Dysphagia 7:234-237 (1992)
l)ysphagia r Springer-VerlagNew York Inc. 1992
Oral Dysfunction Following Nissen Fundoplication 5
Stephen M. Borowitz, M.D. and Kathleen C. Borowitz, M.S. Department o f Pediatrics, Divisions of Gastroenterology and Speech Pathology, University of Virginia Health Sciences Center, Charlottesville, Virginia, USA
In this case report we describe a child with mild neurologic impairment who developed debilitating gagging and retching, and severe oralmotor dysfunction following Nissen fundoplication and gastrostomy tube placement. All oral intake ceased after the operation. Evaluation for postoperative dumping syndrome was negative, and the child's symptoms failed to improve despite numerous medical and surgical measures. However, immediately following reversal of the Nissen fundoplication, the child's gagging and retching ceased, and his oral-motor function began to improve. This is a previously undescribed complication of Nissen fundoplication, a surgical procedure commonly employed in children with neurologic impairment.
Abstract.
Key words:
Nissen fundoplication -- Developmental delay -- Oral-motor function -- Dysphagia - - Gastroesophageal reflux -- Deglutition -- Deglutition disorders.
In children with neurologic impairment, feeding gastrostomies are often used to allay chronic malnutrition and severe feeding difficulties [1]. Symptomatic gastroesophageal reflux is thought to be quite common among these children [2, 3], and recent studies suggest the incidence ofgastroesophageal re-
flux may increase following gastrostomy placement [4]. As a consequence, there has been a trend to perform "protective" antireflux surgery in developmentally disabled children who need feeding gastrostomies [3, 5]. Nissen fundoplication is the most widely employed antireflux procedure in children, and is an effective means of eliminating gastroesophageal reflux [6-8]. While serious perioperative complications are uncommon with Nissen fundoplication, we have become aware of subtle long-term postoperative morbidity associated with this surgical procedure. Of 46 children who have undergone Nissen fundoplication during the past 2 years, we have observed profound postoperative oral-motor dysfunction in four children with neurologic impairment. The oral dysfunction was initially manifested as severe gagging and retching beginning 1 to 4 weeks postoperatively. This was followed by rapid cessation of oral intake associated with pronounced aversive reactions to presentation of food such as lip pursing, head shaking, turning away and crying, or "fighting." In some cases, even nonnutritive oral stimulation resulted in these aversive behaviors. In one case, oral-motor dysfunction was so severe that the fundoplication was taken down. The reversal of surgery resulted in dramatic improvement. That child's history is summarized below. Case Report
Address offprint requests to: Stephen M. Borowitz, M.D., Department of Pediatrics, Box 386, University of Virginia Health Sciences Center, Charlottesville, VA 22908, USA
L.L. is currently a 36-month old caucasian boy. He weighed 2.8 kg at birth. His mother was a 26-year old primigravida who received good prenatal care, and her full-term pregnancy was uncomplicated. L.L. was delivered by emergency cesarian section due to fetal distress. Apgar scores were 5 at 1 and 7 at 10 rain.
S.M. Borowitz and K.C. Borowitz: Oral Dysfunction Following Fundoplication He developed generalized seizures during the first 24 h of life, characterized by bilateral fisting and bicycling; the seizures were easily controlled with phenobarbital. Physical examination was remarkable only for mild hypotonia. Laboratory evaluation was remarkable only for thrombocytopenia which resolved spontaneously over 5 days. Electroecephalogram during the first week of life demonstrated diffuse slowing without any focal abnormalities. Cranial computerized tomography (CT) revealed mildly dilated third and fourth ventricles and patchy periventricular calcifications. Viral cultures and serologies were negative, as were bacterial cultures. His nursery course was complicated by slow oral feeding, and he was discharged receiving supplemental nocturnal nasogastric feedings providing half of his calories. Oral examination at 1 month of age was remarkable for generalized hypotonia, a retracted mandible, posterior tongue carriage, and oral hypersensitivity. A weak and arrhythmical suck was noted during bottle feeding. At 3 months of age, L.L. was admitted to the hospital because of feeding failure. He continued to take half of his calories orally and received the remainder at night through a nasogastric tube. Little change was evident in his oral function or feeding skills. He had no coughing or gagging during feeding. Generalized hypotonia continued to affect his positioning and ability to achieve and maintain a lip seal on the nipple during bottle drinking. An upper gastrointestinal series demonstrated normal anatomy without nasopharyngeal or gastroesophageal reflux. Esophageal motility appeared normal. Hexible endoscopy was normal and esophageal biopsies did not reveal any inflammatory changes. A surgical gastrostomy and Nissen fundoplication were performed without complication. Gastrostomy feedings were begun on the third postoperative day, and oral feedings were restarted 3 days later. At the time of his hospital discharge on the seventh postoperative day, L.L. was taking 2 ounces of formula every 3 h by mouth (half of his calories), with nighttime supplements through the gastrostomy. A repeat upper gastrointestinal series through the gastrostomy tube prior to discharge showed a normal fundoplication without esophageal obstruction. Gastric emptying was normal. Over the 10 days following his discharge, L.L. began gagging and retching with gastrostomy feedings and his oral intake diminished. Within 1 m o n t h of surgery, all oral intake had ceased. He began to experience episodes of irritability, crying, gagging, and clamminess, often, hut not always associated with gastrostomy feedings (2-3 ounces of Pediasure given over 2 h). A repeat upper gastrointestinal series was performed through the gastrostomy tube 2 months after surgery and showed the expected narrowing of the distal esophagus at the site of the fundoplication. Over the ensuing month, L.L. continued to experience severe gagging and retching episodes no longer clearly associated with feedings. Repeat upper gastrointestinal series 2 weeks later (3.5 months following fundoplication) displayed mild narrowing at the site of the fundoplication without gastroesophageal reflux. Rigid esophagoscopy and esophageal dilatation to 18 m m were performed. Feeding evaluation 2 weeks later revealed refusal of all oral intake with intensified aversive reactions, increased drooling, and marked oral defensiveness even with nonnutritive oral stimulation. Additionally, abnormal oral reflexes were observed including jaw thrust, tongue protrusion, tongue fasciculations, and a very prolonged and prominent gag reflex. Evaluation for dumping syndrome was negative; an oral glucose tolerance test was normal, as were multiple postprandial glucose measurements. The gagging and retching persisted and L.L. was readmitted to the hospital 1 month later. Upper gastrointestinal series dem-
235
onstrated a normal fundoplication without esophageal stenosis. Hexible endoscopy was normal with a patent esophagus and pylorus. Esophageal, gastric, and duodenal biopsy specimens were normal, A pyloroplasty was performed with the hopes of improving gastric emptying however the gagging and retching continued unchanged. Trials of continuous infusion of an elemental formula, pectin, sucralfate, rantidine, and glycopyrrolate afforded no improvement. One year after the initial surgery, the fundoplication was taken down. At the time of surgery, the esophageal wrap appeared intact, and no other abnormalities were noted. L.L. experienced minimal vomiting following the operation, and by the time of his discharge 10 days later he was tolerating a constant infusion of Pediasure at 35 m l / h through his gastrostomy. Gagging and retching completely abated immediately after surgery. It is now 24 months since the fundoplication has been reversed. L.L.'s gagging and retching have completely resolved, and he rarely vomits. His oral-motor function has gradually improved. He has significantly less oral defensiveness, and his jaw and tongue thrusts are less prominent. He now shows some nonnutritive sucking and tolerates tactile stimulation to his face, lips, and gums. His drooling has diminished, and he is currently tolerating bolus gastrostomy feedings without difficulty and eating small amounts of strained foods by spoon. He has begun to spontaneously bring toys, spoons, and bottles to his mouth during play. Since the reversal of the fundoplication, the velocity of weight gain and linear growth have improved significantly.
Discussion
Nissen fundoplication is generally thought to be a safe and effective antireflux procedure. Perioperative mortality is generally less than 1%, and major postoperative complications occur in less than 1 5 o of cases [7, 8]. The most common serious postoperative complications are herniation or breakdown of the esophageal wrap and bowel obstruction [8, 9]. During the perioperative period, fundoplication eliminates gastroesophageal reflux in greater than 90% of patients [6-8]. Few authors have followed patients for prolonged periods following surgery to determine whether there is recurrence of symptomatic reflux. In a retrospective study of 60 adults, Negre found a 10-year failure rate of nearly 20% [10]. Randolph followed 67 infants for between 1 and 10 years following fundoplication and documented a 10% failure rate [ 11 ]. Unfortunately, there have been no prospective long-term studies of children following Nissen fundoplication. Subtle postoperative complications may occur quite commonly following Nissen fundoplication. In their retrospective review of 25 children evaluated 4 to 9 years postoperatively, Harnsberger et al. found 36% had significant symptoms attributable to surgery; 32% of the children had difficulties with abdominal distention and discomfort (gas-bloat), 32% had major changes in their eating habits after the surgery, and 25% suffered intermittent solid dys-
236
S.M. Borowitzand K.C. Borowitz: Oral Dysfunction FollowingFundoplication
phagia [12]. O f the 226 adults Negre assessed between 3 and 12 years following the operation, 44% had major changes in their eating habits, 38% suffered from excessive flatus, abdominal distension and discomfort (gas-bloat), and 19% were unable to burp [ 13]. The patient we describe above suffered severe retching and gagging postoperatively. While neither Harnsberger et al. nor Negre mention these distressing symptoms, Stringel et al. observed retching in 13% of 90 children with neurologic impairment following fundoplication [9], and Jolley et al. noted postoperative retching in 18% of the 66 children he evaluated [ 14]. In six ofJolley et al.'s twelve patients the retching was attributed to postoperative dumping syndrome. Dumping syndrome is a complex of postprandial symptoms which may include abdominal distention, diarrhea, irritability, refusal to feed, weakness, and pallor. Dumping syndrome may occur frequently following Nissen fundoplication and generally appears during the first several weeks following surgery [15]. While the pathophysiology of the syndrome is not completely understood, many of the symptoms may be attributable to postprandial hypoglycemia. An oral glucose tolerance test appears to be a sensitive diagnostic measure and demonstrates early hyperglycemia followed by reactive hypoglycemia [15]. While many of L.L.'s symptoms were suggestive of,dumping syndrome, he did not experience any diarrhea, and oral glucose tolerance tests were normal on two occasions. Moreover, he failed to respond to those therapeutic measures often helpful in dumping syndrome such as hyposmolar feedings, constant infusion feedings, and pectin. In six of the twelve patients Jolley et al. described with postoperative retching, dumping syndrome was not present and symptoms were attributed to decreased gastric emptying [14]. Some authors have suggested that performing a gastric outlet procedure to facilitate gastric emptying may lessen the incidence of postoperative retching [16]. While pyloroplasty failed to improve our patient's symptoms, reversal of the fundoplication immediately eliminated his gagging and retching. In addition to L.L., we have observed severe postoperative retching in three other children with neurologic impairment. In all four of these children, retching was associated with rapid decline of oral intake and profound deterioration of oral-motor function. While retching resolved spontaneously over 6 months in two of these four children, oralmotor dysfunction has persisted. In the ease presented above, it was only when the fundoplication
was reversed that oral function began to improve. In none of the published follow-up studies is any mention made of oral function either before or following fundoplication, nor is there any discussion of feeding skills or the method of feeding employed. Among families with children with neurologic impairment, feeding issues are often emotionally charged [17]. Many of these families find feeding their child a gratifying experience; while feeding gastrostomy may be employed to ease the difficulties associated with feeding some of these children, it should not necessarily be used to eliminate all oral intake. While we have observed a variety of complications following Nissen fundoplication in normal children, we have not seen in them the dramatic changes in oral-motor function that we have observed in children with neurologic impairment. Many children with neurologic impairment have difficulty with oral motor control. They may exhibit delayed swallowing reflexes, inefficient swallows, and abnormal swallow frequency which can lead to excessive drooling [18, 19]. They may also exhibit poor coordination between swallowing and ventilation partly explaining their predilection for silent aspiration and respiratory tract infections [ 19, 20]. It is unclear why oral function deteriorates following fundoplication in children with neurologic impairment. Swallowing is an extremely complex motor sequence which depends upon activity of medullary interneurons in the swallowing center. While swallowing is largely reflexive, there is a large voluntary component, and swallowing neurons receive strong inhibitory input from vagal afferent fibers [21]. Some of these afferent fibers may arise from the numerous mechanoreceptors in the distal esophagus, gastroesophageal junction, and gastric fundus [22]. Fundoplication may cause chronic mechanoreceptor activation which inhibits spontaneous activity in the central swallowing center. This may explain some of the changes in eating habits following Nissen fundoplication. Normal children may overcome these inhibitory inputs through the conscious initiation of swallows. In contrast, children with neurologic impairment who already have disordered swallowing and poor oral motor control may be unable to overcome the inhibitory inputs. In our case, reversal of the fundoplication may have alleviated swallow inhibition and facilitated restoration of oral function. The restoration of oral function following reversal of the fundoplication suggests that the problems were not caused by disruption of vagal fibers, but rather by mechanical interference.
S.M. Borowitz and K.C. Borowitz: Oral Dysfunction Following Fundoplication With this report, we relate a previously undescribed long-term complication of Nissen fundoplication. In future studies e x a m i n i n g the m e d i c a l and surgical m a n a g e m e n t o f g a s t r o e s o p h a g e a l reflux in n e u r o l o g i c a l l y i m p a i r e d c h i l d r e n , it w o u l d s e e m w a r r a n t e d to p r o s p e c t i v e l y e v a l u a t e o r a l f u n c t i o n a n d f e e d i n g skills.
References 1. Rempel GR, Colwell SO, Nelson RP: Growth in children with cerebral palsy fed via gastrostomy. Pediatrics 82:857862, 1988 2. Sondheimer JM, Morris BA: Gastroesophageal reflux among severely retarded children. J Pediatr 94:710-714, 1979 3. Wesley JR, Coran AG, Sarahan TM, Klein MD, White SF: The need for evaluation of gastroesophageal reflux in braindamaged children referred for feeding gastrostomy. J Pediatr Surg 16:866-871, 1981 4. Mollitt DL, Golladay ES, Seibert J J: Symptomatic gastroesophageal reflux following gastrostomy in neurologically impaired patients. Pediatrics 75:1124-1126, 1985 5. Jolley SJ, Smith El, Tunell WP: Protective antireflux operation with feeding gastrostomy; experience with children. Ann Surg 201:736-740, 1985 6. Fonkalsrud EW, Foglia RP, Ament ME, Berquist W, Vargas F: Operative treatment for the gastroesophageal reflux syndrome in children. J Pediatr Surg 24:525-529, 1989 7. Vane DW, Harmel RP, King DR, Boles ET: The effectiveness ofNissen fundoplication in neurologically impaired children with gastroesophageal reflux. Surgery 98:662-666, 1985 8. Dedinsky GK, Vane DW, Black CT, Turner MK, West KW, Grosfield FL: Complications and reoperation after Nissen fundoplication in childhood. Am J Surg 153:177-183, 1987 9. Stringel G, Delgado M, Guertin L, Cook JD, Maravilla A, Worthen H: Gastrostomy and Nissen fundoplication in neurologically impaired children. J Pediatr Surg 24:1044-1048, 1989 10. Negre JB, Markkula HT, Keyrilaninen O, Matikainen M:
237
Nissen fundoplication--results at ten year follow-up. Am J Surg 146:635-638, 1983 11. Randolph J: Experience with the Nissen fundoplication for correction of gastroesophageal reflux in infants. Ann Surg 198:579-584, 1983 12. Harnsberger JK, Corey J J, Johnson DG, Herbst FF: Longterm follow-up of surgery for gastroesophageal reflux in infants and children. J Pediatr 102:505-508, 1983 13. Negre JG: Post-fundoplication symptoms--do they restrict the success of Nissen fundoplication? Ann Surg 198:698700, 1983 14. Jolley SG, Tunell WP, Leonard JC, Hoelzer DF, Smith EI: Gastric emptying in children with gastroesophageal reflux. II. The relationship to retching symptoms following antireflux surgery. J Pediatr Surg 22:927-930, 1987 15. Caulfied ME, Wyllie R, Firor HV: Dumping syndrome in children. J Pediatr 110:212-215, 1987 16. Papaila JG, Wilmot D, Grosfeld JL, Grosfield FL, Rescarta FJ, West KW, Vane DW: Increased incidence of delayed gastric emptying in children with gastroesophageal reflux. Arch Surg 124:933-936, 1989 17. Jones PM: Feeding disorders in children with multiple handicaps. Dev Med Child Neuro131:404-406, 1989 18. Sochaniwsky AE, Koheil RM, Bablich K: Oral motor functioning, frequency of swallowing and drooling in normal children and in children with cerebral palsy. Arch Phys Med Rehabil 67:866-874, 1986 19. Helfrich-Miller KR, Rector KL, Straka JA: Dysphagia: its treatment in the profoundly retarded patient with cerebral palsy. Arch Phys Med Rehabi167:520-525, 1986 20. Kenny D J, Casas M J, McPherson KA: Correlation of ultrasound imaging of oral swallow with ventilatory alterations in cerebral palsied and normal children: preliminary observations. Dysphagia 4:112-117, 1989 21. Jean A: Control of the central swallowing program by inputs from the peripheral receptors. J Auton Nerv Syst 10:225233, 1984 22. Falempin M, Mei N, Rousseau JP: Vagal mechanoreceptors of the inferior thoracic oesophagus, the lower oesophageal sphincter, and the stomach in the sheep. Pflugers Arch 373: 25-30, 1978
l)ysphagia r Springer-VerlagNew York Inc. 1992
Oral Dysfunction Following Nissen Fundoplication 5
Stephen M. Borowitz, M.D. and Kathleen C. Borowitz, M.S. Department o f Pediatrics, Divisions of Gastroenterology and Speech Pathology, University of Virginia Health Sciences Center, Charlottesville, Virginia, USA
In this case report we describe a child with mild neurologic impairment who developed debilitating gagging and retching, and severe oralmotor dysfunction following Nissen fundoplication and gastrostomy tube placement. All oral intake ceased after the operation. Evaluation for postoperative dumping syndrome was negative, and the child's symptoms failed to improve despite numerous medical and surgical measures. However, immediately following reversal of the Nissen fundoplication, the child's gagging and retching ceased, and his oral-motor function began to improve. This is a previously undescribed complication of Nissen fundoplication, a surgical procedure commonly employed in children with neurologic impairment.
Abstract.
Key words:
Nissen fundoplication -- Developmental delay -- Oral-motor function -- Dysphagia - - Gastroesophageal reflux -- Deglutition -- Deglutition disorders.
In children with neurologic impairment, feeding gastrostomies are often used to allay chronic malnutrition and severe feeding difficulties [1]. Symptomatic gastroesophageal reflux is thought to be quite common among these children [2, 3], and recent studies suggest the incidence ofgastroesophageal re-
flux may increase following gastrostomy placement [4]. As a consequence, there has been a trend to perform "protective" antireflux surgery in developmentally disabled children who need feeding gastrostomies [3, 5]. Nissen fundoplication is the most widely employed antireflux procedure in children, and is an effective means of eliminating gastroesophageal reflux [6-8]. While serious perioperative complications are uncommon with Nissen fundoplication, we have become aware of subtle long-term postoperative morbidity associated with this surgical procedure. Of 46 children who have undergone Nissen fundoplication during the past 2 years, we have observed profound postoperative oral-motor dysfunction in four children with neurologic impairment. The oral dysfunction was initially manifested as severe gagging and retching beginning 1 to 4 weeks postoperatively. This was followed by rapid cessation of oral intake associated with pronounced aversive reactions to presentation of food such as lip pursing, head shaking, turning away and crying, or "fighting." In some cases, even nonnutritive oral stimulation resulted in these aversive behaviors. In one case, oral-motor dysfunction was so severe that the fundoplication was taken down. The reversal of surgery resulted in dramatic improvement. That child's history is summarized below. Case Report
Address offprint requests to: Stephen M. Borowitz, M.D., Department of Pediatrics, Box 386, University of Virginia Health Sciences Center, Charlottesville, VA 22908, USA
L.L. is currently a 36-month old caucasian boy. He weighed 2.8 kg at birth. His mother was a 26-year old primigravida who received good prenatal care, and her full-term pregnancy was uncomplicated. L.L. was delivered by emergency cesarian section due to fetal distress. Apgar scores were 5 at 1 and 7 at 10 rain.
S.M. Borowitz and K.C. Borowitz: Oral Dysfunction Following Fundoplication He developed generalized seizures during the first 24 h of life, characterized by bilateral fisting and bicycling; the seizures were easily controlled with phenobarbital. Physical examination was remarkable only for mild hypotonia. Laboratory evaluation was remarkable only for thrombocytopenia which resolved spontaneously over 5 days. Electroecephalogram during the first week of life demonstrated diffuse slowing without any focal abnormalities. Cranial computerized tomography (CT) revealed mildly dilated third and fourth ventricles and patchy periventricular calcifications. Viral cultures and serologies were negative, as were bacterial cultures. His nursery course was complicated by slow oral feeding, and he was discharged receiving supplemental nocturnal nasogastric feedings providing half of his calories. Oral examination at 1 month of age was remarkable for generalized hypotonia, a retracted mandible, posterior tongue carriage, and oral hypersensitivity. A weak and arrhythmical suck was noted during bottle feeding. At 3 months of age, L.L. was admitted to the hospital because of feeding failure. He continued to take half of his calories orally and received the remainder at night through a nasogastric tube. Little change was evident in his oral function or feeding skills. He had no coughing or gagging during feeding. Generalized hypotonia continued to affect his positioning and ability to achieve and maintain a lip seal on the nipple during bottle drinking. An upper gastrointestinal series demonstrated normal anatomy without nasopharyngeal or gastroesophageal reflux. Esophageal motility appeared normal. Hexible endoscopy was normal and esophageal biopsies did not reveal any inflammatory changes. A surgical gastrostomy and Nissen fundoplication were performed without complication. Gastrostomy feedings were begun on the third postoperative day, and oral feedings were restarted 3 days later. At the time of his hospital discharge on the seventh postoperative day, L.L. was taking 2 ounces of formula every 3 h by mouth (half of his calories), with nighttime supplements through the gastrostomy. A repeat upper gastrointestinal series through the gastrostomy tube prior to discharge showed a normal fundoplication without esophageal obstruction. Gastric emptying was normal. Over the 10 days following his discharge, L.L. began gagging and retching with gastrostomy feedings and his oral intake diminished. Within 1 m o n t h of surgery, all oral intake had ceased. He began to experience episodes of irritability, crying, gagging, and clamminess, often, hut not always associated with gastrostomy feedings (2-3 ounces of Pediasure given over 2 h). A repeat upper gastrointestinal series was performed through the gastrostomy tube 2 months after surgery and showed the expected narrowing of the distal esophagus at the site of the fundoplication. Over the ensuing month, L.L. continued to experience severe gagging and retching episodes no longer clearly associated with feedings. Repeat upper gastrointestinal series 2 weeks later (3.5 months following fundoplication) displayed mild narrowing at the site of the fundoplication without gastroesophageal reflux. Rigid esophagoscopy and esophageal dilatation to 18 m m were performed. Feeding evaluation 2 weeks later revealed refusal of all oral intake with intensified aversive reactions, increased drooling, and marked oral defensiveness even with nonnutritive oral stimulation. Additionally, abnormal oral reflexes were observed including jaw thrust, tongue protrusion, tongue fasciculations, and a very prolonged and prominent gag reflex. Evaluation for dumping syndrome was negative; an oral glucose tolerance test was normal, as were multiple postprandial glucose measurements. The gagging and retching persisted and L.L. was readmitted to the hospital 1 month later. Upper gastrointestinal series dem-
235
onstrated a normal fundoplication without esophageal stenosis. Hexible endoscopy was normal with a patent esophagus and pylorus. Esophageal, gastric, and duodenal biopsy specimens were normal, A pyloroplasty was performed with the hopes of improving gastric emptying however the gagging and retching continued unchanged. Trials of continuous infusion of an elemental formula, pectin, sucralfate, rantidine, and glycopyrrolate afforded no improvement. One year after the initial surgery, the fundoplication was taken down. At the time of surgery, the esophageal wrap appeared intact, and no other abnormalities were noted. L.L. experienced minimal vomiting following the operation, and by the time of his discharge 10 days later he was tolerating a constant infusion of Pediasure at 35 m l / h through his gastrostomy. Gagging and retching completely abated immediately after surgery. It is now 24 months since the fundoplication has been reversed. L.L.'s gagging and retching have completely resolved, and he rarely vomits. His oral-motor function has gradually improved. He has significantly less oral defensiveness, and his jaw and tongue thrusts are less prominent. He now shows some nonnutritive sucking and tolerates tactile stimulation to his face, lips, and gums. His drooling has diminished, and he is currently tolerating bolus gastrostomy feedings without difficulty and eating small amounts of strained foods by spoon. He has begun to spontaneously bring toys, spoons, and bottles to his mouth during play. Since the reversal of the fundoplication, the velocity of weight gain and linear growth have improved significantly.
Discussion
Nissen fundoplication is generally thought to be a safe and effective antireflux procedure. Perioperative mortality is generally less than 1%, and major postoperative complications occur in less than 1 5 o of cases [7, 8]. The most common serious postoperative complications are herniation or breakdown of the esophageal wrap and bowel obstruction [8, 9]. During the perioperative period, fundoplication eliminates gastroesophageal reflux in greater than 90% of patients [6-8]. Few authors have followed patients for prolonged periods following surgery to determine whether there is recurrence of symptomatic reflux. In a retrospective study of 60 adults, Negre found a 10-year failure rate of nearly 20% [10]. Randolph followed 67 infants for between 1 and 10 years following fundoplication and documented a 10% failure rate [ 11 ]. Unfortunately, there have been no prospective long-term studies of children following Nissen fundoplication. Subtle postoperative complications may occur quite commonly following Nissen fundoplication. In their retrospective review of 25 children evaluated 4 to 9 years postoperatively, Harnsberger et al. found 36% had significant symptoms attributable to surgery; 32% of the children had difficulties with abdominal distention and discomfort (gas-bloat), 32% had major changes in their eating habits after the surgery, and 25% suffered intermittent solid dys-
236
S.M. Borowitzand K.C. Borowitz: Oral Dysfunction FollowingFundoplication
phagia [12]. O f the 226 adults Negre assessed between 3 and 12 years following the operation, 44% had major changes in their eating habits, 38% suffered from excessive flatus, abdominal distension and discomfort (gas-bloat), and 19% were unable to burp [ 13]. The patient we describe above suffered severe retching and gagging postoperatively. While neither Harnsberger et al. nor Negre mention these distressing symptoms, Stringel et al. observed retching in 13% of 90 children with neurologic impairment following fundoplication [9], and Jolley et al. noted postoperative retching in 18% of the 66 children he evaluated [ 14]. In six ofJolley et al.'s twelve patients the retching was attributed to postoperative dumping syndrome. Dumping syndrome is a complex of postprandial symptoms which may include abdominal distention, diarrhea, irritability, refusal to feed, weakness, and pallor. Dumping syndrome may occur frequently following Nissen fundoplication and generally appears during the first several weeks following surgery [15]. While the pathophysiology of the syndrome is not completely understood, many of the symptoms may be attributable to postprandial hypoglycemia. An oral glucose tolerance test appears to be a sensitive diagnostic measure and demonstrates early hyperglycemia followed by reactive hypoglycemia [15]. While many of L.L.'s symptoms were suggestive of,dumping syndrome, he did not experience any diarrhea, and oral glucose tolerance tests were normal on two occasions. Moreover, he failed to respond to those therapeutic measures often helpful in dumping syndrome such as hyposmolar feedings, constant infusion feedings, and pectin. In six of the twelve patients Jolley et al. described with postoperative retching, dumping syndrome was not present and symptoms were attributed to decreased gastric emptying [14]. Some authors have suggested that performing a gastric outlet procedure to facilitate gastric emptying may lessen the incidence of postoperative retching [16]. While pyloroplasty failed to improve our patient's symptoms, reversal of the fundoplication immediately eliminated his gagging and retching. In addition to L.L., we have observed severe postoperative retching in three other children with neurologic impairment. In all four of these children, retching was associated with rapid decline of oral intake and profound deterioration of oral-motor function. While retching resolved spontaneously over 6 months in two of these four children, oralmotor dysfunction has persisted. In the ease presented above, it was only when the fundoplication
was reversed that oral function began to improve. In none of the published follow-up studies is any mention made of oral function either before or following fundoplication, nor is there any discussion of feeding skills or the method of feeding employed. Among families with children with neurologic impairment, feeding issues are often emotionally charged [17]. Many of these families find feeding their child a gratifying experience; while feeding gastrostomy may be employed to ease the difficulties associated with feeding some of these children, it should not necessarily be used to eliminate all oral intake. While we have observed a variety of complications following Nissen fundoplication in normal children, we have not seen in them the dramatic changes in oral-motor function that we have observed in children with neurologic impairment. Many children with neurologic impairment have difficulty with oral motor control. They may exhibit delayed swallowing reflexes, inefficient swallows, and abnormal swallow frequency which can lead to excessive drooling [18, 19]. They may also exhibit poor coordination between swallowing and ventilation partly explaining their predilection for silent aspiration and respiratory tract infections [ 19, 20]. It is unclear why oral function deteriorates following fundoplication in children with neurologic impairment. Swallowing is an extremely complex motor sequence which depends upon activity of medullary interneurons in the swallowing center. While swallowing is largely reflexive, there is a large voluntary component, and swallowing neurons receive strong inhibitory input from vagal afferent fibers [21]. Some of these afferent fibers may arise from the numerous mechanoreceptors in the distal esophagus, gastroesophageal junction, and gastric fundus [22]. Fundoplication may cause chronic mechanoreceptor activation which inhibits spontaneous activity in the central swallowing center. This may explain some of the changes in eating habits following Nissen fundoplication. Normal children may overcome these inhibitory inputs through the conscious initiation of swallows. In contrast, children with neurologic impairment who already have disordered swallowing and poor oral motor control may be unable to overcome the inhibitory inputs. In our case, reversal of the fundoplication may have alleviated swallow inhibition and facilitated restoration of oral function. The restoration of oral function following reversal of the fundoplication suggests that the problems were not caused by disruption of vagal fibers, but rather by mechanical interference.
S.M. Borowitz and K.C. Borowitz: Oral Dysfunction Following Fundoplication With this report, we relate a previously undescribed long-term complication of Nissen fundoplication. In future studies e x a m i n i n g the m e d i c a l and surgical m a n a g e m e n t o f g a s t r o e s o p h a g e a l reflux in n e u r o l o g i c a l l y i m p a i r e d c h i l d r e n , it w o u l d s e e m w a r r a n t e d to p r o s p e c t i v e l y e v a l u a t e o r a l f u n c t i o n a n d f e e d i n g skills.
References 1. Rempel GR, Colwell SO, Nelson RP: Growth in children with cerebral palsy fed via gastrostomy. Pediatrics 82:857862, 1988 2. Sondheimer JM, Morris BA: Gastroesophageal reflux among severely retarded children. J Pediatr 94:710-714, 1979 3. Wesley JR, Coran AG, Sarahan TM, Klein MD, White SF: The need for evaluation of gastroesophageal reflux in braindamaged children referred for feeding gastrostomy. J Pediatr Surg 16:866-871, 1981 4. Mollitt DL, Golladay ES, Seibert J J: Symptomatic gastroesophageal reflux following gastrostomy in neurologically impaired patients. Pediatrics 75:1124-1126, 1985 5. Jolley SJ, Smith El, Tunell WP: Protective antireflux operation with feeding gastrostomy; experience with children. Ann Surg 201:736-740, 1985 6. Fonkalsrud EW, Foglia RP, Ament ME, Berquist W, Vargas F: Operative treatment for the gastroesophageal reflux syndrome in children. J Pediatr Surg 24:525-529, 1989 7. Vane DW, Harmel RP, King DR, Boles ET: The effectiveness ofNissen fundoplication in neurologically impaired children with gastroesophageal reflux. Surgery 98:662-666, 1985 8. Dedinsky GK, Vane DW, Black CT, Turner MK, West KW, Grosfield FL: Complications and reoperation after Nissen fundoplication in childhood. Am J Surg 153:177-183, 1987 9. Stringel G, Delgado M, Guertin L, Cook JD, Maravilla A, Worthen H: Gastrostomy and Nissen fundoplication in neurologically impaired children. J Pediatr Surg 24:1044-1048, 1989 10. Negre JB, Markkula HT, Keyrilaninen O, Matikainen M:
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