Kellermayer R, Tatevian N, Klish W, Shulman RJ. Steroid responsive eosinophilic gastric outlet obstruction in a child. World J Gastroenterol 2008; 14(14): 2270-2271 [PMID: 18407608 DOI: 10.3748/wjg.14.2270]
Corresponding Author of This Article
Richard Kellermayer, MD, PhD, Section of Gastroenterology, Hepatology and Nutrition, Department of Pediatrics, Baylor College of Medicine, 6621 Fannin St., CC1010.00, Houston, TX 77030-2399, United States. kellerma@bcm.tmc.edu
Article-Type of This Article
Case Report
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Richard Kellermayer, William Klish, Robert J Shulman, Section of Gastroenterology, Hepatology and Nutrition, Department of Pediatrics, Baylor College of Medicine, One Baylor Plaza, Houston, TX 77030, United States
Nina Tatevian, Department of Pathology, Baylor College of Medicine, One Baylor Plaza, Houston, TX 77030, United States
Robert J Shulman, Children’s Nutrition Research Center, Department of Pediatrics, Baylor College of Medicine, One Baylor Plaza, Houston, TX 77030, United States
ORCID number: $[AuthorORCIDs]
Author contributions: Kellermayer R managed the patient and prepared the manuscript; Klish W and Shulman RJ managed the patient and revised the manuscript; Tatevian N did the pathological studies and prepared the figures.
Correspondence to: Richard Kellermayer, MD, PhD, Section of Gastroenterology, Hepatology and Nutrition, Department of Pediatrics, Baylor College of Medicine, 6621 Fannin St., CC1010.00, Houston, TX 77030-2399, United States. kellerma@bcm.tmc.edu
Telephone: +1-83-28-223616
Fax: +1-83-28-253633
Received: September 7, 2007 Revised: December 14, 2007 Published online: April 14, 2008
Abstract
Gastric outlet obstruction is a rare complication of eosinophilic gastroenteritis, most commonly treated surgically. We report a case of eosinophilic gastric outlet obstruction in a child that responded to conservative medical management. A brief review of this clinical entity is also provided.
Eosinophilic gastroenteritis or gastroenteropathy (EG) is defined by variable gastrointestinal symptoms and eosinophilic infiltration of one or more areas of the gastrointestinal tract, without evidence of parasitic or extra-intestinal disease[1]. It can be idiopathic, related to food allergies, infections, and rarely infantile inflammatory bowel disease[2]. A very rare complication of this entity is distal gastritis leading to gastric outlet obstruction that has been reported to occur in infancy accompanying, mimicking or generating hypertrophic pyloric stenosis[34] and in adults[56], but has not been described in children as an isolated manifestation of EG. Most commonly patients with eosinophilic gastric outlet obstruction have been treated surgically[357] except for a few infantile cases where protein hydrolysate formula[2] or steroid therapy[48] has been used. Resolution of symptoms with steroid treatment has recently been demonstrated in an adult case also[6]. We report a 2 and half-year-old Caucasian girl with eosinophilic gastric outlet obstruction treated successfully with steroids.
CASE REPORT
The previously healthy girl presented after a 3-mo history of worsening postprandial emesis leading to an inability to tolerate feedings. She had no history of atopy or diet change. She had 0.08 eosinophils (740 × 106 /L) on peripheral blood count, but was not anemic nor hypoalbuminemic. IgE level was normal. Upper gastrointestinal imaging showed marked pyloric narrowing (Figure 1). Endoscopy revealed antral edema and severe pyloric stenosis through which a Pentax 2470 endoscope (8.0 mm diameter) could not be passed. Biopsies from this area were consistent with eosinophilic gastritis (Figure 2A). Methylprednisolone (2 mg/kg per day) was started and she began tolerating liquids within two days. Endoscopy five days later revealed decreased pyloric swelling and the endoscope could be passed through the pylorus. No pyloric ulceration was seen and the duodenum appeared normal. Antral and pyloric mucosal biopsies showed resolution of the eosinophilic cellular infiltrate (Figure 2B). She was advanced to a low roughage diet within a few days, switched to oral prednisolone therapy (0.5 mg/kg per day) and discharged home. The steroids were weaned and discontinued after 8 weeks of treatment. She remained symptom free six months following the cessation of steroids.
Figure 2 Histologic images before and 5 d after steroid therapy.
A: Peripyloric antral sections showed prominent eosinophilic infiltration of the lamina propria (up to 30 eosinophils per single high power field), with occasional degranulation (arrow) of eosinophilic content and infiltration of the muscularis mucosae; B: Biopsies 5 d after intravenous steroid therapy demonstrated only a few eosinophils with a peak count of 2 eosinophils per high power field (HE, × 40).
DISCUSSION
Reported clinical manifestations of eosinophilic gastroenteritis include obstruction at various levels of the gastrointestinal tract, growth failure, weight loss, anemia, melena, diarrhea, protein loosing enteropathy, abdominal pain, pseudo-Crohn’s disease, esophagitis, and eosinophilic ascites[9]. On rare instances EG can even be complicated by gastrointestinal perforation[10]. While eosinophilic inflammation leading to pyloric stenosis and gastric outlet obstruction has been reported in adults and infants, it has not been described in children (pediatric patients more than 2 years of age) as a localized manifestation of EG to our knowledge. We could only identify one earlier case of antral web related gastric outlet obstruction that was complicated by eosinophilic inflammation in a 3-years-old child[11]. Our patient responded briskly to steroid therapy and has remained asymptomatic for more than six months off therapy. Similar clinical response has been recorded earlier in infants with the same condition[48] and very recently in an adult[6]. However, in several instances resolution of the eosinophilic inflammation can be protracted and the course of the disease may wax and wane[410]. Nevertheless, we conclude that steroid therapy should be considered in cases of eosinophilic gastric outlet obstruction prior to surgical interventions in all age groups.
Footnotes
Peer reviewer: Luigi Bonavina, Professor, Department of
Surgery, Policlinico San Donato, University of Milano, via
Morandi 30, Milano 20097, Italy
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