Rumination Syndrome in Children and Adolescents: Diagnosis, Treatment, and Prognosis

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Rumination Syndrome in Children and Adolescents: Diagnosis,

Treatment, and Prognosis

Heather J. Chial, MD; Michael Camilleri, MD; Donald E. Williams, PhD*; Kristi Litzinger, MS, LPP*; and
Jean Perrault, MD‡

R
ABSTRACT. Objectives. To characterize the clinical umination syndrome is characterized by the
features, results of diagnostic testing, and treatment out- effortless regurgitation into the mouth of re-
comes for children and adolescents with rumination syn- cently ingested food followed by rechewing
drome. and reswallowing or expulsion.1,2 Individuals with
Methods. Review of the medical records for all 147 classic rumination syndrome typically do not expe-
patients ages 5 to 20 diagnosed with rumination syn- rience heartburn, abdominal pain, or nausea when
drome at our institution between 1975 and 2000. Data are the regurgitation occurs. The syndrome is most com-
presented as mean ⴞ the standard error of the mean. monly seen in infants and the developmentally dis-
Results. Sixty-eight percent were female. Age at di- abled. However, rumination syndrome does occur in
agnosis was 15.0 ⴞ 0.3 years. Symptom duration before children, adolescents, and adults with normal intel-
diagnosis was 2.2 ⴞ 0.3 years, 73% missed school/work,
ligence.3,4
and 46% had been hospitalized because of symptoms.
Individuals with rumination syndrome are often
Before diagnosis, 16 (11%) underwent surgery for evalu-
ation or management of symptoms. Twenty-four (16%)
misdiagnosed or undergo extensive, costly, and in-
had psychiatric disorders; 3.4% had anorexia or bulimia vasive testing before diagnosis. Insufficient aware-
nervosa. All patients described postprandial regurgita- ness of the clinical features of rumination syndrome
tion after almost every meal (2.7 ⴞ 0.1 meals per day). contributes to the underdiagnosis of this important
Weight loss was described by 42.2% (median: 7 kg). Ad- medical condition. Rumination syndrome is fre-
ditional symptoms included: abdominal pain, 38%; con- quently confused with bulimia nervosa, gastro-
stipation, 21%; nausea, 17%; and diarrhea, 8%. Structural esophageal reflux disease, and upper gastrointestinal
studies were normal. Gastric emptying of solids at 4 motility disorders including gastroparesis and
hours was delayed in 26 of 56 patients. Esophageal pH chronic intestinal pseudo-obstruction. Complications
testing in 24 patients showed reflux/regurgitation in 54%. of rumination syndrome include weight loss, malnu-
Gastroduodenal manometry in 65 patients showed char- trition, dental erosions, halitosis, electrolyte abnor-
acteristic rumination-waves in 40%. Outcome data (at malities, and significant functional disability.3
median follow-up 10 months) were available for 54 pa- Rumination syndrome is a clinical diagnosis based
tients. Symptoms resolved in 16 (30%) and improved in on symptoms and the absence of structural disease.3
30 (56%). Although the Rome II diagnostic groups include
Conclusions. Recognition of the clinical features of childhood functional gastrointestinal disorders, only
rumination syndrome in children and adolescents is es- “infant rumination syndrome” is described in detail
sential; the diagnosis is often delayed and associated and pertains to infants with symptom onset before 8
with morbidity. Extensive diagnostic testing is un- months of age.5 Diagnostic criteria for children and
necessary. Early behavioral therapy is advocated, and
adolescents with rumination beginning after infancy
patient outcomes are generally favorable. Pediatrics 2003;
have not been defined.
111:158 –162; rumination, regurgitation, pediatric, motil-
ity, reflux.
The purpose of this study was to further charac-
terize the clinical features, results of diagnostic
testing, and treatment outcomes for children and
ABBREVIATION. CT, computed tomography. adolescents between the ages of 5 and 20 with rumi-
nation syndrome. We perceived that review of a
large cohort of patients in the 5- to 20-year age group
was necessary to develop future consensus criteria
for rumination syndrome in children and adoles-
From the Clinical Enteric Neuroscience Translational and Epidemiological
cents.
Research (C.E.N.T.E.R.) Program, *Department of Clinical Health Psychol-
ogy, Mayo Clinic Rochester, Rochester, Minnesota; and ‡McGill University METHODS
Health Center, Montreal Children’s Hospital, Division of Gastroenterology
and Nutrition, Montreal, Canada. Patients
Received for publication Mar 26, 2002; accepted Jun 27, 2002. A computerized diagnostic index was used to identify children
Reprint requests to (M.C.) Clinical Enteric Neuroscience Translational and and adolescents between the ages of 5 and 20 diagnosed with
Epidemiological Research (C.E.N.T.E.R.) Program, Charlton 7-154, Mayo rumination syndrome at Mayo Clinic Rochester during the 25-year
Clinic Rochester, 200 First St SW, Rochester, MN 55905. E-mail address: period between 1975 and 2000. One hundred forty-seven patients
camilleri.michael@mayo.edu were identified. Data extracted included demographics, clinical
PEDIATRICS (ISSN 0031 4005). Copyright © 2003 by the American Acad- features, past medical history, social history, evaluation, treat-
emy of Pediatrics. ment, and outcome. The study was approved by the Mayo Insti-

158 PEDIATRICS Vol. 111 No. 1 January 2003


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tutional Review Board, and authorization for the use of the med- patient who underwent multiple operations includ-
ical records for research purposes was confirmed before access ing pyloroplasty, partial colectomy with ileostomy,
was provided to the medical records.
and cholecystectomy. The clinical history of the latter
Statistical Analysis patient was reported elsewhere.2 None of the pa-
Descriptive data are displayed as number of patients (percent),
tients had symptomatic improvement postopera-
or mean ⫾ the standard error or the mean (n ⫽ number of patients tively.
with data available if less than the total number, 147). The Mann-
Whitney U test was used for comparisons of means between
Social History and Family History
groups. The ␹2 test was used for comparisons of proportions.
Eighty-five percent of patients lived at home with
RESULTS their families, and 12% were college students who
Demographics lived on campus (n ⫽ 131); the remaining 3% were
Of the 147 patients identified, 68.0% were female, married, living with a significant other, or living
and 97.2% were Caucasian. The majority (70.8%) with a foster family. Relatively uncommon was the
were from the midwestern United States (Minnesota, use of alcohol (13 patients), tobacco (14 patients), and
Iowa, Wisconsin, Nebraska, North Dakota, Illinois, recreational drugs (4 patients).
Indiana, South Dakota, and Michigan). These demo- Regarding school performance, 90% of the 39 pa-
graphics reflect the usual ethnic and geographic dis- tients with data available earned A and B grade point
tribution of patients attending Mayo Clinic Roches- averages. Using adjectival terms from the medical
ter. records to categorize scholastic performance (n ⫽
73), 18.8% were excellent students, 58.9% were good
Past Medical History students, 2.7% were fair students, and 9.6% were
Forty-eight (32.7%) patients had prior diagnoses of poor students. (Of note, data regarding school per-
gastrointestinal illnesses; 49 (33.3%) had other med- formance were not available for the 7 patients with
ical diagnoses; and 25 (17%) had concomitant psy- developmental disabilities.)
chiatric disorders including depression (6.1%), anxi-
ety disorder (3.4%), attention-deficit/hyperactivity Clinical Features of Rumination Syndrome
disorder (3.4%), adjustment disorder (1.4%), obses- The age at diagnosis was 15.0 ⫾ 0.3 years as shown
sive compulsive disorder (0.7%), somatoform disor- in Fig 1. Symptom duration before diagnosis was
der (0.7%), and posttraumatic stress disorder (0.7%). 2.2 ⫾ 0.3 years (n ⫽ 126). Age at symptom onset was
Anorexia nervosa and bulimia nervosa were docu- 12.9 ⫾ 0.4 years (n ⫽ 126). Males were younger at
mented in 2 (1.4%) and 3 (2.0%) patients, respec- symptom onset (11.0 ⫾ 0.8 vs 13.8 ⫾ 0.5; P ⫽ .003)
tively. A history of physical and/or sexual abuse was and at the time of diagnosis (13.2 ⫾ 0.7 vs 15.8 ⫾ 0.3
documented in 4 (2.7%) patients. Developmental dis- years; P ⫽ .001) than females.
abilities were present in 7 patients including devel- The body mass index at the time of diagnosis was
opmental delay in 5, autism in 1, and dyslexia in 1. 21.5 ⫾ 0.4 kg/m2 (n ⫽ 119); no gender differences for
body mass index were detected (21.2 ⫾ 0.9 kg/m2 for
Past Surgical History males vs. 21.7 ⫾ 0.4 kg/m2 for females; P ⫽ .3).
Before diagnosis of rumination syndrome, 16 Although ‘vomiting’ was often the presenting
(11%) patients had abdominal operations performed complaint, all but 1 of the 147 patients described
elsewhere for evaluation or management of associ- “effortless” postprandial regurgitation. Episodes of
ated symptoms: 6 appendectomies; 5 cholecystecto- regurgitation occurred after 2.7 ⫾ 0.1 meals per day
mies; 3 laparotomies; 1 duodenal enterostomy; and 1 (n ⫽ 34). By the patients’ estimations, regurgitation

Fig 1. Distribution of age at diagnosis.

ARTICLES 159
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commenced 21.1 ⫾ 5.7 minutes postprandially, me- with prior hospitalizations were older at symptom
dian 10 minutes (n ⫽ 23), and persisted 72.3 ⫾ 9.4 onset (15.0 ⫾ 0.8 vs 12.3 ⫾ 0.8 years; P ⫽ .02) and
minutes (n ⫽ 20). Reswallowing of regurgitated food older at the time of diagnosis (16.4 ⫾ 0.6 vs 14.6 ⫾ 0.6
was described by 25 (17.0%) patients. On the other years, P ⫽ .02) than patients who had not been
hand, remastication of regurgitated food was docu- hospitalized. Of the 74 patients with historical data
mented in 4 (2.7%) patients. Nighttime episodes oc- available regarding weight loss and hospitalizations,
curred in only 3 (2.0%) patients. the prevalence of weight loss was significantly
In 15 (10.2%) patients, specific stressors were iden- higher in patients who had been hospitalized (58.8%
tified just before symptom onset including: 3 with vs 30.0% in patients without prior hospitalizations;
deaths in the immediate family; 3 starting school/ P ⫽ .02).
college; 2 accidents; 2 onset of sports season; 1 pa-
rental divorce; 1 geographical relocation; 1 change in Evaluation of Symptoms Before Diagnosis of
job for father; 1 change in church; and 1 going to Rumination Syndrome
camp. Laboratory studies were performed in 93 (63.3%)
patients; results were normal with the exception of
Associated Symptoms hypokalemia in 1 patient who had been previously
Abdominal pain was an associated symptom in 56 diagnosed with Bartter’s syndrome.
(38.1%) patients. Other digestive tract symptoms in- Results from structural studies performed both at
cluded: constipation in 31 (21.1%), nausea in 25 Mayo Medical Center and elsewhere before consul-
(17.0%), diarrhea in 12 (8.2%), bloating in 6 (4.1%), tation at Mayo including upper gastrointestinal bar-
and dental problems in 5 (3.4%). ium series (performed on 53.7%), esophagogas-
Weight loss was reported in 62 (42.2%) patients; troduodenoscopy (53.7%), abdominal ultrasound
the average weight loss in this group was 9.6 ⫾ 1.0 (31.3%), small bowel follow-through (29.3%), abdom-
kg, (median: 7.1 kg; n ⫽ 56). Patients with weight loss inal computed tomography (CT) scan (18.3%), bar-
were older at symptom onset (14.5 ⫾ 0.6 years vs ium enema (4.9%), flexible sigmoidoscopy (4.1%),
11.6 ⫾ 0.6 years without weight loss; P ⫽ .0003) and video swallow evaluation (2.0%), and colonoscopy
at the time of diagnosis (16.4 ⫾ 0.4 years vs 13.8 ⫾ 0.5 (1.4%) were noncontributory. Brain imaging was done
years without weight loss; P ⬍ .0001). in 30 (20.5%) patients; results were normal by CT scan
in 16, magnetic resonance imaging scan in 11, and CT
Medications Before Diagnosis and magnetic resonance imaging scans in 3.
Before evaluation at Mayo Clinic Rochester, 78 Scintigraphic gastric emptying at 4 hours was de-
(53.1%) patients were prescribed medications specif- layed in 26 of the 56 patients assessed. Orocecal
ically for symptoms associated with rumination; on transit at 6 hours, a surrogate marker for small bowel
average, these patients received 1.8 ⫾ 0.1 types of transit, was assessed in 28 patients and was delayed
medications. Of note, no patients had significant in 13 patients. However, 7 of the 13 patients with
symptomatic improvement with medical therapy. delayed orocecal transit also had delayed gastric
Sixty-six (45.0%) patients had been treated with acid- emptying at 4 hours. Scintigraphic colonic transit
blocking medications (histamine [H2] blockers was generally performed in patients with constipa-
and/or proton pump inhibitors) and 56 (38.1%) with tion or diarrhea. The method and normal values
prokinetic medications (cisapride, metoclopramide, have been described elsewhere.6 Colonic transit was
or erythromycin). Forty-three (29.3%) patients re- abnormally slow in 5, and accelerated in 1 of the 12
ceived both acid-blocking and prokinetic medica- patients tested.
tions. Twelve (8.2%) patients had been treated with Esophageal pH testing was performed in 24 pa-
anti-emetic medications, 10 (6.8%) with antidepres- tients; results were normal in 45.8%, and showed
sants, and 3 (2.0%) with narcotic pain medications. reflux/regurgitation in 54.2%. None of the patients
Before evaluation at Mayo Clinic Rochester, 5 tested had significant nocturnal or supine reflux, and
(3.4%) patients had required supplemental enteral the majority with reflux/regurgitation had numer-
(via tube) or parenteral nutrition for management of ous, brief postprandial episodes. Regurgitation/
symptoms, dehydration, or nutritional support. reflux occurred ⬎200 times after a single meal in 1
patient. Gastroduodenal manometry was performed
School/Work Absenteeism in 65 patients; 55.4% were normal, 40% had rumina-
Of the 44 patients with data available, 32 (72.7%) tion-waves, and 4.6% had antral hypomotility. Re-
had missed school or work because of symptoms. In gurgitation/vomiting during the gastroduodenal
these 44 patients, rates of school/work absenteeism manometry assessment occurred only in patients
were not significantly higher in patients with abdom- with rumination-waves on manometry. Esophageal
inal pain; 83.3% of patients with abdominal pain had manometry testing performed using the station-pull-
missed school/work compared with 60.0% of pa- through technique at the conclusion of the gastrodu-
tients without abdominal pain (P ⫽ .2). odenal manometry assessment was normal in all but
2 patients, 1 of whom had evidence of a hypertensive
Hospitalization lower esophageal sphincter and the other a hypo-
Of the 76 patients with data on hospitalizations tonic lower esophageal sphincter.
available in the medical record, 35 (46.1%) had been The average number of the above diagnostic tests
hospitalized for evaluation of symptoms or treat- performed per patient was 3.3 ⫾ 0.2 (median: 3;
ment of complications related to rumination. Patients range: 0 – 8).

160 RUMINATION SYNDROME IN CHILDREN AND ADOLESCENTS


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Treatment and Outcomes nosing this important medical condition. Formal
Behavioral treatment was recommended for 127 of diagnostic criteria for rumination syndrome occur-
the 147 patients, and was undertaken at Mayo Clinic ring in children and adolescents with onset beyond
Rochester in 71 patients. Medical treatment was rec- infancy have not been defined in the consensus cri-
ommended for 25 of the 147 patients including: H2- teria.1
blocker in 9, proton pump inhibitor in 3, prokinetic In the current study, gastric emptying was delayed
medications (cisapride, erythromycin, or metoclo- in 46% of patients assessed. However, there are a
pramide) in 11, tricyclic antidepressants in 3, hyoscy- number of caveats in the interpretation of scinti-
amine in 1, and anti-emetic medication in 1. None of graphic gastric emptying tests in children and ado-
the patients were treated with supplemental enteral lescents with rumination syndrome. Delayed gastric
(via tube) or parenteral nutrition. emptying may result from regurgitation and reswal-
Outcome data were available for 54 (36.7%) pa- lowing of food, which is then delivered intermit-
tients. The mean duration of follow-up was 10.2 ⫾ tently or later to the distal stomach for digestion and
1.4 months. The baseline characteristics and clinical mixing. Another caveat in the interpretation of the
features of rumination for these 54 patients were scintigraphic gastric emptying test is the potential for
similar to those of patients without outcome data expulsion of the radiolabeled meal; when the major-
available. The only significant differences between ity of the test meal is expelled, the study cannot be
the 2 groups were a slightly older age at diagnosis completed. Although an assessment of gastric emp-
(15.8 ⫾ 0.5 vs 14.5 ⫾ 0.4 years; P ⫽ .02) and a higher tying may help to exclude a significant stomach dys-
prevalence of surgeries before diagnosis of rumina- motility and a normal result may reassure the patient
tion syndrome (19.2% vs 5.6%; P ⫽ .02) for patients and family, abnormal results should be interpreted
with outcome data available. Therefore, the outcome carefully in the setting of rumination.
data are likely representative of the group as a In contrast to the relatively common observation of
whole. delayed gastric emptying in the patient population
Fifty-two of the 54 patients with outcome data reported, antral hypomotility was extremely rare on
available had received behavioral treatment consist- formal testing with manometry. Although gastrodu-
ing of biofeedback, relaxation training, instruction in odenal manometry has been advocated as a diagnos-
diaphragmatic breathing, and/or cognitive behav- tic test in the setting of rumination,7 only 40% of the
ioral therapy. Of the 46 patients in this group who 65 patients assessed in our study had characteristic
underwent behavioral treatment at Mayo Clinic R-waves or simultaneous pressure spikes across all
Rochester, the number of treatment sessions with a sensors, suggesting an extraintestinal stimulus and
behavioral medicine professional was 2.7 ⫾ 0.5 (me- contraction of the abdominal musculature. In addi-
dian: 1; range: 1–19). Overall, symptoms resolved in tion, gastroduodenal manometry is an invasive test
16 (29.6%), and improved in 30 (55.5%); therefore, a requiring significant technical and interpretive ex-
positive impact on symptoms was noted in 85.1%. In pertise, and is only available in tertiary care centers.
12.9%, no improvement in symptoms was noted at In general, we do not advocate esophageal pH
the time of follow-up. The 2 patients who did not testing in patients with rumination, because quanti-
receive formal behavioral therapy consultation re- tatively abnormal ‘reflux’ is a consequence rather
ceived reassurance and an explanation of the nature than a cause of the symptoms. When prolonged pH
of their condition, and were noted to have improve- testing is performed for suspected gastroesophageal
ment of symptoms at follow-up. One patient who reflux disease, rumination syndrome is characterized
also suffered from Bartter’s syndrome died during by the predominance of postprandial regurgitation
the course of follow-up. of acid, rather than supine or nocturnal reflux, both
Patients with symptomatic improvement had sig- of which occur in classical gastroesophageal reflux
nificantly higher body mass indices at the time of disease.8,9
diagnosis than those whose symptoms remained un- Abnormal relaxation of the lower esophageal
changed (body mass index: 21.9 kg/m2 vs. 19.0 kg/ sphincter, a common cause of classical gastroesoph-
m2; n ⫽ 46; P ⫽ .02). No other baseline characteristics ageal reflux disease in pediatric patients,10 is also
or clinical features had a significant impact on out- involved in the process of rumination. Thurmshirn et
come. al11 showed the mechanism of rumination involves
relaxation of the lower esophageal sphincter in re-
sponse to lower pressures in the fundus, and in-
DISCUSSION creased gastric sensitivity. In healthy adults, there is
In adults, the diagnosis of rumination syndrome is active contraction at the esophagogastric junction
usually based on eliciting classical symptoms in the and increased lower esophageal sphincter pressure
absence of structural disease.3 Unfortunately, our ex- during periods of increased intraabdominal pres-
perience at a tertiary care center suggests that pedi- sure;12 tonic contraction of the crural diaphragm is
atric and adolescent patients with rumination syn- the proposed mechanism for this response. This
drome often undergo extensive, costly, and invasive mechanism is altered in the setting of rumination
testing, and are frequently misdiagnosed as having syndrome as transient lower esophageal sphincter
gastroesophageal reflux disease or gastroparesis. We relaxations occur after abdominal straining events.13
believe that insufficient awareness of the clinical fea- In addition to postprandial regurgitation, many
tures of rumination syndrome in pediatric and ado- patients in our study had other gastrointestinal
lescent patients contributes to the difficulty in diag- symptoms including abdominal pain (38.1%), consti-

ARTICLES 161
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pation (21.1%), and nausea (17.0%). The presence of TABLE 1. Proposed Criteria for Rumination Syndrome in
additional gastrointestinal symptoms may induce Children and Adolescents
physicians to recommend more extensive diagnostic At least 6 wk, which may not be consecutive, in the previous
testing. However, our study demonstrates that such 12 mo of recurrent regurgitation of recently ingested food
testing is not helpful in the presence of typical clin- which:
1. begins within 30 min of meal ingestion
ical features of rumination. 2. is associated with either reswallowing or expulsion of food
Weight loss was also commonly associated with 3. stops within 90 min of onset or when regurgitant becomes
rumination, despite normal body mass indices at the acidic
time of diagnosis. Considering the female predomi- 4. is not associated with mechanical obstruction
5. does not respond to standard treatment for
nance of the condition and the frequent occurrence of gastroesophageal reflux disease (ie, medical therapy or
weight loss, classical eating disorders such as an- lifestyle modification measures)
orexia nervosa and bulimia nervosa should be con- 6. is not associated with nocturnal symptoms
sidered in the differential diagnosis. However, in our
study population, eating disorders had been diag- vorable at our institution (⬎80% success) that a for-
nosed in only 3.4% of patients. Previous studies have mal, controlled clinical trial has not been pursued.
described a history of eating disorders (primarily Collaboration between gastroenterologists, pediatri-
bulimia nervosa) in a larger proportion of adult pa- cians, and psychologists in addition to educating
tients with rumination syndrome.14 –16 Although patients and family members are key elements to a
weight loss is a concerning symptom in children and successful outcome.
adolescents, we do not believe that weight loss is an
indication for more exhaustive diagnostic testing in ACKNOWLEDGMENTS
the presence of classical clinical features of rumina- This work was supported in part by grants R01-DK54681 and
tion syndrome. K24-DK02638 (to Dr Camilleri) and by General Clinical Research
In general, rumination syndrome is a “benign” Center grant (#RR00585) from the National Institutes of Health.
We thank Cindy Stanislav for excellent secretarial assistance.
condition.17 However, our study demonstrates sig-
nificant functional disability related to weight loss, REFERENCES
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162 RUMINATION SYNDROME IN CHILDREN AND ADOLESCENTS


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Rumination Syndrome in Children and Adolescents: Diagnosis, Treatment, and
Prognosis
Heather J. Chial, Michael Camilleri, Donald E. Williams, Kristi Litzinger and Jean
Perrault
Pediatrics 2003;111;158
DOI: 10.1542/peds.111.1.158
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PEDIATRICS is the official journal of the American Academy of Pediatrics. A monthly


publication, it has been published continuously since 1948. PEDIATRICS is owned, published,
and trademarked by the American Academy of Pediatrics, 141 Northwest Point Boulevard, Elk
Grove Village, Illinois, 60007. Copyright © 2003 by the American Academy of Pediatrics. All
rights reserved. Print ISSN: 0031-4005. Online ISSN: 1098-4275.

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Rumination Syndrome in Children and Adolescents: Diagnosis, Treatment, and
Prognosis
Heather J. Chial, Michael Camilleri, Donald E. Williams, Kristi Litzinger and Jean
Perrault
Pediatrics 2003;111;158
DOI: 10.1542/peds.111.1.158

The online version of this article, along with updated information and services, is
located on the World Wide Web at:
/content/111/1/158.full.html

PEDIATRICS is the official journal of the American Academy of Pediatrics. A monthly


publication, it has been published continuously since 1948. PEDIATRICS is owned,
published, and trademarked by the American Academy of Pediatrics, 141 Northwest Point
Boulevard, Elk Grove Village, Illinois, 60007. Copyright © 2003 by the American Academy
of Pediatrics. All rights reserved. Print ISSN: 0031-4005. Online ISSN: 1098-4275.

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