make i t f r e e l y available for reference and study. I further agree that permission for extensive copying of t h i s thesis for scholarly purposes may be granted by the head of my department or by his or her representatives. I t i s understood that copying or publication of t h i s thesis for f i n a n c i a l gain s h a l l not be allowed without my written permission. Department of The University of B r i t i s h Columbia 1956 Main Mall Vancouver, Canada V6T 1Y3 Pate klTth , /90± DE-6 (3/81) A B S T R A C T C o n g e n i t a l D i s l o c a t i o n o f t h e H i p i n i n f a n t s i s a t o p i c . o f s c r e e n i n g i n t e r e s t i n B r i t i s h C o l u m b i a . W h e n a c a s e o f C o n g e n i t a l D i s l o c a t i o n o f t h e H i p i s n o t d i a g n o s e d a n d t r e a t e d i n t h e n e o n a t a l p e r i o d , t h e i n f a n t m a y l a t e r r e q u i r e s u r g i c a l c a r e . I n B r i t i s h C o l u m b i a , 94% o f s u r g i c a l c a s e s f o r C o n g e n i t a l D i s l o c a t i o n o f t h e H i p c o m e f r o m u n s c r e e n e d c o m m u n i t i e s w h i c h p r o d u c e o n l y 66% o f t h e l i v e b i r t h s i n t h e p r o v i n c e . A c o s t - m i n i m i z a t i o n a n a l y s i s o f s c r e e n i n g a n d c o n s e r v a t i v e t r e a t m e n t v e r s u s n o s c r e e n i n g a n d s u r g e r y s h o w s t h a t t h e c o s t o f s u r g i c a l c a r e i s m o r e t h a n t h r e e t i m e s g r e a t e r . T h i s t h e s i s p r o p o s e s t h a t t h e p r o v i n c e e x t e n d s c r e e n i n g s e r v i c e s t o a l l O b s t e t r i c a l U n i t s i n B r i t i s h C o l u m b i a a n d t r a i n p h y s i c i a n s a n d n u r s e s t o d o t h e s c r e e n i n g . A s u g g e s t i o n i s a l s o m a d e t o i n c o r p o r a t e h i p s c r e e n i n g i n t o a c o m p r e h e n s i v e n e o n a t a l d e v e l o p m e n t a l s c r e e n i n g p a c k a g e . i i i TABLE OF CONTENTS A b s t r a c t i i L i s t o f Tables i v Acknowledgement v CHAPTER 1. C o n g e n i t a l D i s l o c a t i o n o f the Hip i n I n f a n t s 1 CHAPTER 2. Screening Programs i n B r i t i s h Columbia 11 9 Appendix f o r Chapter 2 16 CHAPTER 3. The Scope of the CDH Problem i n B r i t i s h Columbia 18 Appendix f o r Chapter 3 . . . . . . . 22 CHAPTER 4. A C o s t - m i n i m i z a t i o n A n a l y s i s o f Screening f o r C o n g e n i t a l D i s l o c a t i o n of the Hip i n Newborns 27 Appendix f o r Chapter 4 37 CHAPTER 5. P l a n n i n g a P r o v i n c i a l Program f o r CDH 4 2 B i b l i o g r a p h y 4 8 i v LIST OF TABLES TABLE 1.1 Summary of Incidence Rates o f CDH 4 2.1 I d e n t i f i c a t i o n of CDH i n B.C. H o s p i t a l s w i t h No Screening Program: 1967-1971 14 2.2 E f f e c t o f B i r t h P r e s e n t a t i o n on Incidence 17 2.3 Seasonal V a r i a t i o n i n the number o f Cases 17 4.1 Summary of Screening Costs a t Grace H o s p i t a l . . . .32 4.2 Summary of Treatment Costs f o r Screened I n f a n t s w i t h CDH 32 4.3 B r a c i n g Costs 33 4.4 T o t a l S c r e e n i n g Program and Treatment Costs . . . .33 4.5 T o t a l and Average Costs of S u r g i c a l Care f o r 1000 Unscreened I n f a n t s 34 V ACKNOWLEDGEMENTS I would l i k e to express my a p p r e c i a t i o n to those whose e f f o r t on my b e h a l f made t h i s t h e s i s p o s s i b l e . F i r s t , to my Chairman, Dr. Ned G l i c k , who guided the t h e s i s process, s i n c e r e thanks. I am a l s o g r a t e f u l to the Committee members, DrA Sam Sheps and Dr. Dick Splane f o r t h e i r d i r e c t involvement w i t h the t h e s i s content. The f o l l o w i n g F a c u l t y o f the U n i v e r s i t y of B r i t i s h Columbia were i n s t r u m e n t a l i n the ideas and i n f o r m a t i o n gathered i n t h i s t h e s i s : Dr. T e r r y Anderson, Dr. M o r r i s Barer, Dr. Fred Bass, Dr. Mike B e l l , Dr. B l a i r F u l t o n , and Dr. M i c h e l V e r n i e r . I am p l e a s e d t o acknowledge t e c h n i c a l a s s i s t a n c e from Mr. Ronnie S i z t o and Miss Cindy K r o n s t e i n . 1 CHAPTER 1 CONGENITAL DISLOCATION OF THE HIP IN INFANTS O b j e c t i v e s C o n g e n i t a l D i s l o c a t i o n o f the Hip (CDH) i s the most common d e f e c t seen i n newborn i n f a n t s , comprising 75% of a l l c o n g e n i t a l d e f e c t s ( 1 , 2 ) . I t can be i d e n t i f i e d by a simple p h y s i c a l examination ( 3 , 4 , 5 , 6 ) , and i f i d e n t i f i e d e a r l y can be t r e a t e d e f f e c t i v e l y i n a c o n s e r v a t i v e manner u s i n g t r i p l e d i a p e r s o r a simple brace ( 4 , 5 , 7 , 8 ) . Thus, CDH i s an i d e a l c o n d i t i o n to c o n s i d e r f o r l a r g e s c a l e s c r e e n i n g . E a r l y , c o n s e r v a t i v e t r e a t -ment has been suggested to reduce the r i s k o f i d i o p a t h i c Osteo-a r t h r i t i s o f the Hip r e l a t e d to CDH (34,35,36,37,38,39). T h i s t h e s i s examines e x i s t i n g s c r e e n i n g programs f o r CDH, the c o s t s a s s o c i a t e d w i t h s c r e e n i n g , and c o n s i d e r s the v i a b i l i t y o f extending i n f a n t h i p s c r e e n i n g to those areas o f the pr o v i n c e , accounting f o r about two - t h i r d s o f a l l b i r t h s , where l i t t l e o r no CDH s c r e e n i n g now e x i s t s . D e f i n i t i o n o f terms C o n g e n i t a l D i s l o c a t i o n o f the Hip, o r i g i n a l l y d e f i n e d by Hippocrates i n 400 B.C. ( 1 ) , r e f e r s to a complex a s s o c i a t i o n of c l i n i c a l l y d e f i n e d c h a r a c t e r i s t i c s o f the h i p , p r i m a r i l y i n new-borns, which may l e a d to a grad u a l upward displacement o f the femoral head ( 2 ) . A secondary r e a c t i o n a r i s i n g from muscle c o n t r a c t u r e f u r t h e r a l t e r s the weight b e a r i n g c h a r a c t e r i s t i c s o f the h i p , and towards the end of the f i r s t year of l i f e , i m pairs a-: c h i l d ' s a b i l i t y to develop a normal g a i t ( 3 , 5 ) . 2 S e v e r a l terms are used i n t e r c h a n g e a b l y to d e s c r i b e CDH. In t h i s t h e s i s , the term d y s p l a s i a w i l l be used to i d e n t i f y the whole range of degrees of s e v e r i t y of CDH. D y s p l a s i a r e f e r s to a s i t u a t i o n of primary i n s t a b i l i t y of the h i p i n the newborn t h a t can be i d e n t i f i e d by Barlow's t e s t . T h i s t e s t i s a p h y s i c a l m a n i p u l a t i o n of the head of the femur towards the p o s t e r i o r rim of the acetabulum. The degree of movement of the j o i n t i s dependent on the l a x i t y o f the ligaments i n the j o i n t . At the m i l d end of the s c a l e of d y s p l a s i a s i s s u b l u x a t i o n . S u b l u x a t i o n r e f e r s to a s i t u a t i o n where c o n t a c t i s maintained between the femoral head "and the acetabulum, but the c o n t a c t between the a r t i c u l a r s u r f a c e s i s not normal. The j o i n t i s not reduced, nor i s i t completely i n p l a c e . Frank d i s l o c a t i o n means t h a t the head of the femur i s not i n the acetabulum and the s u r f a c e s of the j o i n t do not have any c o n t a c t . T h i s i s the most severe degree of d y s p l a s i a . The term C o n g e n i t a l D i s l o c a t i o n of the Hip, used here, r e f e r s to the whole range of degrees of s e v e r i t y of d y s p l a s i a of the h i p t h a t can be i d e n t i f i e d by the accepted s c r e e n i n g t e s t . When e a r l y d e t e c t i o n of CDH leads to c o n s e r v a t i v e treatment i n the neonate, the h i p d e f e c t can be c o r r e c t e d without s u r g i c a l i n t e r v e n t i o n (3,7,9,10). Untreated, CDH may cause s h o r t e n i n g of the l e g a s s o c i a t e d w i t h the d i s l o c a t e d h i p , l i m p i n g , and a l i m i t e d range of move-ment of the h i p . T o t a l body d i s t o r t i o n i s not uncommon i n severe cases. 3 E t i o l o g y S e v e r a l c h a r a c t e r i s t i c s are known to p l a c e an i n f a n t a t r i s k f o r CDH. The i n c i d e n c e of CDH has been shown i n many s t u d i e s to be h i g h e s t f o r i n f a n t s w i t h a Breech p r e s e n t a t i o n (2,3,5,6,7,9, 11,-17,26). The i n f a n t ' s h i p s , i n t h i s s i t u a t i o n , are h e l d i n an u n s t a b l e p o s i t i o n ' i n u t e r o ' s i n c e the t h i g h s and knees are f u l l y extended. In a Vertex p r e s e n t a t i o n the baby's t h i g h s are f l e x e d and s l i g h t l y abducted, p r o v i d i n g a s t a b l e h i p p o s i t i o n . D e l i v e r y of a baby w i t h Breech p r e s e n t a t i o n can a l s o d i s -l o c a t e h i p s t h a t were merely subluxed ' i n u t e r o ' . Thus, i t i s recommended t h a t abduction of the h i p s be maintained f o r a t l e a s t s e v e r a l days a f t e r b i r t h f o r these i n f a n t s (2,8,11,12,16,35). S t u d i e s of CDH i n i n f a n t s c o n s i s t e n t l y show an i n c i d e n c e three t o f o u r times h i g h e r i n females than i n males (4,5,7,9,11). Von Rosen (11) has suggested t h a t hormonal i n f l u e n c e s s i m i l a r to the ones t h a t cause l a x i t y of the p e l v i c j o i n t s i n the mother d u r i n g the l a s t weeks of pregnancy cause j o i n t l a x i t y i n the i n f a n t , and t h a t t h i s e f f e c t i s more l i k e l y i n a female i n f a n t . On the same b a s i s as Von Rosen, Wynne-Davies (6), McKibbon (8), and C z e i z e l (10) suggest t h a t l a x i t y of the ligaments i n the h i p j o i n t i s a c o n t r i b u t i n g f a c t o r i n CDH. Two of these authors (6,10) suggest t h a t ligamentous l a x i t y may be r e l a t e d to a shallow acetabulum, but a c a u s a l r e l a t i o n s h i p has not been e s t a b l i s h e d . Incidence Rates Table 1.1 summarizes the i n c i d e n c e r a t e s found i n s i x major h o s p i t a l s t u d i e s of d y s p l a s i a of the h i p i n newborns. 4 Comparisons of these s t u d i e s are d i f f i c u l t to make i n absolute terms because d i f f e r e n t degrees of d y s p l a s i a are c o n s i d e r e d i n each study, the p o p u l a t i o n s d i f f e r i n p l a c e and time, and one study compares d i f f e r e n t h o s p i t a l d i s t r i c t s (29). TABLE 1.1 SUMMARY OF INCIDENCE RATES OF CDH Author and Date Rate/1000 i n f a n t s screened Barlow,1962 (19) 14.9/1000 Von Rosen,1968 (11) 1.7/1000 Mi t c h e l l , 1 9 7 2 (33) 10.6/1000 Williamson,1972 (28) 2.3/1000 Artz,1975 (26) 13.3/1000 Jones,1977 (29) 2.3/1000 Barlow, i n S a l f o r d , England (19) , examined 9,289 i n f a n t s d u r i n g t h e i r f i r s t week of l i f e and found 159 abnormal h i p s i n 139 b a b i e s . D i s l o c a t e d h i p s o c c u r r e d 81 times and unstable h i p s an a d d i t i o n a l 78. When both d i s l o c a t e d and u n s t a b l e h i p s are i n c l u d e d as CDH ( h i p d y s p l a s i a ) the i n c i d e n c e i s 14.96 per 1000 b i r t h s . Von Rosen, i n Malmo, Sweden, (11), examined i n f a n t s i n the c i t y ' s one O b s t e t r i c a l department where 99% of a l l i n f a n t s i n 5 Malmo are born. Each c l i n i c a l d i a g n o s i s of CDH was v e r i f i e d by radiography, which l i k e l y the reason f o r the very low i n c i d e n c e . Radiography i s on l y capable of i d e n t i f y i n g the most severe CDH, e l i m i n a t i n g a l l other m i l d e r d y s p l a s i a s from the numbers of p o s i t i v e diagnoses. In 24,000 examinations the i n c i d e n c e was 1.7 per 1000 i n f a n t s . M i t c h e l l (33) conducted a study of CDH i n i n f a n t s i n a l a r g e m a t e r n i t y h o s p i t a l i n Edinburgh, S c o t l a n d . A t o t a l o f 31,961 i n f a n t s were screened by P e d i a t r i c i a n s and those i n f a n t s w i t h a p o s i t i v e t e s t were checked by an Orthopedic Surgeon. The baby's h i p s were c l a s s i f i e d as 'luxated' i f d i s l o c a t i o n was diagnosed, and as 'unstable' i f the j o i n t was l a x . In the study p o p u l a t i o n 3 per 1000 b i r t h s (100 i n f a n t s ) had l u x a t e d h i p s r e q u i r i n g a brace or s p l i n t s . An a d d i t i o n a l 126 i n f a n t s , or 3.9 per 1000 b i r t h s had a d i a g n o s i s of unstable h i p s . The study a l s o found 123 i n f a n t s , 3.7 per 1000 b i r t h s , who had un s t a b l e or l u x a t e d h i p s a t b i r t h t h a t became s t a b l e before d i s c h a r g e . The t o t a l number of i n f a n t s found w i t h a p o s i t i v e t e s t f o r d y s p l a s i a a t s c r e e n i n g was 10.6 per 1000 b i r t h s . In B e l f a s t , Northern I r e l a n d , W i l liamson (2 8) examined data c o l l e c t e d i n two ma t e r n i t y h o s p i t a l s where the O r t o l a n i t e s t was used by a v a r i e t y o f d i f f e r e n t p r o f e s s i o n a l s . (The O r t o l a n i t e s t i s s i m i l a r t o the Barlow t e s t , but the examiner l i s t e n s f o r the h i p to 'clunk' as i t d i s l o c a t e s . ) Each p o s i t i v e case was f o l l o w -ed by an Orthopedic Surgeon. One u n i t recorded 37 cases i n 28,740 b i r t h s or 1.3 per 1000, while the other had 42 cases i n 6,100 b i r t h s or 6.9 per 1000. The average i n c i d e n c e i n the combined u n i t s i n the study was 2.3 per 1000 b i r t h s . 6 T h e B e l f a s t s t u d y a l s o i d e n t i f i e d t h e r a t e o f d i a g n o s e s b e y o n d t h e n e o n a t a l p e r i o d a s 0 . 2 p e r 1 0 0 0 i n t h e f i r s t u n i t , a n d 1 . 9 p e r 1 0 0 0 i n t h e s e c o n d . T h i s i s t h e o p p o s i t e o f w h a t w o u l d b e e x p e c t e d . A r t z e t a l ( 2 6 ) e x a m i n e d t h e h i p s o f n e w b o r n s i n t h e New Y o r k C i t y H o s p i t a l . O f 2 8 , 4 2 4 b i r t h s i n s e v e n a n d o n e - h a l f y e a r s , 2 3 , 4 0 8 w e r e e x a m i n e d . A t o t a l o f 3 3 1 n e w b o r n s w e r e f o u n d w i t h h i p d y s p l a s i a : d i s l o c a t e d o r d i s l o c a t a b l e h i p s . T h e i n c i d e n c e r a t e o f 1 3 . 3 p e r 1 0 0 0 w a s b a s e d o n 3 1 2 o f t h e s e i n f a n t s w h o w e r e f u l l t e r m . J o n e s ( 2 9 ) h a s d e s c r i b e d a f i v e y e a r s e r i e s o f 2 9 , 3 3 6 b i r t h s i n N o r w i c h , . E n g l a n d . H a l f o f t h e s e b i r t h s o c c u r r e d i n h o s p i t a l , a n d h a l f i n t h e r u r a l d i s t r i c t i n c l u d i n g 5 , 4 4 8 b o r n a t h o m e . I n f a n t s w i t h h i p d y s p l a s i a w e r e r e f e r r e d t o O r t h o p e d i c S u r g e o n s . A s i n t h e V o n R o s e n s t u d y , c o n f i r m a t i o n o f C D H w a s m a d e u s i n g r a d i o g r a p h s . T h i s m e t h o d o f d i a g n o s i s i s a b l e t o i d e n t i f y o n l y t h e m o s t s e v e r e c a s e s o f d y s p l a s i a , s o f e w e r c a s e s w o u l d b e e x p e c t e d . H o s p i t a l d o c t o r s w e r e s h o w n t o d e t e c t 2 . 3 c a s e s p e r 1 0 0 0 b i r t h s , a n d r u r a l d o c t o r s a n d m i d w i v e s 1 . 3 p e r 1 0 0 0 b i r t h s . S t u d i e s a c c e p t i n g a w i d e r a n g e o f d e g r e e s o f d y s p l a s i a h a d h i g h e r i n c i d e n c e r a t e s o f C D H t h a n t h o s e s t u d i e s u s i n g r a d i o -g r a p h s a s a c o n f i r m a t i o n o f d y s p l a s i a . S p o n t a n e o u s R e c o v e r y S p o n t a n e o u s r e c o v e r y r e f e r s t o d e c r e a s i n g j o i n t l a x i t y i n i n f a n t s a s t h e y m a t u r e ( 1 9 , 2 6 ) . T h e n u m b e r o f i n f a n t s i n a n e w b o r n p o p u l a t i o n w i t h i d e n t i f i a b l e d y s p l a s i a d e c r e a s e s w i t h a g e ( i n d a y s ) o f t h e i n f a n t . 7 Barlow (19) found t h i s recovery r a t e to be 5 8% i n h i s study; B e l l and T r e d w e l l (7) experienced a r a t e o f 80% i n t h e i r study; and Fredensborg and N i l l s o n (27) found a spontaneous recovery r a t e o f 90%. A l l three s t u d i e s compared i n f a n t s under one week of age w i t h those o l d e r than one week. These r a t e s of recovery w i l l a f f e c t the i n c i d e n c e r a t e s found i n s c r e e n i n g programs f o r CDH depending on the age of the i n f a n t s a t the time o f t e s t i n g . Screening programs f o r neonates w i l l have much h i g h e r i n c i d e n c e r a t e s than programs f o r i n f a n t s as much as fo u r or f i v e days o l d e r . The Screening T e s t and Co n s e r v a t i v e Treatment The most wi d e l y accepted t e s t s f o r CDH are Barlow's t e s t (19) and the O r t o l a n i ' c l i c k ' (11). The s c r e e n i n g p h y s i c i a n or nurse g e n t l y adducts and abducts the i n f a n t ' s h i p s to determine the s t a b i l i t y of the j o i n t . I f the head of the femur s l i p s out of the acetabulum d u r i n g the t e s t , the i n f a n t i s diagnosed as having CDH. E a r l y c o n s e r v a t i v e treatment f o r CDH c o n s i s t s of g e n t l e abduction o f the h i p s w i t h t r i p l e d i a p e r s . A l l i n f a n t s with any degree of d y s p l a s i a are t r e a t e d f o r an average o f s i x weeks, and longer i f the h i p s remain unstable (7,11,19). For those cases o f d y s p l a s i a t h a t do not respond to t r i p l e d i a p e r s , a brace i s p r e s c r i b e d . The brace p l a c e s the baby's h i p s i n more secure abduction (15,19,22), and i t i s e a s i l y removed t o al l o w the mother to care f o r the i n f a n t . The average time an i n f a n t w i t h CDH i s braced i s three months, the brace i s w e l l t o l e r a t e d . The O r t h o t i c s departments of most h o s p i t a l s 8 have the a b i l i t y t o c o n s t r u c t these simple d e v i c e s (3). Treatment f o r 'Late' Cases I n f a n t s with CDH who are i d e n t i f i e d a f t e r the neonatal p e r i o d are termed ' l a t e ' cases. These c h i l d r e n u s u a l l y r e q u i r e s u r g i c a l treatment to c o r r e c t t h e i r d i s a b i l i t y (1,2,15,22,32). B r a c i n g i s not e f f e c t i v e i f i n i t i a t e d i n i n f a n t s over two months of age (35). S u r g i c a l treatment c o n s i s t s of two weeks of body t r a c t i o n , f o l l o w e d by e i t h e r open or c l o s e d r e d u c t i o n of the h i p . In a c l o s e d r e d u c t i o n , the femoral Head i s manipulated i n t o the acetabulum, f r e q u e n t l y w i t h the a s s i s t a n c e of an adductor tenotomy. The c h i l d i s then p l a c e d i n a h i p c a s t f o r 3 to 6 months, wi t h r e g u l a r c a s t changes at 6 week i n t e r v a l s . C h i l d r e n who r e q u i r e an open r e d u c t i o n have s u r g i c a l r e p a i r of the acetabulum, i n c l u d i n g a bone g r a f t i f necessary. The h i p i s u s u a l l y pinned i n p l a c e , and the c h i l d wears a c a s t f o r 6 weeks. At the end of t h i s time, the c h i l d i s r e h o s p i t a l i z e d f o r removal of the p i n . The outcome of s u r g i c a l i n t e r v e n t i o n i n terms of h i p f u n c t i o n i s c o n s i d e r e d to be l e s s s a t i s f a c t o r y than e a r l y c o n s e r v a t i v e treatment w i t h t r i p l e d i a p e r s or a brace (2,11,19). Surgery i s i n v a s i v e and p l a c e s the c h i l d a t r i s k f o r A n a e s t h e t i c death, i n f e c t i o n , and the p o s s i b i l i t y of f a i l u r e of the bone g r a f t or malalignment of the bones. Late h i p r e p a i r d e lays the p o t e n t i a l f o r growth to promote s t a b i l i z a t i o n of the h i p j o i n t as e a r l y as t r i p l e d i a p e r i n g does. 9 Bjerkreim (34) and Somerville (35) have suggested t h a t there i s a strong r e l a t i o n s h i p between CDH i n infancy and O s t e o a r t h r i t i s of the h i p i n middle l i f e . This process has been l i n k e d to avascular n e c r o s i s due to the trauma of surgery (36). C r i t e r i a of a U s e f u l Screening Program Si x common c r i t e r i a have been s e l e c t e d to determine whether screening f o r CDH can be j u s t i f i e d i n B r i t i s h Columbia (46,47,48,49). F i r s t , the c o n d i t i o n of i n t e r e s t should be considered a h e a l t h problem. I t i s known th a t CDH i s the most commonly seen c o n g e n i t a l defect (1,2), and i f undetected and untreated w i l l cause s i g n i f i c a n t d i s a b i l i t y (18). Second, the n a t u r a l h i s t o r y of the disease should be under-stood. The s i g n i f i c a n t r i s k f a c t o r s f o r CDH cannot be a l t e r e d : sex, genetic background, and b i r t h p r e s e n t a t i o n ; e l i m i n a t i n g the p o t e n t i a l f o r primary prevention. Consequently, the optimal management of CDH i s secondary prevention: e a r l y d e t e c t i o n and treatment of the d e f e c t . T h i r d , i n order to i d e n t i f y cases of the disease, the screening t e s t should be r e l i a b l e , acceptable to the person screened and have high p r e d i c t i v e value. Both the modified Barlow's t e s t (19) and the O r t o l a n i ' c l i c k 1 t e s t (11) f u l f i l the f i r s t two c r i t e r i a . The p r e d i c t a b i l i t y of the t e s t has been shown to surpass 85% as Lehmann's study i d e n t i f i e d 6 cases per 1000 l i v e b i r t h s , while 0.06 cases per 1000 l i v e b i r t h s were missed by screening and r e q u i r e d surgery l a t e r (23). 10 F o u r t h , e f f e c t i v e treatment should be a v a i l a b l e f o r a l l cases. E a r l y c o n s e r v a t i v e treatment w i t h t r i p l e d i a p e r s and, i f necessary, a brace, has proved e f f e c t i v e i n many s t u d i e s (3, 7,10) . F i f t h , s c r e e n i n g should be c o s t - e f f e c t i v e . In Chapter 4 t h i s t h e s i s p r o v i d e s a c o s t - m i n i m i z a t i o n a n a l y s i s o f s c r e e n i n g and e a r l y treatment compared to s u r g i c a l i n t e r v e n t i o n . S i x t h , the s c r e e n i n g t e s t should be a v a i l a b l e f o r a l l persons a t r i s k f o r the d i s e a s e . Chapter 5 c o n s i d e r s the v i a b i l i t y o f province-wide s c r e e n i n g f o r CDH i n i n f a n t s . Summary The value o f i n f a n t s c r e e n i n g l i e s i n i t s p o t e n t i a l to i d e n t i f y cases of CDH du r i n g the neonatal p e r i o d when conser-v a t i v e treatment y i e l d s e x c e l l e n t r e s u l t s . Screening thus becomes a mechanism f o r d r a m a t i c a l l y r e d u c i n g the need f o r s u r g i c a l i n t e r v e n t i o n by r e d u c i n g the number o f cases i d e n t i f i e d too l a t e f o r the use of non- i n v a s i v e techniques o f management. 11 CHAPTER 2 SCREENING PROGRAMS IN BRITISH COLUMBIA I n t r o d u c t i o n T h i s chapter examines those CDH s t u d i e s done i n B r i t i s h Columbia i n o rder to determine the i n c i d e n c e r a t e s t h a t c o u l d be expected from a province-wide s c r e e n i n g program. The number of cases o f CDH r e q u i r i n g surgery i n an unscreened p o p u l a t i o n are a l s o i d e n t i f i e d . The Grace H o s p i t a l Study B e l l and F r a s e r (9) conducted a cohort study of r i s k f a c t o r s i n neonates diagnosed w i t h CDH a t Grace H o s p i t a l i n Vancouver. One Orthopedic surgeon examined 16,00 8 i n f a n t s d u r i n g a f i v e year p e r i o d : January 1,19 75 to December 31,1979. Data were c o l l e c t e d on b i r t h weight, sex, age i n days a t the time o f examination, b i r t h p r e s e n t a t i o n , d i a g n o s i s of CDH, and h i p involvement. I d e n t i f i c a t i o n of e t h n i c o r i g i n ( O r i e n t a l and other) was a l s o made. To determine the e f f e c t of these v a r i a b l e s (without examining a l l normal c a s e s ) , a l l p o s i t i v e cases of CDH were p l a c e d i n t o a case group (2 32 c a s e s ) , and a computer-generated random number t a b l e was used to s e l e c t the same number of i n f a n t s from the group of 15,776 u n a f f e c t e d i n f a n t s . A follow-up time of 18 months was c o n s i d e r e d s u f f i c i e n t to d i s c o v e r the presence of any f a l s e - n e g a t i v e diagnoses among the screened i n f a n t s . The i n c i d e n c e of CDH i n the Grace H o s p i t a l p o p u l a t i o n of screened i n f a n t s was 14.6 per 1000. An estimated 2.2% of l i v e b i r t h s were missed by s c r e e n i n g . 12 In the O r i e n t a l group of 3201 i n f a n t s screened, only 5 showed signs of CDH, an incidence of 1.5 per 1000. This low r a t e i n d i c a t e s a reasonable b a s i s f o r f u r t h e r i n v e s t i g a t i o n . B e l l and F r a s e r (9) showed t h a t the only s t a t i s t i c a l l y s i g n i f i - - . cant d i f f e r e n c e between O r i e n t a l i n f a n t s and others.was, b i r t h weight, the O r i e n t a l i n f a n t s weighed l e s s . The r a t i o of females to males was 3:1 and i n f a n t s w i t h CDH had Breech p r e s e n t a t i o n 9 times more oft e n than i n f a n t s w i t h normal h i p s . No s t a t i s t i c a l l y d i f f e r e n c e was found between the cases and the cohort w i t h respect to b i r t h weight, season of b i r t h , or age at time of examination. (Table 2.2 and 2.3, Appendix to t h i s chapter.) These r i s k f a c t o r s have been i d e n t i f i e d i n other s t u d i e s (2,7,10,17,19,24,26) suggesting t h a t screening at Grace H o s p i t a l i s f i n d i n g CDH at s i m i l a r r a t e s and w i t h s i m i l a r patterns of r i s k c h a r a c t e r i s t i c s as other major s t u d i e s . The Vancouver General H o s p i t a l Study B e l l and Tredwell (7) have summarized 9% years of h i p screening i n 32,480'infants at the Vancouver General H o s p i t a l from March 1967 to the end of 1976. The authors estimated that 97% of a l l l i v e b i r t h s were screened. The p a t t e r n of CDH cases at the Vancouver General H o s p i t a l was g e n e r a l l y s i m i l a r to t h a t at Grace H o s p i t a l . The incidence of CDH i n the screened population was 9.9 per 1000, the r a t i o of females to males was 4:1, and the r e l a t i v e r i s k a s s o c i a t e d w i t h Breech p r e s e n t a t i o n was 5:1 compared to Vertex p r e s e n t a t i o n . In B e l l and Tredwell's experience (9) not a l l h i p surgery can be e l i m i n a t e d . The f a l s e - n e g a t i v e r a t e i n t h i s s e r i e s o f examinations was 1 i n 5000, a t o t a l o f 6 c h i l d r e n . The S t . Paul's H o s p i t a l Study Lehmann compiled data on the h i p s c r e e n i n g program a t St. Paul's H o s p i t a l f o r the years 1967 to 1971 (23). The study found an i n c i d e n c e r a t e o f 6.0 per 1000 l i v e b i r t h s , but no d e t a i l s were presented on r i s k f a c t o r s . In a d d i t i o n , Lehmann reviews the i n c i d e n c e r a t e s o f CDH throughout the p r o v i n c e . Although the data f o r t h i s study were c o l l e c t e d a decade ago, i t remains the b e s t comprehensive a n a l y s i s o f the s c r e e n i n g s i t u a t i o n i n t h i s p r o v i n c e . Table 2.1 summarizes the r a t e s r i d e n t i f i e d over the study p e r i o d . Cases o f CDH i n i n f a n t s born . i n ..hospitals where s c r e e n i n g was not system-a t i c o c c u r r e d i n 1.2 per 1000 l i v e b i r t h s diagnosed n e o n a t a l l y , w h i l e 1.4 cases per 1000 l i v e b i r t h s were diagnosed a f t e r the neonatal p e r i o d (1 month of age). Lehmann (2 3) estimates t h a t by 19 81 about o n e - t h i r d o f i n f a n t s born i n B r i t i s h Columbia r e c e i v e d h i p s c r e e n i n g . In terms o f g e o g r a p h i c a l d i s t r i b u t i o n , t h i s o n e - t h i r d are r e s i d e n t i n the Vancouver area. Lehmann recommends a s c r e e n i n g program i n a l l newborn n u r s e r i e s i n the p r o v i n c e , c o - o r d i n a t e d by r e g i o n a l Orthopedic Surgeons, u s i n g e i t h e r p h y s i c i a n s or nurses to adm i n i s t e r the t e s t . C o n s e r v a t i v e treatment o f a l l cases o f d y s p l a s i a i s j u s t i -f i e d s i n c e no c r i t e r i a are a v a i l a b l e to d i s t i n g u i s h those cases f o r whom treatment i s necessary, and those f o r whom i t i s not. i i TABLE 2.1 IDENTIFICATION OF CDH IN B.C. HOSPITALS WITH NO SCREENING PROGRAM: 19 67-1971 (23) HOSPITAL NUMBER OF LIVE BIRTHS NUMBER OF CDH ' S BIRTHS PER 1000 LIVE-Neonatal P e r i o d A f t e r 1 mo. Grace,Vancouver 13,121 0.7 1.2 Royal Columbian, New Westminster 8,724 3.6 1.7 Royal J u b i l e e , V i c t o r i a 8,251 0.7 1.6 Lio n s Gate,Vancouver 7,084 0.7 1.1 P r i n c e George Regional 7,081 0.7 1.1 Burnaby General 6,969 3.2 1.4 Royal Inland,Kamloops 4,924 0.6 0.6 St.Vincent's,Vancouver 4,511 0.7 2.2 V i c t o r i a General 4,486 0.7 0.9 Surrey Memorial 4,472 0.0 1.6 Richmond General 4,191 0.9 2.4 Nanaimo Regional General 3,377 5.9 1.8 Kelowna General 3,178 0.3 1.3 C h i l l i w a c k General 2,702 0.0 0.7 T r a i l R egional 1,996 1.0 1.0 Campbell R i v e r & D i s t r i c t 1,935 0.0 1.5 Maple Ridge 1,897 0.0 1.0 Powell R i v e r General 1,770 0.0 1.7 P e n t i c t o n General 1,574 0.6 0.6 Matsqui-Sumas-Abbotsford 1,331 0 . 0 4.5 TOTALS AND AVERAGES 93,574 1.2 1.4 15 T h i s t r e a t m e n t r e g i m e n a l s o h a s t h e p o t e n t i a l t o r e d u c e t h e r i s k o f i d i o p a t h i c O s t e o a r t h r i t i s o f t h e H i p a s s o c i a t e d w i t h t h e s u r g i c a l c o r r e c t i o n o f C D H , ( 2 3 ) ' . S u m m a r y T h e i n c i d e n c e r a t e s f o r C D H i n n e w b o r n s s c r e e n e d i n B r i t i s h C o l u m b i a a r e : 6 . 0 p e r 1 0 0 0 ( 2 3 ) , 9 . 9 p e r 1 0 0 0 ( 7 ) , a n d 1 4 . 6 p e r 1 0 0 0 ( 9 ) . T h e s e r e p o r t s p r o v i d e a r a n g e o f n u m b e r s o f c a s e s t h a t w o u l d b e f o u n d i n a s c r e e n i n g p r o g r a m . L e h m a n n ' s s t u d y i d e n t i f e d l e s s C D H p e r 1 0 0 0 l i v e b i r t h s t h a n d i d T r e d w e l l a n d B e l l ( 7 ) , o r B e l l a n d F r a s e r ( 9 ) . L e h m a n n ( 2 3 ) a l s o m i s s e d m o r e c a s e s o f C D H a t s c r e e n i n g w h i c h l a t e r r e q u i r e d s u r g e r y . A h i g h e r r a t e o f c a s e s o f C D H d u r i n g t h e n e o n a t a l p e r i o d m a y b e c o n s i d e r e d t o p r o v i d e m o r e e f f e c t i v e s c r e e n i n g s i n c e f e w e r c a s e s o f s u r g e r y w i l l b e r e q u i r e d . T r e d w e . l l a n d B e l l r e p o r t n o c a s e s o f s u r g e r y i n t h e i r s c r e e n e d p o p u l a t i o n s s i n c e 1 9 7 5 ( 7 ) . T h e s u m m a r y o f r a t e s o f d i a g n o s e s i n i n f a n t s o v e r 1 m o n t h o f a g e f r o m h o s p i t a l s w i t h o u t s c r e e n i n g ( T a b l e 2 1 . ( 2 3 ) ) ' o f f e r s a r e a s o n a b l e n u m b e r ( 1 . 4 p e r 1 0 0 0 l i v e i b i r t h s ) f o r c a l c u l a t i n g t h e c o s t o f p r o v i d i n g s u r g i c a l c a r e t o u n s c r e e n e d p o p u l a t i o n s . D r . T r e d w e l l s u g g e s t s t h a t 1 . 5 p e r 1 0 0 0 l i v e b i r t h s w i l l r e q u i r e s u r g i c a l c a r e i f t h e y a r e u n s c r e e n e d ( p e r s o n a l c o m m u n i -c a t i o n f r o m D r . T r e d w e l l , N o v e m b e r , 1 9 8 1 ) . A p p e n d i x f o r C h a p t e r 2 R e s u l t s f r o m t h e " G r a c e H o s p i t a l S t u d y 17 TABLE 2 . 2 EFFECT OF BIRTH PRESENTATION ON INCIDENCE (9) % f o r each group and (numbers) p r e s e n t a t i o n +CDH normal Vertex 74 . 1% (172) 95. 7% (222) Breech 19 .0% ( 44) 2. 2% ( 5) C a e s a r i a n (Vertex) 3 .9% ( 9) 1. 3% ( 3) C a e s a r i a n (Breech) 2 ( 5) 0. 9% ( 2) 100 .0% (232) 100. 0% (232) TABLE 2.3 SEASONAL VARIATION IN THE NUMBER OF CASES (9) % f o r each 3 month p e r i o d and (number) season +CDH normal w i n t e r 22. . 8 % (53) 28. 0% (65) s p r i n g 18. 1 9-, -L t5 (42) 22. 8% (53) summer 33. »6 % (78) 24. 6% (57) f a l l 21. . 1% (49) 24 . 6% (57) 100. . 0% (232) 100. 0% (232) X 2 =6.15 P - 0.20 18 CHAPTER 3 THE SCOPE OF THE CDH PROBLEM IN BRITISH COLUMBIA I n t r o d u c t i o n T h i s chapter e x p l o r e s the outcomes of not s c r e e n i n g f o r CDH i n h o s p i t a l s o u t s i d e Vancouver. The number of c h i l d r e n r e q u i r i n g surgery due to a l a t e d i a g n o s i s o f CDH are i d e n t i f i e d . These cases are then l i n k e d to t h e i r r e s i d e n c e by community to deter,-:'., mine the sources o f s u r g i c a l cases. Data Source The Research D i v i s i o n o f H o s p i t a l Programs i n the M i n i s t r y of Heal t h p r o v i d e d a summary of dis c h a r g e diagnoses o f CDH and the r e s i d e n c e by community o f each c h i l d who r e c e i v e d h o s p i t a l c a r e . The name of the c h i l d was not a v a i l a b l e f o r t h i s a n a l y s i s . Two h o s p i t a l s r e p o r t e d s u r g i c a l d i s c h a r g e s : C h i l d r e n ' s H o s p i t a l and the Vancouver General H o s p i t a l . The h o s p i t a l s r e p o r t i n g a dis c h a r g e d i a g n o s i s o f CDH i n neonates were: the Grace Hos>pital, S t . Paul's H o s p i t a l , and the Vancouver General H o s p i t a l . In a d d i t i o n , the M e d i c a l Records L i b r a r i a n o f the P r i n c e George Regional H o s p i t a l p r o v i d e d d i s c h a r g e data on treatment f o r CDH. Tha data r e p r e s e n t s the most r e c e n t year a v a i l a b l e : f i s c a l 1979 ( A p r i l 1,1979 to March 31, 1980). The d i s c h a r g e l i s t i n g s are i n the Appendix to t h i s chapter. 0 D e f i n i t i o n o f Terms A c h i l d may have one or more procedures l i s t e d w i t h the dis c h a r g e d i a g n o s i s o f CDH. These procedures are c a l l e d an 19 episode of care when they occur w h i l e the c h i l d i s h o s p i t a l i z e d d u r i n g a d i s t i n c t time p e r i o d . When a s e r i e s of episodes are l i n k e d t o gether, as they are i n the s u r g i c a l treatment of CDH, the c h i l d r e c e i v e s what i s termed a p a t t e r n of c are. These p a t t e r n s are d i s t i n c t . A c h i l d r e c e i v i n g an open r e d u c t i o n of the h i p can be d i f f e r e n t i a t e d from one r e c e i v i n g a c l o s e d r e d u c t i o n . The p a t t e r n s of care expected w i t h each procedure were used to determine, from the h o s p i t a l d i s c h a r g e l i s t i n g s , how many c h i l d r e n r e c e i v e d which treatment. As a r e s u l t , i t i s p o s s i b l e to d i s c o v e r how many c h i l d r e n were t r e a t e d , not j u s t the number of d i s c h a r g e s a t t r i b u t a b l e t o CDH. For example, the treatment p a t t e r n f o r a F t . S t . John i n f a n t i n c l u d e d : t r a c t i o n , osteotomy, b l o o d t r a n s f u s i o n , removal of a h i p p i n , and physiotherapy. By i d e n t i f y i n g the age of the c h i l d a t each d i s c h a r g e , the time lapse between procedures may be noted, c o n f i r m i n g the l i n k a g e of each procedure w i t h a p a r t i c u l a r case. Case Excluded from A n a l y s i s An examination of the coded data i n d i c a t e s t h a t one c h i l d who had a d i s c h a r g e d i a g n o s i s of CDH a c t u a l l y underwent a h e r n i a r e p a i r . As a r e s u l t of t h i s apparent i n a c c u r a c y i n the data, a coding e r r o r has been assumed, and t h i s case has been withdrawn from the c a l c u l a t i o n s . S ince o n l y primary d i a g n o s i s was requested from the Research D i v i s i o n o f H o s p i t a l Programs, cases o f CDH a s s o c i a t e d w i t h some oth e r reason f o r h o s p i t a l i z a t i o n w i l l not appear. 20 T h i s k i n d o f o m i s s i o n w i l l u n d e r e s t i m a t e t h e a c t u a l n u m b e r o f s u r g i c a l c a s e s i n t h e p r o v i n c e . F o r e x a m p l e , c h i l d r e n h a n d i -c a p p e d w i t h s e v e r e C e r e b r a l P a l s y t e n d t o e x p e r i e n c e d i s l o c a t e d h i p s d u e t o s p a s t i c i t y o f t h e h i p m u s c u l a t u r e . S u r g i c a l r e p a i r o f t h e s e d i s l o c a t e d h i p s m a y o c c u r , b u t t h e c o n d i t i o n l i s t e d a t d i s c h a r g e m a y b e C e r e b r a l P a l s y . N u m b e r v d f C h i l d r e n R e c e i v i n g S u r g i c a l I n t e r v e n t i o n A n a l y s i s o f t h e d a t a p r e s e n t e d i n t h e . A p p e n d i x o f t h i s c h a p t e r s h o w s t h a t 4 1 c h i l d r e n r e c e i v e d 6 1 e p i s o d e s o f c a r e a t C h i l d r e n ' s H o s p i t a l i n f i s c a l 1 9 7 9 . I n a d d i t i o n , 4 c h i l d r e n r e c e i v e d s u r g i c a l t r e a t m e n t f o r C D H a t t h e V a n c o u v e r G e n e r a l H o s p i t a l , a n d 2 a t t h e P r i n c e G e o r g e R e g i o n a l H o s p i t a l . T h u s , t h e p r o v i n c i a l t o t a l f o r 19 79 i s 47 c a s e s . O f t h i s t o t a l , o n l y 3 c h i l d r e n r e s i d e d i n V a n c o u v e r a t t h e t i m e t h e y w e r e t r e a t e d . I t i s p o s s i b l e t h a t t h e y w e r e n o t b o r n i n a V a n c o u v e r h o s p i t a l , b u t t h i s i n f o r m a t i o n w a s n o t a v a i l a b l e f r o m H o s p i t a l P r o g r a m s . E s t i m a t i n g t h e R a t e o f S u r g i c a l C a s e s T h e i n c i d e n c e r a t e o f c a s e s o f C D H p r e s e n t i n g f o r s u r g e r y f r o m a n u n s c r e e n e d p o p u l a t i o n h a s b e e n e s t i m a t e d b y L e h m a n n a s 1 . 4 p e r 1 0 0 0 l i v e b i r t h s ( 2 3 ) . T r e d w e l l s u g g e s t s t h a t 1 . 5 p e r 1 0 0 0 u n s c r e e n e d i n f a n t s w i l l r e q u i r e s u r g e r y f o r C D H . V o n R o s e n ( 1 1 ) f o u n d 1 . 7 7 c a s e s o f C D H r e c e i v e d s u r g e r y p e r 1 0 0 0 u n s c r e e n e d i n f a n t s i n t h e S w e d i s h p o p u l a t i o n s t u d i e d . 21 If the actual number of cases (47',children) i d e n t i f i e d i n the analysis of data from Hospital Programs i s divided by the number of births i n B r i t i s h Columbia for 1979 (38,639 l i v e births) the incidence rate for s u r g i c a l cases from the unscreened two-thirds of the province (25,537 l i v e births) i s 1.84 per 1000. This assumes that the three babies residing i n Vancouver at the time of CDH surgery were not born i n Vancouver. If these three infants are presumed to have been born i n Vancouver, the surgical rate for the unscreened two-thirds of the province drops to 1.72 per 1000 l i v e b i r t h s , a rate close to Von Rosen's (11) finding, and only 0.32 cases per 1000 higher than, Lehmann's figure (23). Summary This chapter has shown that two-thirds of the l i v e births i n the province are producing 94% of the surgical cases of CDH. The actual rate of cases (1.72 per 1000 l i v e births) i s higher than the estimate of 1.4 per 1000 (23). The cost-minimization analysis w i l l be based on 1.5 cases per 1000 l i v e b i r t h s , acknowledging that t h i s figure may be an underestimate of the actual rate,..but a reasonable approximation. Appendix for Chapter 3 Data from the Research Division of Hospital Programs, Ministry of Health, F i s c a l 1979 (April 1, 1979 to March 31,1980) and Data from the Prince George Regional Hospital, 1981 23 CASES DISCHARGED FROM SPECIFIED BRITISH COLUMBIA HOSPITALS WITH DIAGNOSIS OF CONGENITAL DISLOCATION OF HIP (ICD9 CODE 754.3) APRIL 1, 1979 TO MARCH 31, 1980 Hospital Vancouver General Age l i v e b i r t h 1 day 1 mo. 3 mo. 6 mo. 2 y r . Male Female T o t a l T o t a l 6 1 1 9 17 1 1 20 23 2 1 1 1 1 29-St. Paul's l i v e b i r t h Grace l i v e b i r t h 13 2 9 4 2 Children's Residence Age Sex S u r g i c a l Procedure Abbotsford 1 y r . F 0 3 . . 1 6 Burnaby 9 mo. F 1 1 . . 1 2 I I 1 mo. F 9 1 . . 7 4 • I 6 mo. F 9 5 . . 1 2 , 9 1 . . 7 4 I I 3 mo. F 0 7 . . 2 9 , 0 7 . . 5 3 it 6 mo. F 1 1 . . 1 2 ii 3 mo. F 1 1 . . 1 2 n 1 y r . M 9 5 . . 1 5 , 0 7 . . 5 1 I I 9 mo. F 6 5 . . 0 1 , 6 6 . . 0 0 Chilliwack 1 y r . M 0 3 . . 3 9 , 1 1 . . 8 8 , ti 1 mo. F 9 5 . . 1 2 , 0 7 . . 5 1 Cranbrook 4 y r . F -I I 5 y r . F 0 7 . . 2 9 Delta 3 mo. F 9 1 . . 7 4 , 9 5 . . 7 6 Fort St. John 1 y r . F 0 7 . . 2 9 I I 2 y r . M 9 1 . . 7 4 , 0 7 . . 5 1 H 2 y r . F 8 9 . . 3 4 , 1 3 . . 0 3 I I 2 y r . F 9 0 . . 6 4 , 0 7 . . 3 9 I I 2 y r . M 0 7 . . 5 1 Kamloops 6 mo. F 9 2 . . 2 4 , 9 2 . . 7 4 , Langley 1 y r . F 8 9 . . 3 4 0 7 . 3 9 - 2 -2% Cases Discharged Congenital D i s l o c a t i o n of Hip (continued) Children's Residence Age Sex S u r g i c a l Procedures Langley 1 yr. 89, .34 it 2 y r . M 91. .84, 89, .34 I I 3 mo. F 91, .74, 95, .12 I I 3 mo. F 11. .12 Mackenzie 5 y r . F 91, .74, 07, .44, I I 6 yr. F -Matsqui 3 y r . F 91, .84 New Westminster 1 yr. F 91. .84, 89. .38, I I 1 y r . F 11. .88 North Vancouver 1 y r . F 89. .34, 95. .76 Port Clements 1 mo. F 92. .74, 11. .12 Powell River 6 mo. F 95. .12, 07. .53 Queen Charlotte 7 days M 91. .74, 07. .53 I I 3 mo. F 07. .54 Quesnel 5 y r . F 89. .34 Richmond 9 mo. F 91. .74, 95. .76, I I 9 mo. F 95. .76, 07. .29, Rossland 1 y r . F 91. .74, 95. .12 I I 1 yr. F 11. .12 Saanich 6 y r . F 89. .34 • Squamish 3 mo. F 91. .74, 95. .76 I I 6 mo. F 03. .39, 07. .53 Surrey 2 y r . M 95. .14, 18. .14 I I 3 y r . F 89. .38 Terrace •' 1 y r . F 89. .34, 90. .64 Vancouver 3 mo. F 95. .12, 07. .53 I I 1 y r . F 92. .84 •I 3 mo. M 91. .84 V i c t o r i a 6 y r . F 89. .34 West Vancouver 6 mo. F 91. .74, 95. .12, Williams Lake 1 y r . F 03. .39 1 y r . F 91. .74, 07. .44 •I 1 y r . F 11. .13 from another country 1 yr. F 07. ,29 H 1 yr. F 89. ,34, 90. ,64 •I 2 yr. F 90. 54, 07. ,39 I I 6 mo. F 91. ,74 ti 9 mo. F 07. .29 07. ,53 it 9 mo. F 07. ,51 ii 2 y r . F — Total - 5 3 F 8 ' M 6 1 CODING OF HOSPITAL DISCHARGE DIAGNOSES ICD9. 25 03.16 electroencephalogram 03.39 nonoperative measurements and examinations 07.29 f o r c i b l e correction of a deformity 07.39 other physical therapy 07.44 other s k e l e t a l t r a c t i o n 07.51 application of a plaster jacket 07.53 application of a cast 07.54 application of a s p l i n t 11.12 replacement of a cast on a lower limb 11.88 removal of a cast 13.03 transfusion of whole blood 18.14 presacral sympathectomy 65.01 hernia repair 66.00 i n c i s i o n of the abdominal wall 89.34 d i v i s i o n of the bone of the hip (osteotomy) 89.38 d i v i s i o n of the bones of the pe l v i s 90.64 removal of an in t e r a n l f i x a t i o n device 91.64 debridement of the femur 91.74 closed reduction of d i s l o c a t i o n of the hip 91.84 open reduction of d i s l o c a t i o n of the hip 92.24 d i v i s i o n of j o i n t capsule of the hip 92.74 contrast arthrogram of the hip 95.12 adductor tenotomy 95.14 myotomy for d i v i s i o n (division of muscle) 95.76 other change i n length of muscle, tendon, and f a s c i a 26 P r i n c e George Regional H o s p i t a l C o n g e n i t a l D i s l o c a t i o n o f the Hip, Semi-annual d i a g n o s i s index. prepared by: Mrs. C a r o l Rother, C.C.H.R.A.(A) Su p e r v i s o r , T e c h n i c a l S e r v i c e s M e d i c a l Records Department NUMBER OF CASES OF CONGENITAL DISLOCATION OF THE HIP 1974-1979. Year Number cases S u r g i c a l cases Age group: Newborns l-12mo. 13-24mo. 1974 3 2 1 1 1 1975 4 1 2 2 -1976 2 - - 2 -1977 2 1 1 - 1 1978 9 4 1 3 2 1979 5 2 3 1 1 T o t a l :25 10 8 9 5 27 C H A P T E R 4 A C O S T - M I N I M I Z A T I O N A N A L Y S I S O F S C R E E N I N G F O R C O N G E N I T A L D I S L O C A T I O N O F T H E H I P I N N E W B O R N S I n t r o d u c t i o n T h i s c h a p t e r a d d r e s s e s t h e i s s u e o f w h e t h e r a s c r e e n i n g p r o g r a m f o r C D H i s c o s t - m i n i m i z i n g w h e n c o m p a r e d t o s u r g i c a l c o r r e c t i o n o f C D H i n c a s e s f r o m a n u n s c r e e n e d p o p u l a t i o n . C o s t - e f f e c t i v e n e s s T h e o r y : t h e c a s e o f c o s t - m i n i m i z a t i o n C o s t - e f f e c t i v e n e s s a n a l y s i s p r o v i d e s a f r a m e w o r k f o r c o m -p a r i n g t w o m e t h o d s o f t r e a t i n g i n f a n t s w i t h C D H . I n t h i s e v a l u a t i o n , t h e o u t c o m e s o f b o t h m e t h o d s o f t r e a t m e n t a r e a s s u m e d t o b e t h e s a m e : n o r m a l f u n c t i o n i n g o f t h e a f f e c t e d h i p j o i n t . W h e n t h e o u t c o m e s o f t w o p r o g r a m s a r e t h e s a m e , a s p e c i a l c a s e o f c o s t - e f f e c t i v e n e s s c a n b e u s e d , c o s t - m i n i m i z a t i o n a n a l y s i s . I n a d d i t i o n t o t h e p o s i t i v e b e n e f i t s o f i m p r o v e d h i p f u n c t i o n , s o m e n e g a t i v e p s y c h o l o g i c a l o u t c o m e s o f b o t h t r e a t -m e n t s c a n b e c o n s i d e r e d . T h e s e m a y i n c l u d e t h e p a i n a n d s u f f e r i n g o f t h e i n f a n t b e i n g t r e a t e d , i n c o n v e n i e n c e a n d s t r e s s f o r t h e f a m i l y m e m b e r s , t h e p a r e n t a l c o n c e r n o f h a v i n g a c h i l d l a b e l l e d c o n g e n i t a l l y d e f e c t i v e , a n d t h e e f f e c t o f r e s i d u a l d i s a b i l i t y i n t h e c h i l d . T h e o b j e c t i v e o f t h i s a n a l y s i s i s t o p r o v i d e q u a n t i t a t i v e i n f o r m a t i o n o n t h e c o s t s o f t h e a l t e r n a t i v e p r o g r a m s s o t h a t i t b e c o m e s p o s s i b l e t o c h o o s e t h e l e a s t c o s t l y p r o g r a m , a s s u m i n g e q u a l l y b e n e f i c i a l o u t c o m e s ( 4 3 ) . T h e r e s o u r c e s c o n s u m e d ( c o s t s ) 28 i n both CDH programs, with the e x c e p t i o n of p s y c h o l o g i c a l c o s t s , are accounted f o r i n d o l l a r terms, which are not d i s c o u n t e d . Statement of Program O b j e c t i v e s In c l i n i c a l terms, the o b j e c t i v e of each program i s to produce a normally f u n c t i o n i n g h i p j o i n t i n an i n f a n t w i t h a C o n g e n i t a l D i s l o c a t i o n . The s c r e e n i n g program must f i n d and t r e a t , w h ile the s u r g i c a l program t r e a t s o n l y those cases p r e -sented. The sum o f the outputs of each program i s the number of c h i l d r e n with CDH who are e f f e c t i v e l y t r e a t e d . I t i s a l s o necessary to be able to s t a t e with confidence whether a l l the c h i l d r e n who c o u l d b e n e f i t from the program a c t u a l l y r e c e i v e c a r e . In o t h e r words, i s the s c r e e n i n g program f i n d i n g a l l cases o f CDH, or i s surgery being p r o v i d e d f o r a l l ' l a t e ' cases? The Program Options and Data Sources Two o p t i o n s w i l l be compared. F i r s t , a comprehensive s c r e e n i n g program such as t h a t a t Grace H o s p i t a l (9) or a t the Vancouver General H o s p i t a l (7) w i l l form the data base f o r determining the c o s t s of a s c r e e n i n g program. The i n c i d e n c e r a t e s from these two h o s p i t a l s are used to determine the l i k e l y range o f the number o f i n f a n t s who would r e c e i v e c o n s e r v a t i v e treatment. The c o s t of treatment i s i n c l u d e d i n the s c r e e n i n g program t o t a l c o s t . Second, a s i t u a t i o n of no or i n e f f e c t i v e s c r e e n i n g w i l l be used to estimate the s u r g i c a l c o s t of h i p r e p a i r i n c h i l d r e n w i t h CDH. The data used were s u p p l i e d by the Research D i v i s i o n of H o s p i t a l Programs ( a n a l y s i s i n Chapter 3 ) , and by Dr. Tredwell 29 of the Orthopedic Division of the Department of Surgery at Children's Hospital. The B r i t i s h Columbia Medical Association Statement of Fees for 1981 i s the source of data for the charges levied by Orthopedic Surgeons and Anaesthetists. The Registered Nurses Association of B.C. supplied the 1981 salary schedule for nurses. Dr. Tredwell outlined the patterns of care r e s u l t i n g from screened cases and from surgical cases. Dr. B l a i r Fulton, from the Department of Anaesthesia at the Vancouver General Hospital, described the anaesthetic procedures and protocol of patient care for young children. He also outlined some of the non-monetary costs a c h i l d may face i n a surgical setting. These are: r i s k of anaesthetic death, r i s k of malalignment--or. non-union of bones or bone grafts, and r i s k of infect i o n s and complications. Direct and Indirect Costs: d e f i n i t i o n s This analysis w i l l take into account each component common to the two programs, as well as those which are unique. There are some d i r e c t and i n d i r e c t costs to be considered which vary across programs. Direct costs for the health system include costs generated by screening, follow-up treatment, surgery, and associated hospital and physician costs. Direct costs to the family include braces for conservative treatment of CDH, and tr a v e l costs for those families whose c h i l d requires surgery. Due to the v a r i a b i l i t y of these t r a v e l costs, they are acknowledged but not calculated. Indirect costs are incurred by the families of children 30 r e q u i r i n g s u r g i c a l care, and are made up of l o s t wages and l o s t p r o d u c t i v i t y due to time away from work while t r a v e l l i n g to C h i l d r e n ' s H o s p i t a l . These c o s t s are a l s o acknowledged but not c a l c u l a t e d . There i s no l o s t p r o d u c t i v i t y a t t r i b u t a b l e to h i p s c r e e n i n g done i n the O b s t e t r i c a l U n i t o f a h o s p i t a l , nor to the i n i t i a l treatment o f a p o s i t i v e case: use of a t r i p l e d i a p e r . Minimal i n d i r e c t c o s t s may a r i s e due to time spent f o r follow-up i n the doctor's o f f i c e . S c reening Program and Follow-up Treatment Costs S c r e e n i n g c o s t s a t Grace H o s p i t a l and the Vancouver General H o s p i t a l are comprised o f one h a l f - h o u r o f the screener's time twice weekly i n the i n f a n t nursery. The Orthopedic Surgeons who perform the s c r e e n i n g do not a c t u a l l y 'charge' f o r t h i s s e r v i c e , but the c o s t o f t h e i r time can be estimated a t $50.00 f o r each h a l f hour. (Dr. Tredwe l l ' s estimate) In a d d i t i o n , a member of the nursery s t a f f must be a v a i l a b l e to r e c o r d the r e s u l t s o f the sc r e e n i n g . An estimate o f t h i s c o s t i s $7.00 f o r each h a l f hour s e s s i o n , d u r i n g which an average o f 30 i n f a n t s are screened. I t should be noted t h a t Grace H o s p i t a l and the Vancouver General H o s p i t a l b e n e f i t from economies of s c a l e which may not apply t o o t h e r r e g i o n a l h o s p i t a l s . The i n c i d e n c e o f p o s i t i v e cases o f CDH a t Grace H o s p i t a l i s 14.6 per 1000 i n f a n t s screened ( 9 ) , and a t the Vancouver General H o s p i t a l the i n c i d e n c e i s 9.9 per 1000 l i v e b i r t h s ( 7 ) . In t h i s a n a l y s i s 9.9 has been rounded up to 10.0. Of those i n f a n t s i d e n t i f i e d by s c r e e n i n g and t r e a t e d with t r i p l e d i a p e r s , 80% w i l l undergo recovery by the time o f the f i r s t o f f i c e v i s i t to the Orthopedic Surgeon. I t i s recommended t h a t these babies continue to wear t r i p l e d i a p e r s u n t i l s i x weeks o f age. For the remaining 20% a brace w i l l be p r e s c r i b e d . The brace c o s t v a r i e s from $30.00 t o $60.00 depending on the degree of i m m o b i l i z a t i o n r e q u i r e d . The brace i s p a i d f o r by the parent. Using the data from the 19 81 Fee Schedule (see the Appendix f o r t h i s chapter) the s c r e e n i n g and follow-up treatment c o s t s are c a l c u l a t e d and summarized i n Tables 4.1, 4.2, 4.3, and 4.4. The range of c o s t s expected from s c r e e n i n g and c o n s e r v a t i v e treatment of newborns wi t h CDH i s $3286.00 to $3819.00 per 1000 i n f a n t s e n t e r i n g the program. S u r g i c a l Program Costs Two types o f treatment are used f o r c o r r e c t i n g the mechanical d e f i c i e n c i e s of CDH i d e n t i f i e d when the c h i l d i s o l d e r than one month of age. The f i r s t i s open r e d u c t i o n of the h i p , used i n about 40% of s u r g i c a l cases. The second i s c l o s e d r e d u c t i o n , used f o r the remaining 60%. (These estimates were made by Dr. Tredwell.) Both s u r g i c a l procedure c o s t s are analysed i n d e t a i l i n the Appendix to t h i s chapter. The per diem has been used i n t h i s a n a l y s i s to approximate the c o s t o f h o s p i t a l i z a t i o n and surgery f o r c h i l d r e n r e q u i r i n g treatment f o r CDH. By presuming to e r r on the c o n s e r v a t i v e s i d e o f any estimate of c o s t , t h i s a n a l y s i s w i l l underestimate r a t h e r than overestimate the c o s t of the s u r g i c a l program. The per diem i n c l u d e s o p e r a t i n g room c o s t s , p o s t - a n a e s t h e t i c recovery room c o s t s , ward n u r s i n g c o s t s , ' h o t e l components' such as food and laundry, overhead, and a c t i v i t i e s such as t e a c h i n g and r e s e a r c h . 32 T A B L E 4 . 1 S U M M A R Y O F S C R E E N I N G C O S T S A T G R A C E H O S P I T A L D o c t o r ' s ' c o s t ' t o s c r e e n 3 0 0 0 i n f a n t s p e r y e a r 1 h r / w k x 52 w k s x $ 1 0 0 . . . $ 5 2 0 0 N u r s i n g t i m e f o r 1 y e a r 1 h r / w k x 52 w k s x $14 . . . $ 7 2 8 T o t a l a n n u a l c o s t • . . . $ 5 9 2 8 C o s t p e r 1 0 0 0 i n f a n t s s c r e e n e d . . . $ 1 9 7 6 T A B L E 4 . 2 S U M M A R Y O F T R E A T M E N T C O S T S F O R S C R E E N E D I N F A N T S W I T H C D H O f f i c e v i s i t s f o r e a c h C D H c a s e o c c u r w h e n t h e c h i l d i s : 2 w e e k s o l d . . . $ 5 0 3 m o n t h s . . . $ 2 4 6 m o n t h s . . . $ 2 4 12 m o n t h s . . . $24 T o t a l o f f i c e c o s t p e r C D H c a s e : . . . $ 1 2 2 C o s t o f 10 c a s e s p e r 1 0 0 0 l i v e b i r t h s C o s t o f 1 4 . 6 c a s e s p e r 1 0 0 0 i n f a n t s s c r e e n e d . $ 1 2 2 0 . $ 1 7 0 8 3 3 TABLE 4 . 3 BRACING COSTS Brace c o s t : . . . $ 3 0 to $ 6 0 Average c o s t o f b r a c i n g 20% a t i n c i d e n c e l e v e l s 1 0 / 1 0 0 0 . . . $ 9 0 Average c o s t of b r a c i n g 20% at i n c i d e n c e l e v e l s 1 4 . 6 / 1 0 0 0 . $ 1 3 5 TABLE 4 . 4 TOTAL SCREENING PROGRAM AND TREATMENT COSTS 1 0 / 1 0 0 0 1 4 . 6 / 1 0 0 0 l i v e b i r t h s i n f a n t s screened Screening $ 1 9 7 6 $ 1 9 7 6 O f f i c e v i s i t s $ 1 2 2 0 $ 1 7 0 8 Braces $ 90 $ 1 3 5 T o t a l c o s t s : $ 3 2 8 6 $ 3 8 1 9 Summary The range o f c o s t s generated by a program t h a t screens f o r CDH and t r e a t s a l l p o s i t i v e cases i s $ 3 2 8 6 to $ 3 8 1 9 per 1 0 0 0 i n f a n t s e n t e r i n g the program. 34 Almost a l l surgery f o r CDH i s done a t C h i l d r e n ' s H o s p i t a l where the per diem i s $373.85 (1 9 8 1 ) . The C h i l d r e n ' s H o s p i t a l per diem i s h i g h compared to the per diem of r e g i o n a l and g e n e r a l h o s p i t a l s i n the p r o v i n c e . The average 1981 per diem was not a v a i l a b l e from H o s p i t a l Programs. I f h i p surgery f o r c h i l d r e n was p r o v i d e d a t a r e g i o n a l h o s p i t a l the t o t a l c o s t of care would be lower. Since a l l but 5 cases of CDH surgery f o r the 1979 f i s c a l year used i n t h i s study were t r e a t e d a t C h i l d r e n ' s H o s p i t a l , the use of the a c t u a l per diem (373.85) i s a good approximation o f the r e a l d o l l a r c o s t . Based on the d e t a i l e d c o s t a n a l y s i s i n the Appendix to t h i s chapter, Table 4.5 summarizes the r e l a t i v e c o n t r i b u t i o n of each type o f surgery to the average c o s t of t r e a t i n g one i n f a n t w i t h a l a t e d i a g n o s i s of CDH. TABLE 4.5 TOTAL AND AVERAGE COSTS OF SURGICAL CARE FOR 1000 UNSCREENED INFANTS Cost of c l o s e d r e d u c t i o n of CDH $ 8,631.29 Cost o f open r e d u c t i o n o f CDH $10,393.15 Average c o s t = 60% ($8,631.29) + 40% ($10,393.15) = $9,336.03 per c h i l d t r e a t e d T o t a l c o s t per 1000 i n f a n t s unscreened = ($9,336.03 x 1.5 cases per 1000) = $14,004.05 per 1000 35 Summary This analysis of surgical costs has shown that i n a popula-tion of unscreened infants, 1.5 per 1000 l i v e b i r t h s w i l l require surgery at a cost of $14,004.05. S e n s i t i v i t y Analysis The cost of screening and treating 1000 infants with an incidence rate of 14.6 per 1000 infants screened i s $3,819.00. If a hospital did not experience the economies of scale described for Grace Hospital or the Vancouver General Hospital, the screening costs could r i s e , while the treatment costs remained constant. If a health professional other than an Orthopedic Surgeon performed the screening test, the hourly screening cost would be reduced, lowering the screening cost. I f the screening program used the most expensive screeners but provided the test to only half as many infants per hour, the t o t a l screening cost per 1000 infants would r i s e to $3,952.00. I f nursery s t a f f performed the screening test, the cost of screening 1000 infants would drop to $485.33, assuming that the y i e l d was the same as that of an Orhtopedic Surgeon. The broadest conceivable range of screening costs are therefore $485.33 to $3,952.00. Projected Annual Costs and Savings I f the screening program costs are projected onto a population of 35,000 newborns, the annual cost that could be anticipated would be $145,320.00. The surgical cost of treating 36 the same number of infants l e f t unscreened would be $ 4 9 0 , 1 7 0 . 0 0 for one year. The savings attributable to a province-wide screening program would be $ 3 4 4 , 8 5 0 . 0 0 . The major portion of this saving w i l l occur due to a reduction i n h o s p i t a l i z a t i o n of children requiring surgery for CDH. Conclusions This cost-minimization analysis has shown that the cost of providing surgery to 1 0 0 0 unscreened infants i s three times higher than the cost of providing screening and conservative treatment to 1 0 0 0 infants. In terms of i t s cost-minimization, a screening program for CDH i n newborns i s well j u s t i f i e d i n B r i t i s h Columbia. Appendix for Chapter 4 Exerpts from the 1981 Fee Schedule Detailed Calculation of Surgical Costs for CDH 38 Calculation of Costs 1. Open reduction of the hip i n a c h i l d under 1 year of age. This set of costs have been broken down into each phase of treatment the c h i l d receives. The per diem at Children's Hospital i s $373.85 as of the 1981 f i s c a l year. a) the c h i l d spends 14 days as an inpatient in s k e l e t a l t r a c t i o n . - hospital costs: 14 x 373.85 $ 5,233.90 - doctor's v i s i t s : 14 x 12.00 $ 168.00 b) the c h i l d undergoes surgery for CDH. - surgeon's fee: • . . .$ 500.00 - anaesthetic: $ 134.00 c) the c h i l d spends 5 days post-operatively as an inpatient - hospital costs: 5 x 373.85 $ 1,869.00 - doctor's v i s i t s : 5 x 12.00 $ 60.00 d) 8 weeks afte r f i r s t admission, the c h i l d returns to hospital to have the cast and hip pin removed. - surgeon's fee: •....$ 80.00 - anaesthetic: $ 53.60 e) the c h i l d remains i n hospital 4 days. -hospital costs: 4 x 373.85 $ 1,495.40 - doctor's v i s i t s : 4 x 12.00 $ 48.00 f) the c h i l d i s followed by the doctor u n t i l the c h i l d i s f u l l y mature. - yearly o f f i c e v i s i t s : 15 x 50.00 $ 750.00 Total costs $10,393.15 •39 Calculation of Costs 2. Closed reduction of the hip i n a c h i l d under 1 year of age. This set of costs i s treated as i n item #1. a) the c h i l d spends 14 days as an inpatient in s k e l e t a l t r a c t i o n . - hospital costs: 14 x 373.85 $5,233.90 - doctor's v i s i t s : 14 x 12.00 $ 168.00 b) the c h i l d undergoes a closed reduction most often with an adductor tenotomy. - surgeon's fee: closed reduction $ 180.00 adductor tenotomy $ 127.00 - anaesthetic: $ 53.60 c) the c h i l d spends 3 days post-operatively as an inpatient. - hospital costs: 3 x 373.85 $1,121.55 - doctor's v i s i t s : 3 x 12.00 $ 36.00 d) the c h i l d has an average of 4 cast changes vi a the Day Care Surgery*. - hospital costs: 4 x 126.61 $ 506.44 - surgeon's fee for casts: 4 x 60.00 $ 240.00 - anaesthetic: 4 x 53.70 $ 214.80 e) the c h i l d i s discharged the same day. f) the c h i l d i s followed by the doctor u n t i l the c h i l d i s f u l l y mature. - yearly o f f i c e v i s i t s : 15 x 50.00 . . .$ 750.00 Total costs $8,631.29 *The cost of Day .Care Surgery has been estimated by Evans to be approximately one-third that of the hospital per diem. (49) Data from the 19 81 Fee Schedule of the B r i t i s h Columbia Medical Association Fees for Orthopedic Surgery and Office V i s i t s : Consultation $50.00 Repeat consultation for the same condition within 6 months $24.00 Continuing care by physician: o f f i c e v i s i t $16.00 hospital v i s i t $12.00 home v i s i t $30.00 emergency $60.00 CDH: closed reduction $180.00 open reduction $500.00 Adductor tenotomy: one tendon $127.00 two tendons $212.00 B i l a t e r a l cast change $60.00 41 A n a e s t h e t i c Fees The b a s i c A n a e s t h e t i c u n i t charge f o r 1 9 8 1 i s $ 8 . 4 0 . T h i s u n i t charge i s a p p l i e d to each p a t i e n t ' s care i n four separate components: a) The p a t i e n t ' s h i s t o r y i s eva l u a t e d by the A n a e s t h e t i s t to determine the r i s k t h a t the p a t i e n t p r e s e n t s f o r the a n a e s t h e t i c procedure. T h i s i s a 3 u n i t charge, or 3 x $ 8 . 4 0 $ 2 5 . 2 0 b) A l o a d i n g item i s i d e n t i f i e d f o r every s u r g i c a l procedure (from 1 to 17 ) depending on the a n a e s t h e t i c d i f f i c u l t y of the procedure. For an open r e d u c t i o n o f the h i p , the l o a d i n g item i s 4 , so the t o t a l c o s t of t h i s component i s 4 x $ 8 . 4 0 $ 3 3 . 6 0 c) A time charge i s used based on four components i n each of the f i r s t two hours of an a n a e s t h e t i c and s i x components i n each subsequent hour u n t i l the surgery i s completed. An open r e d u c t i o n i n a c h i l d takes about two hours, so the time charge becomes 8 x $ 8 . 4 0 $ 6 7 . 2 0 d) A u n i t adjustment i s made f o r the degree of e x p e r t i s e r e q u i r e d f o r s p e c i a l c l a s s i f i c a t i o n s of p a t i e n t s . For newborns, an a d d i t i o n a l 5 u n i t s are b i l l e d , f o r c h i l d r e n under one year or age, one a d d i t i o n a l u n i t i s b i l l e d . Most open r e d u c t i o n s are done on c h i l d r e n of one year or l e s s . The u n i t c o s t i s $ 8 . 4 0 Average A n a e s t h e t i c c o s t f o r open r e d u c t i o n of CDH. . . . . $ 1 3 4 . 4 0 Average A n a e s t h e t i c c o s t f o r c l o s e d r e d u c t i o n of CDH. . . . $ 5 3 . 6 0 CHAPTER 5 PLANNING A PROVINCIAL SCREENING PROGRAM FOR CDH I n t r o d u c t i o n To implement a p r o v i n c i a l h i p s c r e e n i n g s e r v i c e f o r neonates, s e v e r a l c o n d i t i o n s w i l l need to be met. These con-d i t i o n s are d i s c u s s e d i n t h i s chapter, based on what i s a l r e a d y known about CDH, the outcomes of e x i s t i n g s c r e e n i n g programs and the c o s t i m p l i c a t i o n s of a l t e r n a t i v e ways of d e v e l o p i n g comprehensive s c r e e n i n g s e r v i c e s . The optimal outcome w i l l be the promotion of e a r l y c o n s e r v a t i v e management of CDH w i t h subsequent r e d u c t i o n of the number o f cases of surgery. The R a t i o n a l e f o r a P r o v i n c i a l Screening Program The f a c t o r s p l a c i n g i n f a n t s at r i s k f o r CDH have been i d e n t i f i e d (2,7,9,17,19,24,26). However, the n a t u r a l h i s t o r y of the d i s e a s e i s not w e l l enough understood to i d e n t i f y which i n f a n t s w i t h h i p d y s p l a s i a a t b i r t h w i l l b e n e f i t from c o n s e r v a t i v e treatment and which w i l l spontaneously recover h i p s t a b i l i t y (4, 23). Thus, a l l i n f a n t s w i t h a p o s i t i v e Barlow's t e s t a t . s c r e e n i n g should be t r e a t e d w i t h t r i p l e d i a p e r s . The methodology f o r a comprehensive s c r e e n i n g program i s known (7,9,11,26) and can be a p p l i e d i n any newborn nur s e r y i n the p r o v i n c e . I n f a n t s screened, and found to demonstrate sig n s o f h i p d y s p l a s i a should be t r e a t e d w i t h t r i p l e d i a p e r s , and f o l l o w e d by a p h y s i c i a n . Recommendations f o r repeat s c r e e n i n g a t two to f o u r months 43 of age have been made (4,23). Parkin has suggested a two phase screening program with neonatal screening followed by a repeat test a few weeks afte r b i r t h . However, he acknowledges that the value of the l a t e r test i s questionable.. (4) . Frankenburg (48) suggests that the main b a r r i e r to CDH screening and early treatment has been that a screening program has not been shown to cost less than l a t e r s u r g i c a l management of established cases of CDH. The cost-minimization analysis presented i n t h i s thesis suggests that CDH screening coupled with conservative treatment costs about one-third as much as su r g i c a l intervention i n established cases. Hip screening at the three Vancouver hospitals has sharply reduced the need for surgery of the hip i n children born and examined for CDH i n t h i s c i t y . Economic and Social Outcomes of Screening Not a l l outcomes of a hip screening program can be i d e n t i f i e d as economic i n nature. A net'dollar saving w i l l occur as more infants are screened throughout the province. In addition, there w i l l be a reduction of morbidity and d i s a b i l i t y associated with hip surgery. The psychological trauma of h o s p i t a l i z a t i o n for young children w i l l be reduced. The d i r e c t cost borne by families who must tr a v e l to Children's Hospital for t h e i r c h i l d ' s surgery w i l l be eliminated, as w i l l the i n d i r e c t costs of income foregone due to l o s t work days. In addition, there i s growing concern among c l i n i c i a n s about the r e l a t i o n s h i p between surgical intervention i n CDH and idiopathic O s t e o a r t h r i t i s of the Hip (23,35,36,37,38). 44 A reduction i n the amount of surgery performed now may well show a reduction i n the incidence of t h i s chronic disease i n these children, thus creating possible additional health care savings i n the future. Policy A p o l i c y of universal services for newborns throughout the province would provide guidelines for provision of CDH screening i n regions where this service i s needed. The decision-making power l i e s primarily within the Ministry of Health. I f the screening was attached to a com-prehensive neonatal developmental assessment i t would have a lower marginal cost and an increased l i k l i h o o d of implemen-tation . Planning and Implementation The planning w i l l encompass the screening program, the f a c i l i t i e s for treatment of pos i t i v e cases, and evaluation of the program. F i r s t , interested health professionals w i l l need to be found who w i l l undertake hip screening. Second, physic:!,. cians w i l l be needed to provide follow-up care for p o s i t i v e cases, and a r e f e r r a l to Orthopedic Surgeons i n regional centres w i l l need to be considered. Third, f a c i l i t i e s for making and f i t t i n g the braces needed for some CDH cases w i l l be needed i n hospitals without Orthotics Departments. A Physio-therapy Department i s an adequate substitute where necessary, as has happened at the Prince George Regional Hospital. 45 S t a f f i n g Physicians and nurses with no previous experience at hip screening would require a short t r a i n i n g period and some supervised practise to develop e f f e c t i v e screening s k i l l s . Consideration i s needed for additional administrative time i n recording test r e s u l t s , managing the data, following missed infants, and r e f e r r a l of pos i t i v e cases for treatment by physicians. The program should begin implementation i n hospitals where an Orthopedic Surgeon i s already i n practise, and progress incrementally to other hospitals i n each region. I f nurses are used as screeners, instead of Orthopedic Surgeons or Family Physicians, the r e l a t i v e cost of screening w i l l decrease, increasing the cost-minimization e f f e c t of screening. However, the rate of i d e n t i f i c a t i o n of positive cases has been shown to decrease when residents were used to screen (2 3); no data are available on the effectiveness of nurses. Lehmann (23) recommends the use of an Orthopedic Surgeon where possible, because the rate of i d e n t i f i c a t i o n of p o s i t i v e cases i s high, and the number of missed cases i s lower than both residents and Family Physicians. Evaluation Evaluation of the P r o v i n c i a l Screening Program w i l l require records to be kept on the outcome of each screening t e s t . In addition, the follow-up of posi t i v e cases w i l l i d e n t i f y the number of infants who experience spontaneous recovery, and the number requiring bracing services. 46 Any cases p r e s e n t i n g f o r surgery a f t e r the i n i t i a t i o n of the s c r e e n i n g program should be t r a c e d to t h e i r h o s p i t a l o f b i r t h f o r c o n f i r m a t i o n o f s c r e e n i n g d i a g n o s i s . The Vancouver General H o s p i t a l study has shown t h a t 1 i n 5000: newborns screened w i l l have no s i g n s of D y s p l a s i a but w i l l r e q u i r e h i p surgery d u r i n g the f i r s t year o f l i f e . The program e v a l u a t i o n w i l l show i f t h i s r a t e , p r o j e c t e d on an annual b i r t h r a t e of approximately 40,000 w i l l produce 8 cases of h i p surgery. Budget I m p l i c a t i o n s There w i l l be an i n i t i a l c o s t f o r the s c r e e n i n g program to t r a i n s c r e e n i n g personnel and pr o v i d e a d d i t i o n a l record-keeping f a c i l i t i e s . However, there w i l l be a more than compensating s a v i n g due to reduced numbers o f s u r g i c a l cases. The c o s t of p r o v i d i n g s u r g i c a l care t o 47 cases i n f i s c a l 1979 i s estimated to be $490,170. I f onl y 8 i n f a n t s r e q u i r e d surgery, the annual c o s t to the M i n i s t r y of Health would be (at $9,336. per c h i l d ) $74,688. The s a v i n g i s $415,482. Con c l u s i o n s T h i s t h e s i s has shown t h a t i t i s p o s s i b l e to i d e n t i f y i n f a n t s a t r i s k f o r C o n g e n i t a l D i s l o c a t i o n o f the Hip, and to t r e a t e f f e c t i v e l y those found a t s c r e e n i n g to have d i s l o c a t e d o r d i s l o c a t a b l e h i p s . S c r e e ning and c o n s e r v a t i v e treatment has been shown to c o s t only o n e - t h i r d as much as s u r g i c a l care per 1000 l i v e b i r t h s . Some of the s a v i n g i n s u r g i c a l c o s t s i s consumed by s c r e e n i n g and treatment c o s t s . Regions of the p r o v i n c e have been i d e n t i f i e d where h i p 47 screening i s not done or i s i n e f f e c t i v e . These regions should receive p r i o r i t y i n establishing new hip screening programs.in neonatal nurseries. Implementation of a province wide screening program for Congenital Dislocation of the Hip i n infants has been supported by the data analysis and proposals made i n th i s thesis. 48 BIBLIOGRAPHY Congenital Dislocation of the'Hip 1. Albee, F.H., Injuries and Diseases of the Hip: Surgery and  Conservative Treatment, Paul B. Hoeber Inc., New York, 19 37 p. 77-133 2. Coleman, S., Congenital Dislocation of the Hip, CV. Mosby and Co., St. Louis, 1978 3. Valman and Finlay, Dislocated and Dislocatable Hip i n the Newborn, B r i t i s h Medical Journal, Jan. 19, 1980, p. 164-166 4. Parkin, D.M., How Successful i s Screening for Congenital Dislocation of the Hip?, American Journal of Public Health, Vol. 71, No. 12, December, 1981, p. 1378-1383 5. Hass, J., Congenital Dislocation of the Hip, Charles C. Thomas, Sp r i n g f i e l d , 111., 1951 6. Wynne-Davies, R., Acetabular Dysplasia and F a m i l i a l Joint Laxity: Two E t i o l o g i c a l Factors i n Congenital Dislocation of the Hip, Journal of Bone and Joint Surgery, Vol. 52, No. 4, 1970 7. Tredwell, S.J., and B e l l , H.M., E f f i c a c y of Neonatal Hip Examination, Journal of Pediatric Orthopedics, Vol. 1, No. 1, 1981 8. McKibbon, B., Anatomical Factors i n the S t a b i l i t y of the Hip Joint i n the Newborn, The Journal of Bone and Joint Surgery, Vol. 52-B, No. 1, 1970, p. 148-159 9. B e l l , H.M., and Fraser, J ., Congenital Dislocation of the Hip in Infants: a cohort study, to be presented at the Inter-national Congress of Orthopedics, Durban, South A f r i c a , March, 1982. 49 10. C z e i z e l , A., Tusnady, G. , Vaczo, G., and Vizkelety, T., The Mechanism of Genetic Predisposition i n Congenital Dislocation of the Hip, Journal of Medical Genetics, Vol. 12, 1975, p. 121-124 11. von Rosen, S., Diagnosis and Treatment of Congenital Dis-location of the Hip i n the Newborn, Journal of Bone and  Joint Surgery, Vol. 44-B, No. 2, 1962 12. Wilkinson, J.A., Prime Factors i n the Etiology of Congenital Dislocation of the Hip, Journal of Bone and Joint Surgery, Vol. 45-B, No. 2, 1963, p. 268-283 13. Thieme, W.T., Wynne-Davies, R., B l a i r , H.A.F., B e l l , E.T. and Loraine, J.A., C l i n i c a l Examination and Urinary Oestrogen Assays i n Newborn Children with Congenital Dislocation of the Hip, Journal of Bone and Joint Surgery, Vol. 50-B, No. 2, p. 546 14. Andren, L., and Borglin, N.E., Disturbed Urinary Excretion Pattern of Oestrogens i n Newborns with Congenital Dislocation of the Hip, Acta Endocrinologica, No. 37, 1961, p. 423 15. Drummond, D.S., and Rogala, E., An Aid i n the Management of Congenital Dislocation of the Hip, The Journal of Bone and  Joint Surgery, Vol. 62-A, No. 8, 1980, p. 1379-1380 16. Howorth, B., Development of Present Knowledge of Congenital Displacement of the Hip, C l i n i c a l Orthopedics and Related  Research, Vol. 125, 1977, p. 68-82 17. Wilkinson, J. and Carter, C , Congenital Dislocation of the Hip, The Journal of Bone and Joint Surgery, Vol. 42-B, No. 6, 1960, p. 669 18. Walker, J.M., Congenital Hip Disease i n a Cree-Ojibwa Pop u l a t i o n : A r e t r o s p e c t i v e Study, Canadian Medical Assoc-i a t i o n J o u r n a l , V o l . 116, 1977, p. 501-504 19. 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Herold, H.Z., U n i l a t e r a l C o n g e n i t a l Hip D i s l o c a t i o n with C o n t r a l a t e r a l A v a s c u l a r N e c r o s i s , C l i n c a l Orthopedics and  Re l a t e d Research, No. 148, 1980, p. 196-202 39. Kalamchi, A., and MacEwan, G.D., A v a s c u l a r N e c r o s i s f o l l o w i n g Treatment of C o n g e n i t a l D i s l o c a t i o n o f the Hip, The J o u r n a l  o f Bone and J o i n t Surgery, V o l . 62-A, No. 6, 1980, p. 876-887 Cost E f f e c t i v e n e s s 40. Sugden, R., and W i l l i a m s , A., The P r i n c i p l e s o f P r a c t i c a l  C o s t - b e n e f i t A n a l y s i s , Oxford U n i v e r s i t y Press, 1978, Chapter 13 41. S h o r t e l l , S.M., and Richardson, W.C., Health Program E v a l -u a t i o n , C V . Mosby Co., 1978, pp. 66-73 42. Stoddart, G.L., On Determining the E f f i c i e n c y o f H e a l t h Programs, 1980, mimeo 43. 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Bay, K.S., Flatman, D., and Nestman, L., The Worth of a S c r e e n i n g Program: An A p p l i c a t i o n of a S t a t i s t i c a l D e c i s i o n Model f o r the B e n e f i t E v a l u a t i o n of Screening P r o j e c t s , American J o u r n a l of P u b l i c H e a l t h , V o l . 66, No. 2, 19 76 p. 145-150 48. Frankenburg, W.K., To Screen or Not to Screen: C o n g e n i t a l D i s l o c a t i o n of the Hip, The American J o u r n a l of P u b l i c  H e a l t h , December, 1981, v o l . 71, no. 12, p. 1311-1313 H o s p i t a l Costs 49. Evans, R.G., and Robinson, G.C., S u r g i c a l Day Care: measure-ments of the economic p a y o f f , Canadian M e d i c a l A s s o c i a t i o n  J o u r n a l , V o l . 123, Nov. 8, 1980, p. 873-880 

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