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A Twenty-one–Year–Old College Student With Postprandial Regurgitation and Weight Loss

2006; Elsevier BV; Volume: 4; Issue: 11 Linguagem: Inglês

10.1016/j.cgh.2006.09.008

1542-7714

Heather J. Chial, Michael Camilleri,

Infant Health and Development

A 21-year-old college student is referred for evaluation of “reflux” symptoms refractory to high-dose proton pump inhibitor medication twice daily for the past 9 months. Her symptoms occur daily after nearly all meals. She describes repeated regurgitation of food into the back of her throat beginning no longer than 20 minutes after she starts eating. When she is eating alone, she will rush to the restroom to spit the food out. When she is eating with others, she eats more slowly and has trained herself to swallow the regurgitated food back down. When she repeatedly swallows the regurgitated food, the latter becomes bitter and sour within approximately 20 minutes, and she has to spit it out rather than re-swallow it. For this reason, she will rarely eat an entire meal when she is in the company of others. She is 5 feet 4 inches tall and currently weighs 100 pounds. She has lost 30 pounds during the past 9 months since the above symptoms began. There is no history of an eating disorder. She perceives that she is too thin and is concerned about her weight loss. She has also developed irregular menstrual periods during the past 6 months. She is not sexually active. Ambulatory esophageal pH testing off of proton pump inhibitor medication showed a pH of less than four 8% of the time in the upright position (normal, less than 6.3%) and no nocturnal reflux (normal, less than 1.2%). A gastric emptying test with a radiolabeled egg meal was conducted during a period of 90 minutes and showed a half-time of 190 minutes (normal for the referral laboratory where the test was performed was 90 minutes). She wants to be “cured” of these symptoms as soon as possible so that she can start medical school in the fall. What is the underlying diagnosis, and how should she be treated? The referring physician wonders whether she is a candidate for Nissen fundoplication or gastric electrical stimulation. The clinical presentation is highly suggestive of rumination syndrome. This is characterized by the effortless regurgitation into the mouth of recently ingested food followed by re-chewing and re-swallowing or expulsion. The syndrome is most commonly seen in infants and the developmentally disabled. However, rumination syndrome does occur in children, adolescents, and adults with normal intelligence. Individuals with rumination syndrome are often misdiagnosed or undergo extensive, costly, and invasive testing before diagnosis. Insufficient awareness of the clinical features of rumination syndrome contributes to the underdiagnosis of this important medical condition. Rumination syndrome is frequently confused with bulimia nervosa, gastroesophageal reflux disease, and upper gastrointestinal motility disorders including gastroparesis and chronic intestinal pseudo-obstruction. Rumination syndrome is a clinical diagnosis based on symptoms and the absence of structural disease. In rumination syndrome, episodes of regurgitation occur with the majority of meals. It has been described as a “meal-in, meal-out, day-in, day-out” behavior. The repetitive regurgitation of gastric contents typically begins within minutes of the start of the meal and can persist for 30 minutes or longer after the meal is completed. In contrast, individuals with gastroparesis typically vomit later in the postprandial period (typically after 1 hour after the end of the meal), and their symptoms rarely occur on a daily basis. The regurgitation is effortless, is not associated with retching, and can be accompanied by a sensation of belching immediately before the regurgitation or arrival of food into the pharynx. The regurgitated material consists of partially recognizable food, which initially has the pleasant taste of the recently ingested food. Patients usually spit out the regurgitated food once it becomes bitter or sour, which usually occurs within 30 minutes of ingestion. Patients make a conscious decision what to do with the regurgitated food in the pharynx depending on the social situation at the time it occurs, the volume regurgitated, and the taste of the material. The primary symptom in rumination syndrome is the regurgitation of recently ingested food. However, many individuals with rumination syndrome have additional symptoms including nausea, heartburn, abdominal discomfort, diarrhea and/or constipation. Weight loss can also be a prominent feature of rumination syndrome, particularly in the adolescent population. In general, rumination syndrome is more common in females than males. The female predominance of the condition and frequent occurrence of weight loss contribute to the misdiagnosis of rumination syndrome as bulimia and/or anorexia nervosa. Patients with additional symptoms might require further medical evaluation before rumination syndrome can be confidently confirmed as a singular diagnosis. In addition to weight loss, the common medical complications of rumination syndrome are relatively mild and variable and include vitamin and mineral deficiencies and dental problems including cavities and erosion of enamel by stomach acid. Fortunately, with appropriate treatment, many of the associated complications are reversible. On the other hand, complications might result from extensive and sometimes invasive testing or treatment before the diagnosis of rumination syndrome is made. Patients frequently miss school and work as a result of symptoms and might even require hospitalization for evaluation and/or management of symptoms before diagnosis. Significant functional disability related to weight loss, work or school absenteeism, social consequences and embarrassment related to rumination behavior at mealtime, hospitalization, and extensive diagnostic testing have been described in pediatric and adolescent patients with rumination syndrome. Early diagnosis and treatment of rumination syndrome are critical to avoiding these adverse consequences. Data regarding the epidemiology and etiology of rumination syndrome are limited. Many patients are high achievers in school, college, work, and society. The pathophysiologic mechanisms involved in rumination syndrome remain somewhat unclear. However, all observations suggest an adaptation of the belch reflex that overcomes the resistance to retrograde flow provided by the lower esophageal sphincter. The propulsion of gastric content in a retrograde or orad direction is thought to be due to an increase in intra-abdominal pressure generated by the contraction of abdominal muscles. On gastroduodenal manometry testing, this results in a characteristic manometric abnormality (in fact, this is an artifact) that is documented in all intra-abdominal pressure sensors simultaneously called rumination waves or “r” waves (Figure 1). Although the presence of “r” waves is highly suggestive of rumination syndrome, up to 50% of patients will not exhibit these waves during the test, suggesting that the diagnostic utility of this invasive test is low. Although “r” waves were originally considered critical for diagnosis, the information obtained by taking a sufficient clinical history is often adequate to make the diagnosis of rumination syndrome. In general, esophageal pH testing is not advocated in patients with rumination syndrome because changes in esophageal pH are a consequence of rumination, rather than a cause for symptoms. Many patients have evidence of “pathological” gastroesophageal reflux because ambulatory esophageal pH monitoring results are frequently abnormal. However, careful study shows that acid reflux typically occurs within the first hour after a meal, which corresponds to periods of regurgitation of intragastric content. In rumination syndrome, there is little or no reflux of acid in the supine position or at nighttime. Another clue to the diagnosis of rumination syndrome is that despite repetitive changes in pH, the total time that the esophageal pH is less than 4 might be paradoxically low, because food buffers the gastric acid to a pH above 4 during the early postprandial period. When gastric emptying is assessed in patients with rumination syndrome, several caveats exist. Some patients will have mildly delayed gastric emptying as a result of the to-and-fro motion of food that occurs during rumination, which slows the transfer of stomach contents into the distal stomach for trituration before emptying into the small bowel. These patients might be misdiagnosed as having gastroparesis. Another caveat is the potential for expulsion of the radiolabeled meal; when the majority of the meal is expelled, the study cannot be interpreted. Therefore, results of scintigraphic gastric emptying studies should be interpreted with caution in patients with symptoms of rumination. A 4-hour scan might be more helpful than measurement of gastric emptying. This is particularly relevant when the data extrapolated from a 90-minute observation period suggest that the half-time is 190 minutes (more than twice as long as the actual observation period), as in the patient discussed here. By using an intragastric balloon in which the pressure of air could be clamped by means of a barostat, Thumshirn et al demonstrated that the mechanism of rumination involves relaxation of the lower esophageal sphincter in response to lower pressures introduced into the fundus and increased gastric sensitivity. In healthy adults, there is active contraction at the esophagogastric junction and increased lower esophageal sphincter pressure during periods of increased intra-abdominal pressure; tonic contraction of the crural diaphragm is the proposed mechanism for this response. This mechanism is altered in the setting of rumination syndrome, because transient lower esophageal sphincter relaxations occur after abdominal straining events. Approximately 50% of patients with rumination syndrome have evidence for impaired gastric accommodation when assessed by using either gastric barostat or gastric volume measurement (single photo emission computed tomography) techniques. The mainstay of treatment for rumination syndrome involves behavioral modification. Wagaman et al introduced a behavioral approach focusing on diaphragmatic breathing as a means to create a competing behavior and break the cycle of activities shown in Figure 1 that result in food regurgitation. The rumination behavior is eliminated because rumination and the competing response cannot be performed at the same time. This approach was so effective that it was difficult to set up a randomized controlled trial. Although controlled treatment trials are lacking, the outcomes for patients with rumination syndrome who undergo behavioral treatment appear to be quite good. In the largest study in the literature of 147 children and adolescents with rumination syndrome by Chial et al, outcome data were available for 46 patients who had undergone behavioral treatment. There was a positive impact on symptoms in 85% of patients, with complete cessation of the behavior in 30% and improvement in 55%. In general, medications including acid-blocking agents, prokinetic medications, antiemetics, anticholinergics, anxiolytics, and antidepressants are not helpful at improving symptoms in patients with rumination syndrome. O’Brien et al documented that a large number of these medications were tried in open-labeled fashion in clinical practice, and none was effective. Fundoplication has not proved to be effective in patients with rumination syndrome. Considering that the apparent delay in gastric emptying is due to the regurgitation behavior rather than gastroparesis, gastric pacing and electrical stimulation are not recommended in patients with rumination syndrome. In general, it is recommended that patients with rumination syndrome eat a balanced diet, although it usually prudent to reintroduce solid food slowly during the first week or two, because the regurgitation might still occur until the patient masters the diaphragmatic breathing. After behavioral treatment, weight loss and nutritional abnormalities should reverse. Rumination syndrome complicated by comorbid medical, psychological, or psychiatric conditions might require additional therapeutic interventions including referral to a psychologist or psychiatrist. Many unanswered questions regarding rumination syndrome remain: What are the prevalence and incidence of rumination syndrome? Who is at risk for developing rumination syndrome? What proportion of patients has impaired gastric accommodation, and does impaired accommodation contribute to the development of symptoms? What is the mechanism of impaired accommodation? What is the mechanism of raised intra-abdominal pressure that propels food in an orad direction? How does diaphragmatic breathing result in improvement in the patient’s symptoms? Do other forms of biofeedback modification work? Rumination syndrome is classified as an eating disorder in the Diagnostic and Statistical Manual, Fourth Edition criteria, and a minority of patients with rumination are bulimic; what is the risk of one syndrome developing into the other? Rumination syndrome is recognized in infants and toddlers, children and adolescents, and adults of normal intelligence. The Rome III consensus criteria have identified the syndrome in these 3 different age groups, and the slight variations in presentation are demonstrated by variation in the symptom-based criteria for diagnosis. These are summarized in Table 1.Table 1Rome III Consensus Criteria for Rumination SyndromeInfantAdolescentAdultMust include all of the following for at least 3 months:1Repetitive contractions of the abdominal muscles, diaphragm, and tongue2Regurgitation of gastric content into the mouth, which is either expectorated or re-chewed and re-swallowed3Three or more of the following:aOnset between 3 and 8 monthsbDoes not respond to management for gastroesophageal reflux disease or to anticholinergic drugs, hand restraints, formula changes, and gavage or gastrostomy feedingscUnaccompanied by signs of nausea or distressdDoes not occur during sleep and when the infant is interacting with individuals in the environmentMust include all of the following:1Repeated painless regurgitation and re-chewing or expulsion of food thatabegin soon after ingestion of a mealbdo not occur during sleepcdo not respond to standard treatment for gastroesophageal reflux2No retching3No evidence of an inflammatory, anatomic, metabolic, or neoplastic process that explains the subject’s symptomsMust include both of the following:1Persistent or recurrent regurgitation of recently ingested food into the mouth with subsequent spitting or re-mastication and swallowing2Regurgitation is not preceded by retchingSupportive criteria:1Regurgitation events are usually not preceded by nausea2Cessation of the process when the regurgitated material becomes acidic3Regurgitant contains recognizable food with a pleasant taste Open table in a new tab The rumination behavior is also recognized in people who are mentally challenged, in whom the treatment, prognosis, and complications differ. The patient underwent a repeat gastric emptying test with a radiolabeled solid egg meal, which showed 25%, 47%, and 97% emptied at 1, 2, and 4 hours, respectively (normal values, 11%–39%, 40%–76%, and 85%–98%, respectively). The patient was referred for behavioral therapy including instruction on the use of diaphragmatic breathing during the intraprandial and postprandial periods. Within 1 week of treatment, her regurgitation frequency decreased from at least 1 episode after each meal to an average of 1 regurgitation episode per day. The frequency steadily decreased and had ceased within 4 weeks. Initially, her diet was reduced in fat and nondigestible solid content, but after 2 weeks, her diet was liberalized. She made an excellent recovery and required no further dietary restriction or medications. The authors thank Mrs Cindy Stanislav for excellent secretarial assistance.