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Resistant esophageal stenosis associated with reflux after repair of esophageal atresia: A therapeutic approach
Resistant Esophageal Stenosis Associated With Reflux After Repair of Esophageal Atresia: A Therapeutic Approach By Raphael Pieretti, Barry Shandling, and Clinton A. Stephens NE OF THE MOST important late complications after repair of esophageal atresia and tracheoesophageal fistula is an obstinate stenosis at the anastomosis. ~4 Such a stenosis may be perpetuated by gastroesophageal reflux and may be cured by antireflux surgery, s This paper presents a retrospective review of cases of resistant postoperative esophageal stenosis after repair of esophageal atresia, treated by antireflux surgery at The Hospital for Sick Children, Toronto, from January 1958 to February 1973.
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MATERIAL AND METHODS During the period studied, 281 children were treated for esophageal atresia and tracheoesophageal fistula. Of the 217 patients who survived, 77 (35%) developed stenosis of various degrees of severity. Repeated dilatations brought no improvement in 15 of these patients, who showed radiographic and/or endoscopic evidence of reflux with or without hiatal hernia. We reviewed the charts of the 15 patients for clinical features, number of pre- and postoperative dilatations, type of treatment, and results.
CLINICAL FEATURES The patients ranged in age from 2 mo to ll yr. All had dysphagia, regurgitation, recurrent episodes of aspiration, pneumonia, and failure to thrive. End-to-end esophagoesophagostomy with division and closure of the fistula had been done in ten children, and end-to-side procedure with transfixion and/or ligation of the fistula in five. Leakage had occurred within the first postoperative week in five patients. Fistula recurred in four children, two of whom had had an evident leak, and one child had a second recurrence.
RADIOGRAPHIC FINDINGS The diagnosis of reflux and hiatal hernia took some time and effort to establish in these patients (on average, 2.7 esophagograms were required to confirm the diagnosis). In two cases, it was first made by endoscopic examination and subsequently confirmed by radiography. Free reflux without hiatal hernia was found in five cases and with hiatal hernia in nine: in one child, radiography showed a mild degree of stenosis at the gastroesophageal junction presumably due to reflux.
From the Department of Surgery, The Hospital for Sick Children, Toronto, Ontario, Canada. Presented at the American Academy of Pediatrics, Surgical Section, Chicago, Ill., October 20-24, 1973. Raphael Pieretti, M.D.: Chief Resident, Division of General Surgery, The Hospital for Sick Children, Toronto. Barry Shandling, M.B.: Staff Surgeon, The Hospital for Sick Children, Toronto. Clinton A. Stephens, M.D.: Chief, Division of General Surgery, The Hospital for Sick Children, Toronto. Address for reprint requests: Barry Shandling, M.B., The Hospital for Sick Children, 555 University Avenue, Toronto M5G 1)(8, Ontario, Canada. 9 1974 by Grune & Stratton, Inc.
Journal of Pediatric Surgery, Vol. 9, No. 3 (June), 1974
355
356
PIERETTI, SHANDLING, AND STEPHENS
ENDOSCOPIC FINDINGS All 15 patients had tight anastomotic stenoses. Granulation tissue at the site of the anastomosis was seen in three. Eight had distal esophagitis, seven with hiatal hernia, and one with stenosis at the gastroesophageal junction.
TREATMENT Conservative treatment of the reflux was tried without success on six infants. These babies were given thickened feedings in small amounts at frequent intervals and with upright nursing. The average number of dilatations before the antireflux operation was 11 per patient; the frequency varied according to the severity of the stenosis from once every 3 days to once a month. Two patients with massive reflux underwent fewer dilatations because for obvious reasons an antireflux operation was indicated earlier. One child required 43 dilatations, and the diagnosis was missed entirely until a laparotomy established that the upper gastrointestinal bleeding was secondary to reflux esophagitis. The antireflux operation consisted of a modified Allison type of repair in 14 cases and a fundal plication in one.
RESULTS T h e r e s u l t s o f a n t i r e f l u x s u r g e r y w e r e d r a m a t i c . Six p a t i e n t s d i d n o t r e q u i r e a n y f u r t h e r d i l a t a t i o n a n d t h e r e m a i n d e r r e q u i r e d o n l y o n e to t h r e e p o s t o p e r a tive dilatations each. Table 1 compares the number of dilatations necessary for each patient before and after antireflux surgery.
DISCUSSION T h e p e r s i s t e n t s t e n o s e s seen in t h e s e c h i l d r e n a f t e r r e p a i r o f e s o p h a g e a l a t r e s i a a n d t r a c h e o e s o p h a g e a l fistula w e r e a p p a r e n t l y d u e to g a s t r o e s o p h a g e a l reflux. T h i s s e e m s to c a u s e o r a g g r a v a t e i n f l a m m a t i o n a n d l e a d s t o u n d u e c i c a t r i z a t i o n at t h e site o f t h e a n a s t o m o s i s . T h e d i a g n o s i s o f reflux is n o t easy. In t h o s e c a s e s w i t h a t i g h t a n d p e r s i s t e n t
Table i. Dilatations Required by Each Patient Before and After Antireflux Surgery Patient
Preoperative Dilatations
Postoperative Dilatations
Age at Operation (yr)
Age Now (yr)
1 2 3 4 5 6 7 8 9 10 11 12 13 14 15
19 12 43 15 11 4 7 9 5 8 18 8 7 5 10
9* 0 2 2 2 2 1 8t" 0 1 1 0 0 0 0
11 1 3/12 1 5 1 8/12 4/12 6/12 9/12 10/12 2/12 8/12 9/12 6/12 6/12 9/12
17 14 14 13 12 8 7 5 2 7/12 2 1 6/12 1 4/12 1 1/12 1 1
*Nine dilatations for gostroesophageal junction stenosis. Reflux cured. t"l:ight esophagoscopies for removal of foreign body due to uncoordinated esophageal motility. Reflux and anastomotic stenosis cured.
RESISTANT ESOPHAGEAL STENOSIS
357
stenosis we m a k e every effort to d e m o n s t r a t e the a s s o c i a t i o n o f the t w o conditions. Several b a r i u m s w a l l o w s a n d e n d o s c o p i c e x a m i n a t i o n s m a y be necessary. O u r results s h o w t h a t the p r o g n o s i s for p e r s i s t e n t a n a s t o m o t i c stenosis in p a t i e n t s with r e p a i r e d e s o p h a g e a l atresia a s s o c i a t e d with g a s t r o e s o p h a g e a l reflux is excellent. In i n s t a n c e s o f p e r s i s t e n t stenosis, g a s t r o e s o p h a g e a l reflux s h o u l d be suspected; if present, it s h o u l d a l w a y s be t r e a t e d . SUMMARY
This p a p e r reviews 15 p a t i e n t s with severe, p e r s i s t e n t sten0sis after r e p a i r o f t r a c h e o e s o p h a g e a l fistula a n d e s o p h a g e a l a t r e s i a o v e r a 15-yr p e r i o d 1958 1973. T h e s e o b s t i n a t e stenoses c o u l d be s e c o n d a r y to g a s t r o e s o p h a g e a l reflux, w h i c h seems to cause i n f l a m m a t i o n a n d u n d u e c i c a t r i z a t i o n at the site o f the a n a s t o m o s i s . A t t e m p t s at c o n s e r v a t i v e m a n a g e m e n t were n o t successful. In all 15 p a t i e n t s antireflux o p e r a t i o n s c u r e d the stenoses. REFERENCES
1. Haight C: The management of congenital esophageal atresia and tracheoesophageal fistula. Surg Clin North Am 41:1281, 1961 2. Holder TM, Ashcraft KW: Esophageal atresia and tracheoesophageal fistula. Ann Thorac Surg 9:445, 1970 3. Holinger PH, Brown WT, Maurizi DG: Endoscopic aspects of post-surgical manage-
ment of congenital tracheoesophageal fistula. J Thorac Cardiovasc Surg 49:22, 1965 4. Schultz LR, Clatworthy HW Jr: Esophageal strictures after anastomosis in esophageal atresia. Arch Surg 87:120, 1963 5. Daum R: Postoperative complications following operation for oesophageal atresia and tracheo-esophageal fistula. Progr Pediatr Surg 1:209, 1970
O
MATERIAL AND METHODS During the period studied, 281 children were treated for esophageal atresia and tracheoesophageal fistula. Of the 217 patients who survived, 77 (35%) developed stenosis of various degrees of severity. Repeated dilatations brought no improvement in 15 of these patients, who showed radiographic and/or endoscopic evidence of reflux with or without hiatal hernia. We reviewed the charts of the 15 patients for clinical features, number of pre- and postoperative dilatations, type of treatment, and results.
CLINICAL FEATURES The patients ranged in age from 2 mo to ll yr. All had dysphagia, regurgitation, recurrent episodes of aspiration, pneumonia, and failure to thrive. End-to-end esophagoesophagostomy with division and closure of the fistula had been done in ten children, and end-to-side procedure with transfixion and/or ligation of the fistula in five. Leakage had occurred within the first postoperative week in five patients. Fistula recurred in four children, two of whom had had an evident leak, and one child had a second recurrence.
RADIOGRAPHIC FINDINGS The diagnosis of reflux and hiatal hernia took some time and effort to establish in these patients (on average, 2.7 esophagograms were required to confirm the diagnosis). In two cases, it was first made by endoscopic examination and subsequently confirmed by radiography. Free reflux without hiatal hernia was found in five cases and with hiatal hernia in nine: in one child, radiography showed a mild degree of stenosis at the gastroesophageal junction presumably due to reflux.
From the Department of Surgery, The Hospital for Sick Children, Toronto, Ontario, Canada. Presented at the American Academy of Pediatrics, Surgical Section, Chicago, Ill., October 20-24, 1973. Raphael Pieretti, M.D.: Chief Resident, Division of General Surgery, The Hospital for Sick Children, Toronto. Barry Shandling, M.B.: Staff Surgeon, The Hospital for Sick Children, Toronto. Clinton A. Stephens, M.D.: Chief, Division of General Surgery, The Hospital for Sick Children, Toronto. Address for reprint requests: Barry Shandling, M.B., The Hospital for Sick Children, 555 University Avenue, Toronto M5G 1)(8, Ontario, Canada. 9 1974 by Grune & Stratton, Inc.
Journal of Pediatric Surgery, Vol. 9, No. 3 (June), 1974
355
356
PIERETTI, SHANDLING, AND STEPHENS
ENDOSCOPIC FINDINGS All 15 patients had tight anastomotic stenoses. Granulation tissue at the site of the anastomosis was seen in three. Eight had distal esophagitis, seven with hiatal hernia, and one with stenosis at the gastroesophageal junction.
TREATMENT Conservative treatment of the reflux was tried without success on six infants. These babies were given thickened feedings in small amounts at frequent intervals and with upright nursing. The average number of dilatations before the antireflux operation was 11 per patient; the frequency varied according to the severity of the stenosis from once every 3 days to once a month. Two patients with massive reflux underwent fewer dilatations because for obvious reasons an antireflux operation was indicated earlier. One child required 43 dilatations, and the diagnosis was missed entirely until a laparotomy established that the upper gastrointestinal bleeding was secondary to reflux esophagitis. The antireflux operation consisted of a modified Allison type of repair in 14 cases and a fundal plication in one.
RESULTS T h e r e s u l t s o f a n t i r e f l u x s u r g e r y w e r e d r a m a t i c . Six p a t i e n t s d i d n o t r e q u i r e a n y f u r t h e r d i l a t a t i o n a n d t h e r e m a i n d e r r e q u i r e d o n l y o n e to t h r e e p o s t o p e r a tive dilatations each. Table 1 compares the number of dilatations necessary for each patient before and after antireflux surgery.
DISCUSSION T h e p e r s i s t e n t s t e n o s e s seen in t h e s e c h i l d r e n a f t e r r e p a i r o f e s o p h a g e a l a t r e s i a a n d t r a c h e o e s o p h a g e a l fistula w e r e a p p a r e n t l y d u e to g a s t r o e s o p h a g e a l reflux. T h i s s e e m s to c a u s e o r a g g r a v a t e i n f l a m m a t i o n a n d l e a d s t o u n d u e c i c a t r i z a t i o n at t h e site o f t h e a n a s t o m o s i s . T h e d i a g n o s i s o f reflux is n o t easy. In t h o s e c a s e s w i t h a t i g h t a n d p e r s i s t e n t
Table i. Dilatations Required by Each Patient Before and After Antireflux Surgery Patient
Preoperative Dilatations
Postoperative Dilatations
Age at Operation (yr)
Age Now (yr)
1 2 3 4 5 6 7 8 9 10 11 12 13 14 15
19 12 43 15 11 4 7 9 5 8 18 8 7 5 10
9* 0 2 2 2 2 1 8t" 0 1 1 0 0 0 0
11 1 3/12 1 5 1 8/12 4/12 6/12 9/12 10/12 2/12 8/12 9/12 6/12 6/12 9/12
17 14 14 13 12 8 7 5 2 7/12 2 1 6/12 1 4/12 1 1/12 1 1
*Nine dilatations for gostroesophageal junction stenosis. Reflux cured. t"l:ight esophagoscopies for removal of foreign body due to uncoordinated esophageal motility. Reflux and anastomotic stenosis cured.
RESISTANT ESOPHAGEAL STENOSIS
357
stenosis we m a k e every effort to d e m o n s t r a t e the a s s o c i a t i o n o f the t w o conditions. Several b a r i u m s w a l l o w s a n d e n d o s c o p i c e x a m i n a t i o n s m a y be necessary. O u r results s h o w t h a t the p r o g n o s i s for p e r s i s t e n t a n a s t o m o t i c stenosis in p a t i e n t s with r e p a i r e d e s o p h a g e a l atresia a s s o c i a t e d with g a s t r o e s o p h a g e a l reflux is excellent. In i n s t a n c e s o f p e r s i s t e n t stenosis, g a s t r o e s o p h a g e a l reflux s h o u l d be suspected; if present, it s h o u l d a l w a y s be t r e a t e d . SUMMARY
This p a p e r reviews 15 p a t i e n t s with severe, p e r s i s t e n t sten0sis after r e p a i r o f t r a c h e o e s o p h a g e a l fistula a n d e s o p h a g e a l a t r e s i a o v e r a 15-yr p e r i o d 1958 1973. T h e s e o b s t i n a t e stenoses c o u l d be s e c o n d a r y to g a s t r o e s o p h a g e a l reflux, w h i c h seems to cause i n f l a m m a t i o n a n d u n d u e c i c a t r i z a t i o n at the site o f the a n a s t o m o s i s . A t t e m p t s at c o n s e r v a t i v e m a n a g e m e n t were n o t successful. In all 15 p a t i e n t s antireflux o p e r a t i o n s c u r e d the stenoses. REFERENCES
1. Haight C: The management of congenital esophageal atresia and tracheoesophageal fistula. Surg Clin North Am 41:1281, 1961 2. Holder TM, Ashcraft KW: Esophageal atresia and tracheoesophageal fistula. Ann Thorac Surg 9:445, 1970 3. Holinger PH, Brown WT, Maurizi DG: Endoscopic aspects of post-surgical manage-
ment of congenital tracheoesophageal fistula. J Thorac Cardiovasc Surg 49:22, 1965 4. Schultz LR, Clatworthy HW Jr: Esophageal strictures after anastomosis in esophageal atresia. Arch Surg 87:120, 1963 5. Daum R: Postoperative complications following operation for oesophageal atresia and tracheo-esophageal fistula. Progr Pediatr Surg 1:209, 1970