- Email: [email protected]
Rumination syndrome in adolescents
R
Rumination syndrome in adolescents Seema Khan, MD, Paul E. Hyman, MD, Jose Cocjin, MD, and Carlo Di Lorenzo, MD
Objectives: To evaluate the clinical presentation and to assess the usefulness of antroduodenal manometry (ADM) and the results of multidisciplinary team management in 12 neurologically normal adolescents (9 girls) with rumination. Study design: All patients had extensive investigations that ruled out other causes of their chronic symptoms. We performed ADM in all patients. A multidisciplinary approach was used for the nutritional and behavioral rehabilitation of these patients. Results: The median age at presentation was 14 years (range, 9-19 years), and the average duration of symptoms was 17 months. All patients complained of postprandial, effortless regurgitation, and the majority had weight loss and abdominal pain. Results of fasting ADM were normal in all. The postprandial ADM showed brief, simultaneous pressure increases at all recording sites, associated with regurgitation in 8 patients. No emesis was observed in the other 4 children during the study. Treatment included nutritional support in combination with antidepressants and anxiolytics (n = 6), cognitive therapy with biofeedback or relaxation techniques (n = 7), and pain management (n = 2). Resolution or improvement of symptoms was seen in 10 of the 12 patients, and successful transition to oral feedings was achieved in all during the follow-up period, which ranged from 5 to 36 months. Conclusions: Rumination is a distinct functional gastrointestinal disorder of otherwise healthy children and adolescents, which can be diagnosed on the basis of clinical features. The ADM shows a characteristic pattern and rules out motility disorders that are often confused with rumination. A multidisciplinary team approach is associated with satisfactory recovery in most patients. (J Pediatr 2000;136:528-31)
From the Divisions of Pediatric Gastroenterology, Children’s Hospital of Pittsburgh, Pittsburgh, Pennsylvania; and Children’s Hospital of Orange County, Orange, California.
Submitted for publication Apr 8, 1999; revisions received July 7, 1999, and Oct 8, 1999; accepted Oct 20, 1999. Reprint requests: Carlo Di Lorenzo, MD, Associate Professor of Pediatrics, Division of Gastroenterology, Children’s Hospital of Pittsburgh, 3705 Fifth Ave, Pittsburgh, PA 15213-2583. Copyright © 2000 by Mosby, Inc. 0022-3476/2000/$12.00 + 0 9/21/103953 doi:10.1067/mpd.2000.103953
528
In mentally handicapped individuals the prevalence of rumination is 6% to 10%.1 Reports of rumination in adults with normal intelligence suggest that its prevalence is underestimated.2,3 In children and adolescents, rumination is confused with other conditions that cause vomiting, such as gastroesophageal reflux disease, gastroparesis, motility disorders, and eating disorders. Many clinicians do not consider rumination among the possible diagnoses, and therefore the symptoms of rumination
syndrome often continue untreated for long periods. Rumination is defined by DSM III-R criteria as “repeated regurgitation of food, with weight loss or failure to gain expected weight, developing after a period of normal functioning. Partially digested food is brought back into the mouth without nausea, retching, disgust, or associated gastroenterological disorders. The food is then ejected from the mouth or reswallowed.”4 Several theories have been proposed, but none fully explain the pathophysiologic mechanisms involved in rumination.2,3,5,6 Recently, Thumshirn et al7 showed that rumination syndrome is characterized by higher gastric sensitivity and decreased threshold for lower esophageal sphincter relaxation during gastric distention. Few studies describe rumination in healthy children and adolescents. The aims of this study are to describe the clinical characteristics, assess the utility of antroduodenal manometry as a diagnostic tool, and report outcomes in children and adolescents with rumination.
ADM Antroduodenal manometry TPN Total parenteral nutrition
PATIENTS AND METHODS We identified patients with rumination by a retrospective review of the data collected between 1994 and 1998 at 2 diagnostic centers for children suspected of having gastrointestinal motility disorders. Either before or after admission, we assessed complete blood counts, serum electrolytes, renal function, amylase, lipase, and liver en-
KHAN ET AL
THE JOURNAL OF PEDIATRICS VOLUME 136, NUMBER 4 zymes; we performed upper gastrointestinal series with small bowel follow through and upper gastrointestinal endoscopies in all patients and radionuclide gastric emptying scan and computerized tomography or magnetic resonance imaging of the brain in some. Anti-secretory agents and prokinetic agents had failed to control the symptoms. Two patients were receiving total parenteral nutrition, and one patient was being fed through a nasojejunal tube. For the purpose of screening subjects for this review, we defined rumination as effortless, postprandial regurgitation of partially digested food into the mouth, followed by either reswallowing or extrusion. We performed ADM in all patients to determine the physiology associated with symptoms. The ADM catheter assembly consisted of 6 to 8 radially oriented orifices, spaced 3 or 5 cm apart. The catheters were perfused with distilled, deionized water by a low-compliance, capillary infusion system at a rate of 0.1 mL/min. After an overnight fast and when adequate sedation was achieved for each patient, we placed the catheter using endoscopic or fluoroscopic guidance with at least one recording site in the antrum and 4 in the small bowel. We began manometric studies only after the patient recovered from sedation. We recorded pressure changes for at least 4 hours during fasting and 2 hours after the voluntary ingestion of a mixed solid and liquid meal. We compared the ADM findings in patients with rumination with those previously reported in children with no upper gastrointestinal symptoms but referred for ADM to rule out generalized gastrointestinal motility disorders.8 We used a multidisciplinary approach for the nutritional and behavioral rehabilitation of the patients. The rehabilitation team included a pediatric gastroenterologist, a mental health professional, a dietician, and sometimes a pain management specialist. Effective reassurance that we made
Figure. Postprandial ADM demonstrates brief, simultaneous pressure increases at all recording sites, coinciding with rumination episodes (+). an, Antrum; du, duodenum.
the correct diagnosis was essential to developing a therapeutic alliance with the child and family.
RESULTS We identified 12 children and adolescents (9 girls) with rumination. We studied these patients between the ages of 9 and 19 years (median age, 14 years) after an average symptom duration of 17 months (range, 1-72 months). None of the patients were developmentally delayed or neurologically handicapped. The working diagnoses before ADM were chronic intestinal pseudo-obstruction in 3 patients, intractable vomiting in 4, post-viral gastroparesis in 2, gastroesophageal reflux disease in 2, and gastritis in the remaining patient. One patient also had a diagnosis of neurofibromatosis type 1. Emesis was universally present in all the patients as an effortless, postprandial event, and in two thirds it occurred within the first 30 minutes of a meal. None of the patients reported reswallowing or nocturnal symptoms. The other associated symp-
toms were abdominal pain (n = 10), weight loss >5 pounds (n = 7), nausea (n = 4), and heartburn (n = 2). We obtained a psychiatric consultation for 10 patients. The mental health diagnoses included depression (n = 3), somatoform disorders (n = 3), Asperger’s syndrome (n = 1), and pain disorder (n = 1). An eating disorder was suspected in 2 patients. In 5 patients the period of school absence was >4 weeks when we initiated the evaluation. The investigations helped to rule out other causes of vomiting before reaching the diagnosis of rumination. Anatomic anomalies were ruled out with contrast radiography in all patients at initial presentation, including a patient who several months later had new findings of superior mesenteric artery syndrome, as a consequence of a 21-pound weight loss. The repeat upper gastrointestinal series showing retention of barium in the descending duodenum to the right of the spine was prompted by observations on ADM of persistent elevation of baseline pressure in the third portion of the duodenum. Two patients had mild histologic 529
KHAN ET AL
esophagitis and gastritis, and all biopsy specimens were negative for Helicobacter pylori. The patient with neurofibromatosis type 1 had depression, but magnetic resonance imaging of the brain, repeated periodically during follow-up, showed no intracranial disease to explain the emesis. Other investigations included an abnormal acid reflux score in 1 of 3 patients who had intraesophageal pH monitoring studies, mildly delayed gastric emptying for solids and liquids in 2 of 8 children, and normal findings on esophageal manometry in all 4 tested patients. Results of ADM were normal during fasting in all patients. In 8 patients brief, simultaneous, postprandial pressure increases associated with regurgitation were observed at all recording sites (Figure). The episodes of regurgitation were not associated with antral contractions or retrograde peristalsis. The other 4 patients had no emesis during the study. Except for the simultaneous pressure increases, the postprandial pattern was normal in all. We used nasojejunal, gastrostomy, or gastrojejunostomy tube feedings in 6 patients and total parenteral nutrition for a period of 2 weeks in one and intermittently for 2 years in another patient. In 6 children, anxiolytics or antidepressants were used in combination with nutritional and behavioral rehabilitation. Psychotherapy, biofeedback, and relaxation techniques were recommended to all and used for 7 patients. Over time, there was an improvement, as assessed by a decrease in the frequency of rumination episodes, in 10 patients, with eventual resolution in 9. All patients previously requiring nutritional support returned to exclusively oral feedings during the course of management. The diagnosis of rumination was initially unacceptable to parents of 2 patients, one of whom has continued to experience symptoms despite medical treatment. The other patient has only occasional rumination, and the diagnosis has become acceptable to the family after repeated reassurance. The patient with 530
THE JOURNAL OF PEDIATRICS APRIL 2000 neurofibromatosis type 1 continues to experience rumination episodes.
DISCUSSION Rumination is classified as a functional bowel disorder in adults.9 By definition, functional disorders are chronic or recurrent symptoms that occur in the absence of any readily defined anatomic or biochemical abnormality (ie, arising in the absence of disease). Traditionally, functional conditions were diagnosed by exclusion, after efforts to identify metabolic, infectious, neoplastic, and structural diseases had been exhausted. This approach has one serious limitation: for patients with a functional disorder and the clinicians who care for them, the discomfort, anxieties, and frustrations increase with each negative test result and each treatment failure. This study demonstrates that rumination occurs in otherwise healthy children and adolescents. Diagnosis can be based on characteristic symptoms with exclusion of other causes of chronic vomiting such as gastroparesis or neurologic disorders. The earliest human reports of rumination date back to the 17th century and include its description by the famous physician, Edouard Brown Sequard, who exhibited rumination as a learned behavior while self-experimenting with swallowed sponges and gastric acid responses to various foods.10 The timing of rumination constitutes an important differentiating point from gastroparesis, in which vomiting is frequently long delayed after eating. Patients with gastroparesis also have delayed gastric emptying and often postprandial antral hypomotility,11 a feature absent in all our study patients. Our patients had a high frequency of associated symptoms such as nausea and abdominal pain in the absence of organic disease; this may be because of somatization, the unconscious conversion of emotional distress into physical symptoms. Several patients had visceral pain-
associated disability syndrome.12 Heartburn was reported infrequently; it is reported that heartburn usually abolishes rumination.3,13 The mild esophagitis and gastritis in 2 patients were not of sufficient severity to explain their symptoms. We doubt that severe esophagitis occurs in patients with rumination, because the vomitus contains mainly food, and acid, if present, does not have prolonged contact with the esophageal mucosa. Rumination is believed to be due to coexistence of functional and psychiatric abnormalities.14,15 Some patients with rumination have serious psychologic disturbances such as depression and suicidal ideation.2,16 In our series one child of divorced parents had onset of symptoms coinciding with the news of her father’s visit. Rumination may be self-limited in response to situational stress in healthy children.17 Two of the patients we report were believed to have eating disorders, and rumination was considered a learned behavior to control weight. Other studies have proposed a role for stress in the onset of eating disorders.3,18 Among other factors, a preceding viral illness is also thought to be a possible cause, subsequently leading to a learned behavior.2,3,15 The referring diagnosis of post-viral gastroparesis in 2 patients was ruled out by normal findings on gastric emptying studies and the adequate antral motor responses after ingestion of a solid meal. The physical consequences of rumination can be serious and include weight loss, malnutrition, chronic pain producing functional disability, halitosis, dental erosions, and electrolyte disturbances in long-standing cases. In the series of 38 adolescents and adults described by O’Brien et al,3 reswallowing was present in 50%, weight loss in 42%, and bulimia nervosa in 13%. It has been hypothesized that patients with rumination use neural pathways to induce voluntary lower esophageal sphincter relaxation by abdominal wall contractions. ADM shows brief simultaneous pressure increases (named R
KHAN ET AL
THE JOURNAL OF PEDIATRICS VOLUME 136, NUMBER 4 waves) associated with regurgitation, which represent a conscious or unconscious act to increase the intra-abdominal pressure to induce regurgitation. In adults with normal intelligence and rumination, weight loss was present in 38%, R waves were observed in 87% of patients who had ADM, and decreased lower esophageal sphincter pressure was identified in a minority of patients who underwent esophageal manometry.16 The management of rumination in a healthy child or adolescent starts with patient and family education and reassurance. Successful management of rumination with the use of behavior modification, biofeedback, and various relaxation techniques in uncontrolled studies has also been reported.2,16,19-21 From our retrospective data, we cannot conclude that the morbidity of childhood rumination is related to the misdiagnosis and over-medicalization of functional symptoms. Still, it seemed that after the rumination diagnosis was made, an average of 17 months after the initial symptoms, the focus shifted from diagnosis to nutritional, educational, and/or emotional rehabilitation. Recovery was satisfactory in the majority of patients once we stopped uncomfortable diagnostic procedures and initiated treatment. In follow-up ranging from 5 to 36 months, rumination improved in 10 patients and eventually ceased in 9; all 12 returned to normal activities, including school attendance. Even though the diagnosis of rumination can be made without ADM, the pattern of brief, simultaneous pressure increases in all recording sites associated with emesis during the study is characteristic. When compared with children given a diagnosis of intestinal pseudoobstruction, cyclic vomiting syndrome, or post-viral gastroparesis, the R waves stand out as a unique feature of ADM in rumination.22 ADM requested by the referring physicians confirmed a symptom-based diagnosis of rumination in 8 of 12 patients. Moreover, the presence of migrating motor complexes during fasting and a normal postprandial motor
pattern on ADM, as previously described in children without upper gastrointestinal complaints,8 was inconsistent with the working diagnoses of pseudo-obstruction or gastroparesis. In conclusion, there is a need to recognize rumination as a functional gastrointestinal disorder in children and adolescents. Our experience suggests that a multidisciplinary team approach reduces symptoms and associated disturbances in most patients. The specific modalities of this approach may be modified according to the associated features of any psychiatric illnesses or other functional gastrointestinal symptoms in the child and family. It is hoped that a growing recognition of its presentation by pediatricians will lead to early intervention and hence prevention of its attendant medical complications, as well as invasive and sometimes unnecessary investigations.
REFERENCES 1. Fredericks DW, Carr JE, Williams WL. Overview of the treatment of rumination disorder for adults in a residential setting. J Behav Ther Exp Psychiatry 1998;29:31-40. 2. Amarnath RP, Abell TL, Malagelada JR. The rumination syndrome in adults. A characteristic manometric pattern. Ann Intern Med 1986;105:513-8. 3. O’Brien MD, Bruce BK, Camilleri M. The rumination syndrome: clinical features rather than manometric diagnosis. Gastroenterology 1995;108:1024-9. 4. American Psychiatric Association. Diagnostic and statistical manual of mental disorders. 3rd ed, revised. Washington (DC): American Psychiatric Association; 1987. 5. Reynolds RPE, Lloyd DA. Manometric study of a ruminator. J Clin Gastroenterol 1986;8:127-30. 6. Malcolm A, Thumshirn MB, Camilleri M, Williams DE. Rumination syndrome (review). Mayo Clin Proc 1997;72:646-52. 7. Thumshirn M, Camilleri M, Hanson RB, Williams DE, Schei AJ, Kammer PP. Gastric mechanosensory and lower esophageal sphincter function in rumination syndrome. Am J Physiol 1998; 275:G314-21. 8. Uc A, Hoon A, Di Lorenzo C, Hyman
9.
10. 11.
12.
13.
14.
15.
16.
17.
18. 19.
20.
21.
22.
PE. Antroduodenal manometry in children with no upper gastrointestinal symptoms. Scand J Gastroenterol 1997;32:681-5. Drossman DA, Richter JE, Talley NJ, Thompson WG, Corazziari E, Whitehead WE. The functional gastrointestinal disorders: diagnosis, pathophysiology, and treatment. Boston: Little, Brown, Co; 1994. p. 1-370. Kanner L. Historical notes on rumination in man. Med Life 1936;43:27-60. Sigurdsson L, Flores A, Putnam PE, Hyman PE, Di Lorenzo C. Postviral gastroparesis: presentation, treatment and outcome. J Pediatr 1997;130:751-4. Bursch B, Walco GA, Zeltzer L. Clinical assessment and management of chronic pain and pain-associated disability syndromes. J Dev Behav Pediatr 1998;19:45-53. Shay SS, Johnson LF, Wong RKH, Curtis DJ, Rosenthal R, Lomett JR, et al. Rumination, heartburn and daytime gastroesophageal reflux. J Clin Gastroenterol 1986;8:115-26. Fleisher DR. Functional vomiting disorders in infancy: innocent vomiting, nervous vomiting, and infant rumination syndrome. J Pediatr 1994;125:S84-S94. Levine DF, Wingate DL, Pfeffer JM, Butcher P. Habitual rumination: a benign disorder. Br Med J 1983;287:255-6. Soykan I, Chen J, Kendall B, McCallum RW. The rumination syndrome. Clinical and manometric profile, therapy, and long-term outcome. Dig Dis Sci 1997;42:1866-72. Reis S. Rumination in two developmentally normal children: case report and review of the literature. J Fam Pract 1994;38:521-3. Fairburn CG, Cooper PJ. Rumination in bulimia nervosa. BMJ 1984;288:826-7. Parther CM, Litzinger KL, Camilleri M, Thumshirn M, Williams DE. An open trial of cognitive behavioral intervention in the treatment of rumination syndrome [abstract]. Gastroenterology 1997;112:A808. Johnson WG, Corrigan SA, Crusco AH, Jarell MP. Behavioral assessment and treatment of post-prandial regurgitation. J Clin Gastroenterol 1987;9:679-84. Wagaman JR, Williams DE, Camilleri M. Behavioral intervention for the treatment of rumination [case report]. J Pediatr Gastroenterol Nutr 1998;27:596-8. Khan S, Flores AF, Sigurdsson L, Di Lorenzo C. Antroduodenal manometry (ADM) in the evaluation of children with chronic vomiting [abstract]. Gastroenterology 1999;116:G2444. 531
Rumination syndrome in adolescents Seema Khan, MD, Paul E. Hyman, MD, Jose Cocjin, MD, and Carlo Di Lorenzo, MD
Objectives: To evaluate the clinical presentation and to assess the usefulness of antroduodenal manometry (ADM) and the results of multidisciplinary team management in 12 neurologically normal adolescents (9 girls) with rumination. Study design: All patients had extensive investigations that ruled out other causes of their chronic symptoms. We performed ADM in all patients. A multidisciplinary approach was used for the nutritional and behavioral rehabilitation of these patients. Results: The median age at presentation was 14 years (range, 9-19 years), and the average duration of symptoms was 17 months. All patients complained of postprandial, effortless regurgitation, and the majority had weight loss and abdominal pain. Results of fasting ADM were normal in all. The postprandial ADM showed brief, simultaneous pressure increases at all recording sites, associated with regurgitation in 8 patients. No emesis was observed in the other 4 children during the study. Treatment included nutritional support in combination with antidepressants and anxiolytics (n = 6), cognitive therapy with biofeedback or relaxation techniques (n = 7), and pain management (n = 2). Resolution or improvement of symptoms was seen in 10 of the 12 patients, and successful transition to oral feedings was achieved in all during the follow-up period, which ranged from 5 to 36 months. Conclusions: Rumination is a distinct functional gastrointestinal disorder of otherwise healthy children and adolescents, which can be diagnosed on the basis of clinical features. The ADM shows a characteristic pattern and rules out motility disorders that are often confused with rumination. A multidisciplinary team approach is associated with satisfactory recovery in most patients. (J Pediatr 2000;136:528-31)
From the Divisions of Pediatric Gastroenterology, Children’s Hospital of Pittsburgh, Pittsburgh, Pennsylvania; and Children’s Hospital of Orange County, Orange, California.
Submitted for publication Apr 8, 1999; revisions received July 7, 1999, and Oct 8, 1999; accepted Oct 20, 1999. Reprint requests: Carlo Di Lorenzo, MD, Associate Professor of Pediatrics, Division of Gastroenterology, Children’s Hospital of Pittsburgh, 3705 Fifth Ave, Pittsburgh, PA 15213-2583. Copyright © 2000 by Mosby, Inc. 0022-3476/2000/$12.00 + 0 9/21/103953 doi:10.1067/mpd.2000.103953
528
In mentally handicapped individuals the prevalence of rumination is 6% to 10%.1 Reports of rumination in adults with normal intelligence suggest that its prevalence is underestimated.2,3 In children and adolescents, rumination is confused with other conditions that cause vomiting, such as gastroesophageal reflux disease, gastroparesis, motility disorders, and eating disorders. Many clinicians do not consider rumination among the possible diagnoses, and therefore the symptoms of rumination
syndrome often continue untreated for long periods. Rumination is defined by DSM III-R criteria as “repeated regurgitation of food, with weight loss or failure to gain expected weight, developing after a period of normal functioning. Partially digested food is brought back into the mouth without nausea, retching, disgust, or associated gastroenterological disorders. The food is then ejected from the mouth or reswallowed.”4 Several theories have been proposed, but none fully explain the pathophysiologic mechanisms involved in rumination.2,3,5,6 Recently, Thumshirn et al7 showed that rumination syndrome is characterized by higher gastric sensitivity and decreased threshold for lower esophageal sphincter relaxation during gastric distention. Few studies describe rumination in healthy children and adolescents. The aims of this study are to describe the clinical characteristics, assess the utility of antroduodenal manometry as a diagnostic tool, and report outcomes in children and adolescents with rumination.
ADM Antroduodenal manometry TPN Total parenteral nutrition
PATIENTS AND METHODS We identified patients with rumination by a retrospective review of the data collected between 1994 and 1998 at 2 diagnostic centers for children suspected of having gastrointestinal motility disorders. Either before or after admission, we assessed complete blood counts, serum electrolytes, renal function, amylase, lipase, and liver en-
KHAN ET AL
THE JOURNAL OF PEDIATRICS VOLUME 136, NUMBER 4 zymes; we performed upper gastrointestinal series with small bowel follow through and upper gastrointestinal endoscopies in all patients and radionuclide gastric emptying scan and computerized tomography or magnetic resonance imaging of the brain in some. Anti-secretory agents and prokinetic agents had failed to control the symptoms. Two patients were receiving total parenteral nutrition, and one patient was being fed through a nasojejunal tube. For the purpose of screening subjects for this review, we defined rumination as effortless, postprandial regurgitation of partially digested food into the mouth, followed by either reswallowing or extrusion. We performed ADM in all patients to determine the physiology associated with symptoms. The ADM catheter assembly consisted of 6 to 8 radially oriented orifices, spaced 3 or 5 cm apart. The catheters were perfused with distilled, deionized water by a low-compliance, capillary infusion system at a rate of 0.1 mL/min. After an overnight fast and when adequate sedation was achieved for each patient, we placed the catheter using endoscopic or fluoroscopic guidance with at least one recording site in the antrum and 4 in the small bowel. We began manometric studies only after the patient recovered from sedation. We recorded pressure changes for at least 4 hours during fasting and 2 hours after the voluntary ingestion of a mixed solid and liquid meal. We compared the ADM findings in patients with rumination with those previously reported in children with no upper gastrointestinal symptoms but referred for ADM to rule out generalized gastrointestinal motility disorders.8 We used a multidisciplinary approach for the nutritional and behavioral rehabilitation of the patients. The rehabilitation team included a pediatric gastroenterologist, a mental health professional, a dietician, and sometimes a pain management specialist. Effective reassurance that we made
Figure. Postprandial ADM demonstrates brief, simultaneous pressure increases at all recording sites, coinciding with rumination episodes (+). an, Antrum; du, duodenum.
the correct diagnosis was essential to developing a therapeutic alliance with the child and family.
RESULTS We identified 12 children and adolescents (9 girls) with rumination. We studied these patients between the ages of 9 and 19 years (median age, 14 years) after an average symptom duration of 17 months (range, 1-72 months). None of the patients were developmentally delayed or neurologically handicapped. The working diagnoses before ADM were chronic intestinal pseudo-obstruction in 3 patients, intractable vomiting in 4, post-viral gastroparesis in 2, gastroesophageal reflux disease in 2, and gastritis in the remaining patient. One patient also had a diagnosis of neurofibromatosis type 1. Emesis was universally present in all the patients as an effortless, postprandial event, and in two thirds it occurred within the first 30 minutes of a meal. None of the patients reported reswallowing or nocturnal symptoms. The other associated symp-
toms were abdominal pain (n = 10), weight loss >5 pounds (n = 7), nausea (n = 4), and heartburn (n = 2). We obtained a psychiatric consultation for 10 patients. The mental health diagnoses included depression (n = 3), somatoform disorders (n = 3), Asperger’s syndrome (n = 1), and pain disorder (n = 1). An eating disorder was suspected in 2 patients. In 5 patients the period of school absence was >4 weeks when we initiated the evaluation. The investigations helped to rule out other causes of vomiting before reaching the diagnosis of rumination. Anatomic anomalies were ruled out with contrast radiography in all patients at initial presentation, including a patient who several months later had new findings of superior mesenteric artery syndrome, as a consequence of a 21-pound weight loss. The repeat upper gastrointestinal series showing retention of barium in the descending duodenum to the right of the spine was prompted by observations on ADM of persistent elevation of baseline pressure in the third portion of the duodenum. Two patients had mild histologic 529
KHAN ET AL
esophagitis and gastritis, and all biopsy specimens were negative for Helicobacter pylori. The patient with neurofibromatosis type 1 had depression, but magnetic resonance imaging of the brain, repeated periodically during follow-up, showed no intracranial disease to explain the emesis. Other investigations included an abnormal acid reflux score in 1 of 3 patients who had intraesophageal pH monitoring studies, mildly delayed gastric emptying for solids and liquids in 2 of 8 children, and normal findings on esophageal manometry in all 4 tested patients. Results of ADM were normal during fasting in all patients. In 8 patients brief, simultaneous, postprandial pressure increases associated with regurgitation were observed at all recording sites (Figure). The episodes of regurgitation were not associated with antral contractions or retrograde peristalsis. The other 4 patients had no emesis during the study. Except for the simultaneous pressure increases, the postprandial pattern was normal in all. We used nasojejunal, gastrostomy, or gastrojejunostomy tube feedings in 6 patients and total parenteral nutrition for a period of 2 weeks in one and intermittently for 2 years in another patient. In 6 children, anxiolytics or antidepressants were used in combination with nutritional and behavioral rehabilitation. Psychotherapy, biofeedback, and relaxation techniques were recommended to all and used for 7 patients. Over time, there was an improvement, as assessed by a decrease in the frequency of rumination episodes, in 10 patients, with eventual resolution in 9. All patients previously requiring nutritional support returned to exclusively oral feedings during the course of management. The diagnosis of rumination was initially unacceptable to parents of 2 patients, one of whom has continued to experience symptoms despite medical treatment. The other patient has only occasional rumination, and the diagnosis has become acceptable to the family after repeated reassurance. The patient with 530
THE JOURNAL OF PEDIATRICS APRIL 2000 neurofibromatosis type 1 continues to experience rumination episodes.
DISCUSSION Rumination is classified as a functional bowel disorder in adults.9 By definition, functional disorders are chronic or recurrent symptoms that occur in the absence of any readily defined anatomic or biochemical abnormality (ie, arising in the absence of disease). Traditionally, functional conditions were diagnosed by exclusion, after efforts to identify metabolic, infectious, neoplastic, and structural diseases had been exhausted. This approach has one serious limitation: for patients with a functional disorder and the clinicians who care for them, the discomfort, anxieties, and frustrations increase with each negative test result and each treatment failure. This study demonstrates that rumination occurs in otherwise healthy children and adolescents. Diagnosis can be based on characteristic symptoms with exclusion of other causes of chronic vomiting such as gastroparesis or neurologic disorders. The earliest human reports of rumination date back to the 17th century and include its description by the famous physician, Edouard Brown Sequard, who exhibited rumination as a learned behavior while self-experimenting with swallowed sponges and gastric acid responses to various foods.10 The timing of rumination constitutes an important differentiating point from gastroparesis, in which vomiting is frequently long delayed after eating. Patients with gastroparesis also have delayed gastric emptying and often postprandial antral hypomotility,11 a feature absent in all our study patients. Our patients had a high frequency of associated symptoms such as nausea and abdominal pain in the absence of organic disease; this may be because of somatization, the unconscious conversion of emotional distress into physical symptoms. Several patients had visceral pain-
associated disability syndrome.12 Heartburn was reported infrequently; it is reported that heartburn usually abolishes rumination.3,13 The mild esophagitis and gastritis in 2 patients were not of sufficient severity to explain their symptoms. We doubt that severe esophagitis occurs in patients with rumination, because the vomitus contains mainly food, and acid, if present, does not have prolonged contact with the esophageal mucosa. Rumination is believed to be due to coexistence of functional and psychiatric abnormalities.14,15 Some patients with rumination have serious psychologic disturbances such as depression and suicidal ideation.2,16 In our series one child of divorced parents had onset of symptoms coinciding with the news of her father’s visit. Rumination may be self-limited in response to situational stress in healthy children.17 Two of the patients we report were believed to have eating disorders, and rumination was considered a learned behavior to control weight. Other studies have proposed a role for stress in the onset of eating disorders.3,18 Among other factors, a preceding viral illness is also thought to be a possible cause, subsequently leading to a learned behavior.2,3,15 The referring diagnosis of post-viral gastroparesis in 2 patients was ruled out by normal findings on gastric emptying studies and the adequate antral motor responses after ingestion of a solid meal. The physical consequences of rumination can be serious and include weight loss, malnutrition, chronic pain producing functional disability, halitosis, dental erosions, and electrolyte disturbances in long-standing cases. In the series of 38 adolescents and adults described by O’Brien et al,3 reswallowing was present in 50%, weight loss in 42%, and bulimia nervosa in 13%. It has been hypothesized that patients with rumination use neural pathways to induce voluntary lower esophageal sphincter relaxation by abdominal wall contractions. ADM shows brief simultaneous pressure increases (named R
KHAN ET AL
THE JOURNAL OF PEDIATRICS VOLUME 136, NUMBER 4 waves) associated with regurgitation, which represent a conscious or unconscious act to increase the intra-abdominal pressure to induce regurgitation. In adults with normal intelligence and rumination, weight loss was present in 38%, R waves were observed in 87% of patients who had ADM, and decreased lower esophageal sphincter pressure was identified in a minority of patients who underwent esophageal manometry.16 The management of rumination in a healthy child or adolescent starts with patient and family education and reassurance. Successful management of rumination with the use of behavior modification, biofeedback, and various relaxation techniques in uncontrolled studies has also been reported.2,16,19-21 From our retrospective data, we cannot conclude that the morbidity of childhood rumination is related to the misdiagnosis and over-medicalization of functional symptoms. Still, it seemed that after the rumination diagnosis was made, an average of 17 months after the initial symptoms, the focus shifted from diagnosis to nutritional, educational, and/or emotional rehabilitation. Recovery was satisfactory in the majority of patients once we stopped uncomfortable diagnostic procedures and initiated treatment. In follow-up ranging from 5 to 36 months, rumination improved in 10 patients and eventually ceased in 9; all 12 returned to normal activities, including school attendance. Even though the diagnosis of rumination can be made without ADM, the pattern of brief, simultaneous pressure increases in all recording sites associated with emesis during the study is characteristic. When compared with children given a diagnosis of intestinal pseudoobstruction, cyclic vomiting syndrome, or post-viral gastroparesis, the R waves stand out as a unique feature of ADM in rumination.22 ADM requested by the referring physicians confirmed a symptom-based diagnosis of rumination in 8 of 12 patients. Moreover, the presence of migrating motor complexes during fasting and a normal postprandial motor
pattern on ADM, as previously described in children without upper gastrointestinal complaints,8 was inconsistent with the working diagnoses of pseudo-obstruction or gastroparesis. In conclusion, there is a need to recognize rumination as a functional gastrointestinal disorder in children and adolescents. Our experience suggests that a multidisciplinary team approach reduces symptoms and associated disturbances in most patients. The specific modalities of this approach may be modified according to the associated features of any psychiatric illnesses or other functional gastrointestinal symptoms in the child and family. It is hoped that a growing recognition of its presentation by pediatricians will lead to early intervention and hence prevention of its attendant medical complications, as well as invasive and sometimes unnecessary investigations.
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