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Multilocus Heterotopic Gastric Mucosa of Ileum Masquerading As VEOIBD in Anewborn Livia Lindoso, MD,A Cortney R

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Multilocus Heterotopic Gastric Mucosa of Ileum Masquerading As VEOIBD in Anewborn Livia Lindoso, MD,A Cortney R Multilocus Heterotopic Gastric Mucosa of Ileum Masquerading as VEOIBD in aNewborn Livia Lindoso, MD,a Cortney R. Ballengee, MD,a Kiran P. Patel, MD,b,c Rene Romero, MD,a,c Shelley Caltharp, MD,c,d Adina L. Alazraki, MD,c,e Subra Kugathasan, MDa,c Heterotopic gastric mucosa (HGM) is defined as the presence of gastric abstract mucosa outside of the stomach, which is documented by histologic finding. HGM is typically a solitary lesion; however, in our Case Report, the patient presented with multilocus HGM, an uncommon form in which the small bowel is extensively involved. We report a unique case of multilocus HGM mimicking very early–onset inflammatory bowel disease with recurrent gastrointestinal bleeding, chronic inflammation, and stricturing in a newborn patient. Divisions of aGastroenterology and bAllergy and d fi Immunology, Departments of Pediatrics, Pathology and Histologic ndings from the ileocecal specimen revealed multiple ulcers Laboratory Medicine, and eRadiology and Imaging Science, surrounded by chronic inflammation. Subsequently, a Technetium-99m School of Medicine, Emory University, Atlanta, Georgia; and c ’ pertechnetate scan demonstrated an increased tracer uptake in the remaining Childrens Healthcare of Atlanta, Atlanta, Georgia ileum. This radiologic finding, in combination with the discovery of gastric Dr Lindoso reviewed the patient chart and drafted mucosa within the remainder of resected ileal specimen, led to the diagnosis the initial manuscript; Dr Caltharp did the histological analysis of the specimens and revised of HGM. Omeprazole was initiated, and the patient is now asymptomatic the manuscript; Dr Alazraki did the analysis of without further gastrointestinal bleeding. Increased awareness of this rare the magnetic resonance enterography and disease and performing a Technetium-99m pertechnetate early can correctly Technetium-99m nuclear scan and revised the manuscript; Drs Ballengee, Patel, Romero, and diagnose HGM and prevent disease complication. Kugathasan critically revised the draft and initial manuscript; and all authors approved the final manuscript as submitted. DOI: https://doi.org/10.1542/peds.2018-2398 Heterotopic gastric mucosa (HGM) is the We report a unique case of multilocus Accepted for publication Jan 11, 2019 presence of gastric mucosa outside of HGM mimicking very early–onset fl Address correspondence to Subra Kugathasan, MD, the stomach documented by histologic in ammatory bowel disease (VEOIBD) Division of Pediatric Gastroenterology, Department of findings. Literature suggests that HGM can in a newborn with recurrent Pediatrics, Emory University School of Medicine, 1760 occur throughout various locations in the gastrointestinal bleeding, chronic gut Haygood Dr, W427, Atlanta, GA 30322. E-mail: body, including the esophagus, duodenum, inflammation, and stricturing. This Case [email protected] ileum, rectum, gallbladder, and (rarely) Report is a reminder to clinicians to PEDIATRICS (ISSN Numbers: Print, 0031-4005; Online, 1098-4275). outside the gastrointestinal tract in the consider HGM in the differential mediastinum, scrotum, urinary bladder, diagnosis of rectal bleeding in Copyright © 2019 by the American Academy of Pediatrics airways, and spinal cord.1 The origin of newborns. An early Technetium-99m nuclear scan would have led to a firm FINANCIAL DISCLOSURE: The authors have indicated HGM can be congenital or acquired during diagnosis of HGM and avoided the they have no financial relationships relevant to this the progress of repair of damaged mucosa, costly workup for VEOIBD and possibly article to disclose. although it is typically confined to 1 area.2,3 shortened the length of total parenteral FUNDING: No external funding. It commonly occurs in conjunction with nutrition (TPN) in our case. POTENTIAL CONFLICT OF INTEREST: The authors have other congenital malformations, including indicated they have no potential conflicts of interest Meckel diverticulum and gastrointestinal to disclose. tract duplication. The clinical presentation CASE PRESENTATION of HGM may vary depending on size and A premature boy born at 33 weeks’ To cite: Lindoso L, Ballengee CR, Patel KP, et al. location but typically includes abdominal gestation presented with hematochezia, Multilocus Heterotopic Gastric Mucosa of Ileum pain, bleeding, perforation, ulceration, and diarrhea, anemia, severe Masquerading as VEOIBD in a Newborn. Pediatrics. 2019;143(4):e20182398 intussusception. hypoalbuminemia, and partial bowel Downloaded from www.aappublications.org/news by guest on October 2, 2021 PEDIATRICS Volume 143, number 4, April 2019:e20182398 CASE REPORT obstruction during the first week associated with VEOIBD (including had not previously been observed in of life. He was initially presumed to IL10R and TTC7a) did not reveal any the previous 2 resections (Fig 3). have necrotizing enterocolitis and known pathogenic variants. Although the patient had 2 previous therefore treated with bowel rest and Furthermore, an extensive workup ileal resections and several antibiotics. Recovery was slow, and for immunodeficiency and infection colonoscopies with biopsies, this was the patient continued to show partial (including dihydrorhodamine, the first time HGM was found. The bowel obstruction despite antibiotic X-linked inhibitor of apoptosis patient subsequently underwent treatment. Ultimately, he required protein, T-regulatory panel, a Technetium-99m nuclear scan, prolonged TPN because of the antienterocyte antibody, lymphocyte which confirmed the presence of extended period of bowel rest. An enumeration panel, serum multilocus gastric mucosa in the exploratory laparotomy was immunoglobulins, and remaining ileal loops, which was performed at 4 weeks of age because cytomegalovirus polymerase chain evident by the increased tracer of the continued rectal bleeding and reaction in blood and urine) was uptake (Fig 4). Because of the bowel obstruction. The surgical unremarkable. The patient’s clinical presence of multilocus HGM, the specimen revealed severe thickening course lacked symptoms suggestive patient was initiated on high-dose of the ileum as well as a stricture. of systemic inflammation, multiorgan proton-pump inhibitor (PPI) therapy The patient consequently underwent autoimmunity, recurrent or severe (omeprazole, 20 mg twice per day), ileocecal resection with ileostomy infection, or perianal fistulas, any of and eventually, he was advanced to and colonic mucous fistula. Histology which would be consistent with full enteral nutrition without further revealed chronic organizing and VEOIBD. The patient failed multiple need for TPN by the age of 24 perforating ulcerations surrounded medical therapies, including months. Because of the significant by chronic inflammation. Despite the corticosteroids and azathioprine, and ileal loss and the risk for vitamin B12 resection, the patient continued to continued to have intermittent rectal deficiency and bile salt–induced have an ileal obstruction and bleeding, poor enteral tolerance, and diarrhea, the patient was started on underwent repeat ileal segmental poor growth. The patient continued regular vitamin B12 injections and resection secondary to recurrent to be dependent on TPN at 18 months cholestyramine. The patient has been stricture 7 weeks after the initial first of age because enteral nutrition doing well with improved growth procedure. Interestingly, the advancement was repeatedly delayed (height 10th percentile; weight 25th pathology report from the specimen by intermittent small-bowel percentile) while on omeprazole with revealed crypt abscesses and obstruction. no signs of gastrointestinal bleeding ulceration (Fig 1), which was at the age of 30 months. consistent with VEOIBD. A A magnetic resonance enterography colonoscopy was performed and (MRE) was performed and revealed DISCUSSION revealed macroscopic chronic yet another ileal stricture (Fig 2). The inflammation of the ileum. A patient underwent a third ileal The most common causes of lower commercially available targeted gene segmental resection given the MRI gastrointestinal bleeding in the panel (MM160 Inflammatory Bowel results and symptoms of bowel neonatal and infantile period are Disease: Sequencing Panel; EGL obstruction. During this operation, Meckel diverticulum, polyps, clotting Genetics, Tucker, GA), which scattered HGM was noted throughout disorders, arteriovenous fistulas, and sequences 26 genes known to be the affected segment of ileum, which (uncommonly) VEOIBD.3,4 Although FIGURE 1 Crypt abscesses and ulceration. A, Stricture with circumferential mucosal ulceration (arrows) with focal fissuring ulceration (rectangular) to the muscularis propria. No residual crypts are seen. B, Adjacent acute cryptitis (arrows). C, Crypt abscesses. Downloaded from www.aappublications.org/news by guest on October 2, 2021 2 LINDOSO et al mucosa outside of the stomach confirmed by histology (Fig 3). These histologic findings can be divided into type I, consisting of gastric glands and foveolar epithelium, and type II, consisting of only foveolar epithelium.22 Type I is thought to have a congenital origin, whereas type II may have originated from metaplastic processes.22 Other histologic findings associated with HGM are foveolar hyperplasia and mild lymphocytic infiltration.22 In our FIGURE 4 case, the patient presented with type Technetium-99m pertechnetate Meckel scintig- I, which according to the literature is FIGURE 2 raphy. Shown is a linear increased focus of the most common histologic T2 fat-suppressed coronal image from MRE radiotracer
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