Rumination syndrome is defined by the Rome III criteria as repeated painless regurgitation and rechewing or expulsion of food within minutes of starting a meal (1). It is a phenomenon considered normal in ruminant animals, but its occurrence in humans is always pathologic. This syndrome had been reported in the past as being prevalent among infants and adults with developmental disabilities (2,3), but more recently its recognition has increased among children, adolescents, and adults with normal intelligence (4,5). The pathophysiology of rumination remains incompletely understood. The aim of this article is to describe 2 cases of adolescent girls with rumination syndrome in which we identified a new pathophysiologic mechanism similar to what has been described in supragastric belching (6). We termed it “supragastric vomiting” and to our knowledge it has not been described before.
A 14-year-old girl, previously healthy, experienced an episode of viral gastroenteritis with profuse vomiting for several days followed by several months of effortless vomiting of both liquids and solids immediately after or during meals. There was no pain or nausea and she had a 7-kg weight loss in 6 months. There were no nocturnal symptoms. She underwent an extensive workup, which included routine serum, urine, stool studies, upper gastrointestinal series, esophagogastroduodenoscopy, abdominal ultrasound, computed tomographic scan, and magnetic resonance imaging of the head and hepatobiliary iminodiacetic acid scan, all providing normal results. She was treated with erythromycin, metoclopramide, cisapride, domperidone, ondansetron, and omeprazole without significant improvement. Subsequently, she developed an inability to swallow solids and liquids and gastrojejunal feedings were initiated.
A nuclear medicine esophageal transit study showed that when the radioactive water was swallowed there was bolus movement through the esophagus distally, but only a small amount entered the stomach. Within 2 seconds there was reverse transit back to the upper esophagus, followed by vomiting. An antroduodenal manometry demonstrated normal motility. An esophageal manometry showed normal esophageal motility when saliva was swallowed and a series of deep inspirations followed by simultaneous pressure waves associated with gagging and vomiting when water was offered (Fig. 1A).
An intensive multidisciplinary approach was instituted with participation of members from gastroenterology and psychiatry. There was a slow, steady improvement of symptoms over 12 months with the ability to swallow food followed subsequently by the ability to avoid regurgitation. She has resumed all of her normal activities and is able to tolerate full feedings by mouth.
A 12-year-old girl was in her usual state of health until she had an episode of several days of emesis believed to be secondary to a viral gastroenteritis. For the following 6 months she complained of a variety of somatic symptoms including vomiting during and immediately after each meal, abdominal pain, weight loss, and fatigue. There was no nausea and no vomiting at night. She was hospitalized several times and underwent extensive investigations including magnetic resonance imaging and magnetic resonance spectroscopy of the brain; 2 esophagogastroduodenoscopies, which showed normal mucosa; an upper gastrointestinal series, which demonstrated no anatomical defect; and a nuclear medicine gastric emptying study of a solid meal, which showed several episodes of reflux and a mildly delayed gastric emptying. Several medications were tried including esomeprazole, metoclopramide, domperidone, erythromycin, tegaserod, hyoscyamine, amitriptyline, ondansetron, sucralfate, and citalopram without improvement in her symptoms. Because of persistent vomiting, a gastrojejunal feeding tube was placed. Her symptoms changed from chronic vomiting to difficulty in swallowing solid food.
At the time of her visit to our motility center, she was able to swallow water and saliva without difficulty; however, when eating solids she immediately brought the food up to the point that it was unclear whether she even swallowed the food. An esophagogram revealed that the barium reached the stomach without difficulties when given as such, but when a cracker was coated with barium, the material was swallowed, and after reaching the lower part of the esophagus, part of it was regurgitated. It was then followed by normal esophageal peristalsis, which cleared the residual barium. An antro-duodenal manometry showed no abnormalities suggestive of an intrinsic motility disorder. An esophageal manometry demonstrated normal peristaltic sequences when she was swallowing her own saliva or a small amount of water. When given pudding the patient gulped air followed by regurgitation of some of the swallowed food. This phenomenon was associated with an increase in pressure throughout the esophagus followed by a peristaltic sequence (Fig. 1B). The findings of the manometry study and the esophagogram were discussed with the child and her parents and allowed us to point out the behavioral nature of the problem. Behavioral changes were instituted and the patient began to be able to swallow food again. After another apparent “viral gastroenteritis,” she developed more classic rumination behavior, underwent a 3-week hospitalization, and is currently making great progress while receiving further behavioral treatment consisting of diaphragmatic breathing and progressive, slow increase in the size and consistency of food ingested.
The etiology of rumination syndrome remains unknown, but the history of viral gastroenteritis at the onset of symptoms in both our patients supports the theory that a stressful event such as mucosal disease or emotional arousal may trigger a learning behavior that leads to rumination (4,7–9). Recent reports suggest that rumination can be diagnosed on a clinical basis alone, yet it is not uncommon for patients with this condition to endure extensive, invasive, and often unnecessary workup before this entity is recognized (10). Lack of awareness of this disorder may constitute the main reason to perform such extensive testing. The recent publication of the pediatric Rome III criteria for functional gastrointestinal disorder with the inclusion of adolescent rumination among the entities discussed should facilitate such diagnosis in the future (1). In our tertiary referral center, we now diagnose approximately 10 to 15 adolescents with rumination syndrome every year. The diagnosis of rumination may be challenging because symptoms may overlap with those of gastroesophageal reflux, gastroparesis, chronic intestinal pseudoobstruction, and eating disorders (bulimia) (11). Antroduodenal manometry was performed in both our patients and failed to prove the presence of the characteristic “R” waves described by Amarnath et al (7), which were also reported to be present in only 40% of the pediatric patients in a study done by Chial et al (10). Interestingly, neither of our patients vomited during the antroduodenal manometry study. We have observed that under the pressure of being in a laboratory setting with constant attention being paid to their symptoms, adolescents with rumination are often able to go through a meal without symptoms. Making the patients aware of their behavior when they contract their abdominal muscles to facilitate regurgitation or when they swallow air before ingesting the bolus of food in the case of supragastric vomiting is an important factor in initiating a successful treatment plan.
In the cases described, the patients' behavior changed from effortless emesis to “inability to swallow.” It is unclear whether this behavior is part of a spectrum of disorders related to eating or a different functional disorder. The fact that the children seemed to start with a more classic rumination behavior and evolved into the inability to swallow (and the second child eventually reverted to ruminating again) suggests that the psychopathology underlying these entities may be the same. The esophagogram and the nuclear medicine transit study indicated that some of the ingested material never reached the stomach, an unusual finding even for the ruminators. The esophageal manometry was a revealing test demonstrating that before swallowing a bolus of food the patients took a deep breath, trapped the air in the esophagus, and then regurgitated the material from the esophagus. This is similar to a phenomenon previously reported by Bredenoord et al (6) in patients with aerophagia, which the authors termed “supragastric belching.” Because our patients regurgitated food instead of air as in aerophagia, we have elected to call it “supragastric vomiting,” a finding never reported before in pediatrics or in patients with rumination syndrome.
Despite the parents and referring physicians' concern that either an obstructive lesion or a motility problem was responsible for the symptoms, the behavioral nature of this disorder became clear during the motility tests performed. In both patients, there was a decrease in the number of episodes when attention was driven or taken away from the act of eating, as described before in patients with other functional disorders like aerophagia or rumination (12). Pointing out to the patients and to the parents how saliva was easily swallowed but even 2 mL of water was causing symptoms was particularly enlightening. The lack of improvement with several antisecretory and prokinetic medications also made this association more evident and decreased the likelihood that a true motility disorder was present (a concern that had brought both patients to our center for manometry testing). The immediacy of vomiting related to the ingestion of food did not fit well the presentation of gastroparesis. Techniques like combined multichannel intraluminal impedance and manometry, which have been used to diagnose both supragastric belching and rumination in adults, could help better explain and further characterize this entity in the pediatric population (13,14) and may indicate when the behavior changes from classic rumination to inability to swallow.
Rumination syndrome is an entity increasingly diagnosed in adolescents with normal intelligence. The phenotype of rumination may vary from the classic regurgitation of material ingested a few minutes before to the instantaneous vomiting of food and inability to swallow most solids and liquids except saliva (the latter behavior being described in this report as supragastric vomiting). Manometry studies may help to clarify the pathophysiology of this condition and alert the medical provider to the presence of supragastric vomiting. Behavioral interventions in such cases are associated with a favorable outcome.
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