Journal of Pediatric Gastroenterology and Nutrition 31:204207 August 2000 Lippincott Williams & Wilkins, Inc.

, Philadelphia

Case Report

Aerophagia as Cause of Gaseous Abdominal Distention in a Toddler

Vera LoeningBaucke
Department of Pediatrics, University of Iowa, Iowa City, Iowa, U.S.A.

Aerophagia involves excessive air swallowing causing progressive abdominal distension. The symptoms in children are a nondistended abdomen in the morning, progressive abdominal distension during the day, visible, often audible, air swallowing, and excessive flatus. Resolution of the abdominal distension occurs during the night by absorption of gas and by flatulence. Although aerophagia in childhood is rarely discussed in the literature, it is a common disorder seen by adult gastroenterologists (15). We present a 32-month-old toddler with a long history of functional constipation in whom gaseous abdominal distension suddenly developed. Increase in his daily laxative dosage did not improve the abdominal distension. Mechanical intestinal obstruction, chronic intestinal pseudo-obstruction, bacterial overgrowth, Hirschsprungs disease, and malabsorption were considered and ruled out. The extensive gaseous abdominal distension was due to aerophagia and withholding of stool and flatus. CASE REPORT A 32-month-old boy was referred to the Encopresis Clinic at the University of Iowa because of abdominal distension during the past 4 weeks. The parents reported that his abdomen would become more and more distended as the day progressed. During the night, he had extensive episodes of flatulence. On awakening, his room was foul-smelling and his abdomen was flat. He had been treated with milk of magnesia for constipation for the past 18 months. He had been withholding stool for the past 7 months. He sat on the floor 12 to 20 times during the day and withheld stool and, in addition, flatus. The parents reported that because of the abdominal distension, his daily milk of magnesia dosage was increased from 25 mL to 40 mL with no improvement. He had a
Received February 3, 2000; revised May 25, 2000; accepted May 30, 2000. Address correspondence and reprint requests to Dr. Vera LoeningBaucke, University of Iowa Hospitals and Clinics, Dept. of Pediatrics, JCP 2555, 200 Hawkins Drive, Iowa City, IA, 52242, U.S.A.

soft bowel movement in his diaper every 3 days and eight small soiling episodes per day. He had not had a formed stool in the past 6 months. He vomited occasionally. A dietary history revealed that he was a picky eater, liked milk, had poor fruit intake, and rarely ate meat. He was the product of an uncomplicated pregnancy and delivery. Birth weight was 3710 g. At 6 months of age he had several days of constipation. He then had normal daily bowel movements until reaching 12 months of age, when diarrhea developed with six to eight bowel movements daily and a severe diaper rash. The stools did not contain blood or mucus. Laboratory evaluation at that time included several stool cultures and stool examinations for ova, parasites, and Giardia lamblia, fecal white cells, Clostridium difficile, Clinitest (Bayer, West Haven, CT, U.S.A.) pH, and fat; complete blood count; electrolytes; serum immunoglobulin (Ig)A; and gliadin and endomysial IgA and IgG antibodies. All laboratory results were normal. At 14 months of age, constipation was diagnosed and he had had problems with constipation since. He was breast-fed until 14 months of age. He began consuming cheese and yogurt 9 months of age and cows milk at 12 months of age. Motor development was normal, and speech development was delayed. A physical examination revealed an anxious, uncooperative toddler. He could speak words but produced a constant whining noise. His weight was 14 kg (50th percentile), his height was 95 cm (68th percentile), and his head circumference was 50 cm (64th percentile). No skin rash was present. The physical examination was remarkable for findings during the abdominal and rectal examinations. The abdomen was severely distended, nontender, hypertympanitic but soft, and bowel sounds were active. No hepatomegaly or splenomegaly was noted. The rectal examination revealed no perineal erythema, a normal anus, and a hugely dilated rectal ampulla filled with gas; no stool was present. A neurologic examination was normal, and muscle strength and tone were appropriate for age. The abdominal radiographs were reviewed. The supine abdominal radiograph from 2 weeks prior (Fig. 1) showed gastric distension with air and prominent air in 204

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felt throughout the abdomen. No gaseous distension was appreciated. The rectal examination revealed a large soft stool in the rectal ampulla. A supine abdominal radiograph revealed only gas within the large bowel and a significant quantity of stool, predominantly in the ascending and sigmoid colon. He received two phosphate enemas (135 mL each) with large stool output. Afterward the abdomen was flat, and the distended rectal ampulla was empty of stool. Senna was discontinued and a 10-mg bisacodyl suppository was administered every other day resulting in a his passing stool every other day into the toilet. The abdomen remained nondistended. DISCUSSION
FIG 1. Supine abdominal radiograph shows gastric distension with air and prominent air in the small and large bowel. Some stool was present in the ascending and transverse colon. There were no airfluid levels, and no obstruction was seen.

the small and large bowel. Some stool was present in the ascending and transverse colon. There were no airfluid levels, and no obstruction was seen. The supine abdominal radiograph from 1 week prior, after a phosphate enema and an increased daily dose of milk of magnesia to 40 mL, showed increased amounts of gas in the gastrointestinal tract, moderate dilatation of the right colon, and gas in the transverse and sigmoid colon. Liquid stool was present in the right colon and rectum. The amount of stool was less in comparison to that seen in the radiograph obtained 1 week prior. The parents said they had not seen swallowing movements when he was not eating or drinking and had not noticed belching. On further questioning, they reported that he had been making unusual squeaky noises for the past few weeks, which suggested air swallowing. Anorectal manometry was performed and showed the rectosphincteric reflex to be present. Treatment and Follow-up The treatment of aerophagia consisted of reassurance and explanation to the parents and child. He stopped making the squeaky noise soon after he was evaluated by the author, and the daily abdominal distension stopped. The treatment of fecal retention consisted of discontinuing milk of magnesia and administering senna daily. Senna was increased to 15 mL (3 teaspoonfuls) daily with breakfast and was administered for 1 month, but no regular stool output occurred. Elimination of all milk products for a 2-week period did not change the stool pattern. Because of his persistent soiling, his mother discontinued the senna. When he was evaluated 3 weeks later, the abdomen appeared distended, tender, and firm, and stool could be

Gas is normally present throughout the lumen of the gut from the mouth to the anus. This gas is derived from four major sources: swallowed air, interaction of gastric acid with alkaline secretions of food, diffusion of gas from the blood stream into the gut lumen, and bacterial fermentation (6). It is estimated that 70% of the gastrointestinal gas is swallowed, 20% is caused by diffusion of gases from the blood, and 7% to 10% is the result of bacterial decomposition (1). Under normal circumstances swallowed air is the predominant source of gastric gas, with a minor contribution from gas refluxing through the pylorus (7). Air swallowing is universal and involuntary. It occurs with eating and drinking, salivation, and chewing gum. During infancy, excessive air swallowing may result from inappropriate feeding practices or crying, nursing from an empty bottle, or sucking on a pacifier. In older children, large amounts of air can be swallowed while drinking through a straw or chewing gum. The volume of air swallowed when drinking a glass of milk is two to three times the volume of the milk drunk, whereas the maximal amount of gas that can be produced in the colon by bacterial action after eating a large helping of starchy food is infinitely less (8). Very few articles have been written about aerophagia (eructio nervosa) in childhood (15). Recently, RasquinWeber et al. (9) and Hyman et al. (10) published the Rome II criteria for aerophagia. The Rome II diagnostic criteria are at least 12 weeks, which need not be consecutive, in the preceding 12 months of two or more of the following signs and symptoms: air swallowing, abdominal distension due to intraluminal air, and repetitive belching and/or increased flatus. Aerophagia is a rarely observed behavior among healthy children and adults (0,14%) but involves 8.8% of the institutionalized mentally retarded population (11). In many of the reported pediatric patients, the diagnosis was missed initially (3). Usually, parents do not volunteer the excessive air swallowing, because they do not recognize it as such. Parents may deny frequent swallowing in the child because they may not realize that it is happening, but when asked about gulping sounds or other frequent noises that they
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V. LOENINGBAUCKE fully with osmotic laxatives, in particular with lactulose. If withholding is present, then abdominal bloating is pronounced. Gastric distension is not observed. The laxative should be discontinued, and a stimulant laxative, such as senna, should be substituted. Interference with intestinal transport results from mechanical bowel obstruction. It also results from luminal patency when peristalsis is abnormal or absent due to neuromuscular disease, ischemia, and intrinsic or extrinsic inflammation and in postoperative states. Gastroparesis, megaduodenum, and/or megacolon have been described in children and adults with chronic intestinal pseudo-obstruction. The most common symptoms in children with chronic intestinal pseudo-obstruction are nausea, vomiting, abdominal distension, constipation, and failure to thrive (18). The diagnosis is suspected on the basis of history, physical examination, and basic radiologic studies. The plain abdominal radiograph shows generalized small bowel and/or colonic distension. A working group recently suggested that this term be used only in patients with the most severe forms of motility disorders, in whom any segment of the gastrointestinal tract may be dilated or is intermittently dilated (19). Such severe occurrences are rare, whereas less severe forms of disordered motility are much more frequently observed. Patients with altered gastrointestinal motility may have bacterial overgrowth, resulting in diarrhea, bloating, and cramps. In them, breath hydrogen testing is useful. Many investigators now consider Hirschsprungs disease as part of pseudo-obstruction. Hirschsprungs disease is characterized by absence of ganglion cells in the distal bowel, beginning at the internal sphincter and extending proximally for varying distances. The ganglionated bowel is dilated above the aganglionic segment and contains large amounts of stool and gas. The aganglionic segment is constricted or normal but never dilated. Therefore, this diagnosis was unlikely in our patient with dilatation of the colon and rectum down to the anal level. The presence of a rectosphincteric reflex during anorectal manometric testing excluded Hirschsprungs disease (20). Our toddler had two functional disorders: extensive gaseous abdominal distension as a complication of aerophagia and withholding of stool and flatus because of functional fecal retention. Recognition of excessive air swallowing prevented unnecessary studies and treatment. The air swallowing stopped spontaneously after several weeks. In addition, he had functional fecal retention, which did not respond to oral laxatives and oral stimulants, but responded to bisacodyl suppositories. It is hoped that with time, the rectal stimulant can be replaced by a mild laxative. REFERENCES
1. Stone RT, Morgan MC. Aerophagia in children. Am Fam Physician 1971;3:945. 2. Rosenbach Y, Zahavi I, Nitzan M, et al. Pathologic childhood aerophagy: an under-diagnosed entity. Eur J Pediatr 1988;147:4223.

recognize, they may report sounds that are suggestive of air swallowing. In our patient, the diagnosis of aerophagia was made because the abdominal radiographs showed an airdistended stomach and increased gas in the small and large bowel and because of the squeaky noises that suggested air swallowing. In addition, he had a nondistended abdomen in the morning, progressive abdominal distention during the day, and nocturnal flatulence. Our patient did not have excessive flatulence during the day, which may have been absent because of his other functional disease, which was fecal retention with retentive posturing. Functional fecal retention has been defined by the Rome II criteria as a history of at least 12 weeks of passage of large-diameter stools at intervals of less than two times per week and retentive posturingthat is, avoiding defecation by purposely contracting the pelvic floor. As the pelvic muscles fatigue, the child uses the gluteal muscles, squeezing the buttocks together (9,10). Aerophagia represents a functional gastrointestinal disorder rather than disease and is usually self-limited, lasting weeks to months, as in our patient. A careful history and a minimal number of diagnostic studies should differentiate it from organic disease, such as malabsorption or intestinal obstruction. The correct diagnosis helps in alleviating anxiety and prevents unnecessary testing, treatments, and hospital admission. Treatment is usually by reassurance, as it was in our patient. Occasionally, psychological counseling and behavior modification are necessary if symptoms persist, particularly in profoundly mentally retarded children (12,13). Gastric decompression by nasogastric tube has been necessary in a few patients in whom respiration was hindered by the extreme elevation of the diaphragm (3). Rarely has the massive distension of the bowel lead to ileus, volvulus, and necrosis (14,15). Other causes of increased amounts of gas in the gastrointestinal tract were considered, such as drinking carbonated beverages or chewing gum. The family denied that the child consumed either. Normal adults consuming a standard diet excrete variable amounts of gas, rarely exceeding 100 mL per hour (16), and have 20 episodes of flatulence per day (17). Malabsorption of lactose or other carbohydrates and bacterial fermentation of ingested carbohydrates and vegetables containing nonabsorbable carbohydrates result in increased gas production. However, most of the gas present in the intestine is absorbed through the mucosa. Milk and all milk products were withdrawn for 14 days in our patient without a change in his condition. Extreme flatus with a greatly distended abdomen is seen in children with mucosal disease such as celiac disease. Clinical manifestations of malabsorption are diarrhea and steatorrhea. A previous work-up for celiac disease ruled out this diagnosis. Increased gas in the bowel and flatulence are often seen in constipated children who are treated unsuccessJ Pediatr Gastroenterol Nutr, Vol. 31, No. 2, August 2000

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3. Gauderer MW, Halpin TC Jr, Izant RJ Jr. Pathologic childhood aerophagia: a recognizable clinical entity. J Pediatr Surg 1981;16:3015. 4. Holburn CS. Aerophagia: an uncommon form of self-injury. Am J Ment Def 1986;91:2013. 5. Lecine T, Michaud L, Gottrand F, et al. Les enfants avaleurs dair. Arch Pediatr 1998;5:12248. 6. Sferra TJ, Heitlinger LA. Gastrointestinal gas formation and infantile colic. Pediatr Clin North Am 1996;43:489510. 7. Lasser RB, Bond JH, Levitt MD. The role of intestinal gas in functional abdominal pain. N Engl J Med 1976;293:5246. 8. Hunt T. Aerophagy and flatulence. Practitioner 1970;205:7878. 9. Rasquin-Weber A, Hyman PE, Cucchiara S, et al. Childhood functional gastrointestinal disorders. Gut 1999;45(suppl 2):11608. 10. Hyman PE, Rasquin-Weber A, Fleisher DR, et al. Childhood functional gastrointestinal disorders. In: Drossman DA, Corazziari E, Talley NJ, et al. eds. The Functional Gastrointestinal Disorders. 2nd ed. Lawrence, KS: Allen Press, Inc.; 2000:53375. 11. Lekkas CN, Lentino W. Symptom-producing interposition of the colonic: Clinical syndrome in mentally deficient adults. JAMA 1978;240:74750. 12. Barrett RP, McGonigle JJ, Ackles PK, et al. Behavioral treatment of chronic aerophagia. Am J Ment Def 1987;91:6205.

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13. Holburn CS, Dougher MJ. Behavioral attempts to eliminate airswallowing in two profoundly mentally retarded clients. Am J Ment Def 1985;89:52436. 14. van der Kolk MB, Bender MH, Goris RJ. Acute abdomen in mentally retarded patients: role of aerophagia: report of nine cases. Eur J Surg 1999;165:50711. 15. Trillis F Jr, Gauderer MWL, Ponsky JL, et al. Transverse colon volvulus in a child with pathologic aerophagia. J Pediatr Surg 1986;21:9668. 16. Steggerda FR. Gastrointestinal gas following food consumption. Ann NY Acad Sci 1968;150:5766. 17. Levitt MD, Furne J, Aeolus MR, et al. Evaluation of an extremely flatulent patient: case report and proposed diagnostic and therapeutic approach. Am J Gastroenterol 1998;93:227681. 18. Di Lorenzo C. Pseudo-obstruction: current approaches. Gastroenterology 1999;116:9807. 19. Rudolph CD, Hyman PE, Altschuler SM, et al. Diagnosis and treatment of chronic intestinal pseudo-obstruction in children: report of a consensus workshop. J Pediatr Gastroenterol Nutr 1997;24:10212. 20. Loening-Baucke V, Pringle KC, Ekwo EE. Anorectal manometry for the exclusion of Hirschsprungs disease in neonates. J Pediatr Gastroenterol Nutr 1985;4:596603.

Clinical Quiz, continued from page 203. Answer: The abdominal computed tomography scan demonstrated soft tissue thickening along the left anterolateral abdominal wall, from the left rib margin to below the iliac crest. There were collections of fluid and air bubbles in the fascial planes around the external oblique muscles (Fig. 2). The findings were suggestive of necrotizing fasciitis. The patient required immediate surgery with removal of the G-tube and debridement of the necrotic tissue. She also required further de bridements. Additional therapy included continued triple antibiotic therapy, full parenteral nutritional support, treatment with hyperbaric oxygen, and transfusions of packed red blood cells (PRBC) and immunoglobulin. The G-tube was surgically reinserted at the original site 6 days after the initial debridement. The patient slowly improved and was discharged after 2 weeks with the abdominal wall wounds healing well. The patient continued to use the G-tube for feeding. She has subsequently had a successful bone marrow transplantation for her Blackfan-Diamond anemia. Comment: Although percutaneous endoscopic G-tube (PEG) placement is a safe procedure, it is not without complications. Both minor and major complications can occur. Minor complications include stomal leakage, peristomal infection, transient pneumoperitoneum, transient emphysema, mild upper gastrointestinal tract bleeding, catheter obstruction and dislodgment of the tube (1,2). Major complications include gastrocolic and enterocutaneous fistulae, peritonitis, necrotizing faciitis, seeding cancer metastasis, aspiration pneumonia, and serious systemic wound infections (1,2). Minor complications have been reported in up to 50% of adult cases, and FIG. 2. major complications in 3% to 9% (1,2). Studies of PEG in children have shown similar types of complications, but no fasciitis (35). The most commonly reported complications are infections at the G-tube site (25). These range from superficial cellulitis to abscess formation and lifethreatening necrotizing fasciitis (NF) (69). Occurrence of NF after PEG placement has been sparsely reported in adults, and there have been no reported cases in children. Mortality associated with NF is high; two of the three recently reported adult cases resulted in death (68). NF is a rapidly progressive bacterial infection characterized by necrosis of the subcutaneous tissue and fascia, and it can be accompanied by severe systemic toxicity. NF can follow surgical or traumatic wounds, and it generally occurs in the extremities, perineum, and abdominal wall. There is an increased frequency and mortality in the compromised host. Risk factors for NF include malnutrition, diabetes, obesity, alcoholism, varicella infection, and corticosteroid-related immunosuppression. A high index of suspicion allows early diagnosis and treatment. Initial symptoms are often nonspecific and may include fever, malaise, edema, induration, and severe pain at the involved site. Subcutaneous crepitus may be felt. Radiographs may reveal gas or fluid in the fascial planes, which can be confirmed by CT. Absence of these findings, however, does not negate a diagnosis of NF, and suspicion should remain in the case of rapidly progressing cellulitis and a deteriorating clinical status. The causative organisms of NF are variable. Staphylococcus aureus, enterobacteriae, and hemolytic streptococcal species are frequently present. A combination of facultative aerobic and anaerobic bacteria may be isolated, and synergistic action of such a combination may result in a fulminant course. The diagnosis is confirmed by operative exploration of the wound, which reveals edema and necrosis. Treatment consists of aggressive surgical debridement and wide spectrum antibiotics. Hyperbaric oxygen has been reported to be useful in the treatment of group A streptococcal necrotizing infections, but its use as standard therapy for NF is controversial. Physician awareness and early diagnosis with prompt surgical and medical intervention remain the greatest predictive factors for a favorable outcome.

REFERENCES
1. Steffes C, Weaver DW, Bouwman DL. Percutaneous endoscopic gastrostomy. Am Surg 1989;55:2736. 2. Ganga UR, Ryan JJ, Schafer LW. Indications, complications, and long-term results of percutaneous endoscopic gastrostomy: a retrospective study. South Dakota J Med 1994;47:14952. 3. Gauderer MWL. Percutaneous endoscopic gastrostomy: A 10-year experience with 200 children. J Pediatr Surg 1991;26:28892. 4. Marin OE, Glassman MS, Schoen BT, Caplan DB. Safety and efficacy of percutaneous endoscopic gastrostomy in children. Am J Gastroenterol 1994;89:35761. 5. Behrens R, Lang T, Muschweck H, Richter T, Hofbeck M. Percutaneous endoscopic gastrostomy in children and adolescents. J Pediatr Gastroenterol Nutr 1997;25:48791. 6. Haas DW, Dharmaraja P, Morrison JG, Potts JR. Necrotizing fasciitis following percutaneous endoscopic gastrostomy. Gastrointest Endo 1988;34:4878. 7. Grief JM, Ragland JJ, Ochsner MG, Riding R. Fatal necrotizing fasciitis complicating percutaneous endoscopic gastrostomy. Gastrointest Endo 1986;32:2923. 8. Cave DR, Robinson WR, Brotschi EA. Necrotizing faciitis following percutaneous endoscopic gastrostomy. Gastrointest Endo 1986;32:2946. 9. Person JL, Brower RA. Necrotizing faciitis/myositis following percutaneous endoscopic gastrostomy. Gastrointest Endo 1986;32:309.

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