Surgical Treatment of Reflux Stricture of the Esophagus By Youkatsu Ohhama, Akio Tsunoda, Toshiji Nishi, Ryoji Yamada, and Hiroshi Y a m a m o t o Yokohama, Japan 9 Ten children, aged 7 months to 15 years, with peptic esophageal stricture, were treated surgically. In four of the children, the stricture had occurred after esophageal anastomosis. Peptic stricture was diagnosed by esophagography, pH monitoring, manometry, and esophagoscopy. Barrett's esophagus was found in t w o children. Nine children underwent transabdominal Nissen fundoplication initially. In the first child of this series, a tight anastomotic stricture had been excised 2 weeks before fundoplication. Seven children became complaint-free within 2 or 3 months after fundoplication without any dilatation, and two children with anastomotic stricture improved after 1 or 2 postoperative dilatations. The condition of one boy, with a 6-year history of tight stricture, did not improve with repeat Nissen and subsequent dilatations. Histological examination showed proliferation of smooth muscle cells in the submucosa. A conservative surgical approach is effective for the management of peptic esophageal stricture in children, and direct surgical intervention for stricture should be attempted only in cases of stricture resistant to antireflux surgery with a long history of reflux. 9 1990 by W.B. Saunders Company. INDEX WORDS: Gastroesophageal reflux; esophageal stricture; Nissen fundoplication.

H A L A S I A or gastroesophageal reflux (GER) is a well-recognized entity in childhood. If the symptoms persist long enough, chronic esophagitis may lead to stricture formation. From studies by Carre 1 to define the natural history of GER, patients who continued to suffer from troublesome symptoms up to the age of 4 years or longer were considered to be at risk of developing esophageal stricture, and this group represented about 5% of all patients in his study. However, other reported incidences of esophageal stricture associated with GER range from 2.5% to 50% of all patients undergoing antireflux surgery. 2-4 This complication is considered to be an indication for antireflux surgery. In a significant proportion of cases, antireflux surgery has successfully restored normal esophageal function. Surgery for GER with stricture formation is more

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complicated than surgery for GER alone. In this paper, we review our experience at Kanagawa Children's Medical Center with regard to the diagnosis and therapy of peptic reflux stricture of the esophagus and their outcome. MATERIALS AND METHODS

Between 1971 and 1988, 53 children underwent antireflux surgery. Of these, 10 children aged between 7 months and 15 years had peptic esophageal stricture. The most common symptoms were vomiting and dysphagia. The onset of documented symptoms of reflux ranged from shortly after birth to 14 years of age. The average period from onset of GER symptoms to surgery was 1.5 years (range, 3 months to 4.9 years). However, in one patient who responded favorably to position therapy, esophageal stricture developed at 6 months of age without any documented symptoms. There was a history of repair of esophageal atresia in three patients, and excision of congenital lower esophageal stenosis in one patient. Three developed a small leak at the anastomosis, which was subsequently sealed, and thereafter suffered anastomotie stricture requiring esophageal dilatation on two or more occasions to control dysphagia before antireflux surgery was performed. Two children had significant neurological impairment. For patients with lower or midesophageal stricture, 24-hour pH monitoring was performed, placing the pH probe above the stricture, or for patients with upper stricture, 2 to 3 cm above the upper border of the lower esophageal sphincter (LES) as measured by manometry. By calculating the number of standard deviation equivalents about percent time pH under four and number of refluxes over 5 minutes, the pH score consists of scores for two factors. The usefulness of the pH score for evaluation of GER has been reported previously.5 Esophageal manometry was performed using the technique of slow constant pull-through. A catheter-tip pressure transducer was used to measure the pressure in the LES at end expiration, as well as the length of the lower esophageal high-pressure zone. The patients have been followed-up for between 2.7 and 12 years from the time of surgery (average, 7.2 years). RESULTS

From the Department of Surgery, Kanagawa Children's Medical Center, Yokohama, Japan. Presented at the 22nd Annual Meeting of the Pacific Association of Pediatric Surgeons, Portland and Sun River, Oregon, May 22-26, 1989. Address reprint requests to Youkatsu Ohhama, MD, Kanagawa Children's Medical Center, Mutsukawa 2-138-4, Minami-Ku, Yokohama, 232 Japan. 9 1990 by W.B. Saunders Company. 0022-3468/90/2507-0013503.00/0

M u l t i p l e d i a g n o s t i c tests w e r e u s e d for e v a l u a t i n g b o t h t h e c a u s e of e s o p h a g e a l s t r i c t u r e a n d t h e s y m p toms. B a r i u m c o n t r a s t e x a m i n a t i o n d e m o n s t r a t e d an e s o p h a g e a l s t r i c t u r e in all 10 p a t i e n t s . T h r e e s t r i c t u r e w e r e in t h e u p p e r e s o p h a g u s , t w o w e r e in t h e m i d d l e e s o p h a g u s , a n d five in t h e distal t h i r d o f t h e e s o p h a g u s . In f o u r p a t i e n t s t h e s t r i c t u r e was at t h e level of a p r e v i o u s r e p a i r of e s o p h a g e a l a t r e s i a or c o n g e n i t a l l o w e r e s o p h a g e a l stenosis t h a t h a d b e e n excised transabdominally. S e v e n of t h e 10 p a t i e n t s w e r e f o u n d to h a v e associa t e d h i a t u s h e r n i a . In n i n e o f t h e p a t i e n t s , b a r i u m c o n t r a s t studies d e m o n s t r a t e d G E R . In e i g h t p a t i e n t s , 2 4 - h o u r e s o p h a g e a l p H m o n i t o r i n g

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Journal of Pediatric Surgery, Vol 25, No 7 (July), 1990: pp 758-761

TREATMENT OF REFLUX ESOPHAGEAL STRICTURE

was performed, and six were found to have a high pH score. One patient with a large hiatus hernia had no apparent reflux and had a normal score. Eight of the 10 patients underwent esophageal manometry. In six, LES pressure was lower than in normal subjects, and the LES length was short for age. Endoscopy with biopsy of the stricture, or of the area immediately below it, was performed in all 10 patients. A fiberscope 7 mm in diameter could not be passed through the stricture. Biopsy specimens were positive for esophagitis in nine, and diagnosis of Barrett's esophagus was made in two. When the diagnosis of persistent esophageal stricture associated with GER was apparent+ antireflux surgery was performed. A total of 12 Nissen fundoplications were performed in 10 patients using the transabdominal 360 ~ wrap-around technique. Two patients underwent a repeat Nissen after the first procedure was disrupted within 6 months after surgery. A tube gastrostomy was placed in the two severely retarded children for feeding purpose. The gastric drainage operation, such as pyloromyotomy or pyloroplasty, was performed in all patients. Dilatation of the anastomotic stricture was performed in two cases before associated GER was diagnosed. However, dysphagia failed to improve in both patients. There were three incidences of surgical complication. These were two disrupted Nissen fundoplications, which were subsequently repeated, and one esophageal perforation at the site of stricture during esophageal dissection, which was closed longitudinally. Nine patients underwent Nissen fundoplication initially. In the first patient of this series, a tight anastomotic stricture had been excised 2 weeks before fundoplication. In nine patients it was possible to position the fundoplication within the abdomen with closure of the crural defect. Only in one instance was it necessary to place part of the fundoplication within the mediastihum because of a severely short esophagus. Twenty-four-hour pH monitoring was performed postoperatively, demonstrating control of GER in all patients. Nine patients became complaint-free within 2 to 3 months after fundoplication. More importantly, there was resolution of the stricture, and seven patients needed no postoperative esophageal dilatation. Two patients with anastomotic stricture required one or two dilatations before resolution of the stricture. The remaining one patient who had undergone repeated Nissen showed no improvement of dysphagia and mid esophageal stricture, even after several postoperative dilatations with dissection of the web by electrocautery under endoscopy. He had a 6-year history of persistent GER and Barrett's esophagus. While waiting for resection of the web in the midesophagus, he died of

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septicemia 3 years after the repeat Nissen. Histological examination showed proliferation of smooth muscle cells in the submucosa at the stricture. Of the four anastomotic strictures, only one was resolved without the need for postoperative dilatation. On the other hand, five of six strictures with hiatus hernia were improved both radiographically and clinically without any postoperative dilatation. DISCUSSION

It is established that diagnosis of GER may be made by various methods. We believe that the usual tests for demonstrating reflux in patients with established esophageal stricture may vary according to the type of stricture present. In this series, one of 10 patients did not show GER on barium contrast study. Another who had a tight anastomotic stricture after previous repair of esophageal atresia did not demonstrate GER, but after bouginage free reflux was demonstrated by barium examination. One of eight patients who underwent 24-hour pH monitoring had a normal pH score. It is our belief that in some instances the stricture produced by the GER may act as a barrier to reflux higher in the esophagus, and that the flutter valve action of the esophagus produced by a herniated stomach may also act as a barrier to reflux. Therefore, repeated studies may be necessary in order to show GER in patients with persistent esophageal stricture. A 20% incidence of anastomotic stricture after repair of esophageal atresia has been reported. 6 Patients who require early dilatation of symptomatic postoperative anastomotic narrowing should be examined for anastomotic leakage or recurrent tracheoesophageal fistula. After these have been excluded, persistent anastomotic narrowing may be due to GER. Recognition that reflux was the causal factor responsible for persistent anastomotic narrowing in three of the seven patients with severe GER in this series, who had undergone previous repair of esophageal atresia, allowed antireflux surgery to be performed. Hill et al 7 and others 8 have promoted a conservative surgical approach to the management of reflux peptic strictures of the esophagus in adults. They feel that correction of GER with both concomitant and postoperative dilatation as necessary is sufficient in most instances, and suggest that more aggressive forms of surgical management for esophageal stricture should not commonly be required. Randolph 9 and Leape t~ have stated that most cases of stricture in children respond if reflux is eliminated. Our experience of one case in which the esophagus was injured and had to be sutured longitudinally, followed by complete healing after reflux had been eliminated, tends to support this notion.

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More conservative methods have also been suggestedJ 1 That is, bougienage and aggressive medical therapy are reported to be effective for most benign peptic esophageal strictures in adults. However, because most of such patients require multiple dilatations for a year or more, such an approach would hardly be acceptable for most infants and children, who might suffer from irreparable growth retardation during that time, and the need for repeated dilations would expose children to the risks of repeated anesthesia and esophageal perforation. Dilatation or surgical correction of a peptic stricture prior to correction of reflux is usually bound to fail. Although Monero et a112 expected that preoperative dilatation might provide a better opportunity to improve nutrition in children with weight loss prior to surgery, this approach has not worked effectively. Today we know that intravenous hyperalimentation with cimetidine therapy before antireflux surgery is more effective than one or two dilations, making oral food intake possible. One of the patients showed improved nutrition by tube enteral feeding with cimetidine therapy. The consensus in the conflict about surgical management of reflux esophageal stricture presently favors conservative antireflux procedures rather than resection. ~3"~4We also feel that concomitant and postoperative dilatation is unnecessary for most patients, especially for stricture unrelated to anastomosis. However, in one patient, stricture had not been improved by repeat Nissen and subsequent dilatations. Because he had a 6.7-year history of persistent GER, it seemed that there was a time limit for effective resolution of stricture using conservative means. The histological features of the stricture in this case were markedly different from the scarring stricture commonly noted, and rather resembled the rare condition of congenital lower esophageal stenosis due to muscular hypertrophy. However, it was unclear whether the muscle cell proliferation was a late consequence of reflux esophagitis, or a consequence of rebound proliferation after electrocautery extending into the lamina muscularis propria. The Nissen fundoplication is our antireflux procedure of choice. This procedure has proved to be the most effective means of controlling reflux. The most important point concerning surgery is the procedure that should be selected for cases of short esophagus. We were able to reposition the sphincter in the abdomen and performed repair under slight tension in all patients except one. In a 15-year-old girl with a long stricture and short esophagus, we could not perform tension-free repair. After abdominal wrapping, we left

OHHAMA El" AL

part of the fundic cuff in the mediastinum, and anchored the cuff to the crural edge in its natural position. This procedure is easily performed, and some have recommended that the Nissen procedure in the mediastinum is useful in patients with an acquired short esophagus when the surgeon cannot reduce the wrap below the diaphragm.15 However, a rare postoperative complication of ulceration within the wrap itself has been reported. 16 Postoperative en bloc herniation into the mediastinum is similar to positioning the Nissen fundic cuff in the mediastinum with regard to shape and efficacy against GER. We have followed a child who underwent fundoplication at another hospital, whose fundic cuff had herniated en bloc into the mediastinum, and he has been complaint-free for over 10 years. Also, the previously mentioned female case has been complaint-free for 5 years postoperatively. On the other hand, some groups have pointed out the value of the Collis-Nissen procedure for cases of short esophagus. 17 However, Collis-Nissen provides physiological unnatural shape in comparison with simple Nissen; furthermore, it is undesirable that a Collis gastric tube, which is impossible to restore, will be applied in growing children. In fact, follow-up data about this procedure are limited even in adults. For these reasons, we prefer to place the Nissen in the mediastinum, as previously mentioned. Floppy Nissen is best for avoiding excess narrowing of the lumen and subsequent gas-bloat syndrome. The procedure for wrapping the abdominal esophagus is an important technical point of the Nissen operation. Although the tightness of the fundoplication is usually determined by inserting a bougie down the esophagus, the size of bougie suitable for children of various ages is a difficult problem to solve. Hill ~s has developed the technique of intraoperative manometry. It has been suggested that esophageal pressure monitoring during Nissen fundoplication may be a helpful guide for avoiding gas-bloat syndrome, and some reports have recommended an adequate pressure for wrap-around, although there are no clinical guidelines for practical techniques of controlling wrap pressure, as in Hill's procedure. In practice, we do not perform either insertion of a bougie down to esophagus or intraoperative manometry. Our standard Nissen is performed using a simple technique (Fig 1). After measuring the abdominal esophageal circumference, we design a fundic wrap to fit it. Like fitting a shirt according to neck size, fundoplication was made by a fundic wrap fitted to the size of the abdominal esophagus. The length of the fundic cuff is defined as 1.5 to 2.5 cm depending on the age of the patients, and no more than 3 cm. We have

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Fig 1. Technique of our fundoplication procedure. A f t e r measuring the abdominal esophageal circumferance, a fundic wrap is made to fit it. Then w e w r a p the abdominal esophagus left posteriorly and suture it with five stitches.

never experienced s y m p t o m a t i c g a s - b l o a t s y n d r o m e in patients t r e a t e d with this simple technique. T h e indication of c o n c o m i t a n t gastric d r a i n a g e operation is a controversial one. Based on clinical experiences, we have had an impression that the resting i n t r a g a s t r i c pressure after Nissen seems to be slightly higher than t h a t of h e a l t h y children, and when subclinical elevation o f i n t r a g a s t r i c pressure is persistently present, the risk of cuff disruption m i g h t be increased. Therefore, a c o n c o m i t a n t gastric d r a i n a g e p r o c e d u r e is essential for severe G E R e v a l u a t e d by p H monitoring. It is speculated t h a t p y l o r o m y o t o m y m i g h t be sufficient for gastric d r a i n a g e . F r o m our experience, we conclude t h a t a conservative surgical a p p r o a c h is effective for the m a n a g e m e n t of peptic esophageal stricture in children, and t h a t direct surgical intervention is unnecessary. However, patients with stricture resistant to antireflux surgery and with a long history of reflux should be t r e a t e d b y resection. It is far better to p e r f o r m fundoplication in patients with chronic G E R prior to the time of stricture formation.

REFERENCES

1. Carre LJ: Postural treatment of children with a partial thoracic stomach (hiatus hernia). Arch Dis Child 35:569-580, 1960 2. O'Neil JA Jr, Betts J, Ziegler MM, et al: Surgical management of reflux stricture of the esophagus in childhood. Ann Surg 196:453-459, 1982 3. Berlaizky Y, Choen OM, Freund HR, et al: Surgical treatment of gastroesophageal reflux with esophageal stricture in infancy and childhood. Am J Surg 143:205-208, 1982 4. Hicks LM, Christie DL, Hall DG, eta[: Surgical treatment of esophageal stricture secondary to gastroesophageal reflux. J Pediatr Surg 15:863-868, 1980 5. Ohhama Y, Tsunoda A, Nishi T, et al: Twenty-four hour pH monitoring for children--A special criteria for evaluation for gastroesophageal reflux. Jpn J Pediatr Surg 14:647-656, 1982 6. Strodel WE, Coran AG, Kirsh MM, et al: Esophageal atresia, a 41-year experience. Arch Surg 114:523-527, 1979 7. Hill LD, Gelfand M, Bauermeister D: Simplified management of reflux esophagitis with stricture. Ann Surg 172:638-651, 1970 8. Larraian A, Csendes A, Pope IICE: Surgical correction of reflux, an effective therapy for esophageal stricture. Gastroenterology 69:578-583, 1975 9. Randolph JG: Hiatal hernia and gastroesophageal reflux, in Ravitch MM, Welch KJ, Benson CD, et al (eds): Pediatric Surgery. Chicago, IL, Year Book, 1979, pp 475-482

10. Leape LL: Gastroesophageal reflux, in Holder TM, Ashcraft KW (eds): Pediatric Surgery. Philadelphia, PA, Saunders, 1980, pp 292-312 11. Lanza FL, Graham DY: Bougienage is effective therapy for most benign esophageal stricture. JAMA 240:844-847, 1978 12. Monereo J, Cortes L, Blesa E: Peptic esophageal stenosis in children. J Pediatr Surg 8:475-478, 1973 13. Mercer CD, Hill LD: Surgical management of peptic esophageal stricture, twenty-year experience. J Thorac Cardiovasc Surg 91:371-378, 1986 14. Watson A: Reflux stricture of the esophagus. Br J Surg 74:443-448, 1987 15. Maher JW, Hocking MP, Woodward ER: Supradiaphragmatic fundoplication: Long-term follow-up and analysis of complications. Am J Surg 147:181-186, 1984 16. Richardson JD, Larson GM, Polk HC: Intrathoracic fundoplication for shortened esophagus: Treacherous solution to a challenging problem. Am J Surg 143:29-35, 1982 17. Orringer MB: The combined Collis-Gastroplasty-Nissen fundoplication for gastroesophageal reflux, in Demeester TR, Skinner DB (eds): Esophageal Disorder: Pathophysiology and Therapy. New York, NY, Raven, 1985, pp 203-208 18. Hill LD: Intraoperative management of lower esophageal sphincter pressure. J Thorac Cardiovasc Surg 78:378-382, 1978