Literature DB >> 26823541

Concurrent Ulcerative Colitis and Neurofibromatosis Type 1: The Question of a Common Pathway.

William Adams1, Lisa Mitchell2, Roberto Candelaria-Santiago3, Jody Hefner2, Joseph Gramling2.   

Abstract

Patients with neurofibromatosis type 1 (NF1) are prone to the development of gastrointestinal stromal tumors, which may present clinically with hematochezia, obstruction, or abdominal pain. These symptoms are also commonly associated with the presentation of ulcerative colitis (UC). Within the past 5 years, there have been 2 reports of concurrent NF1 and UC and a common pathophysiologic pathway involving mast cells has been postulated. We present the case of a 15-year-old boy with a known history of NF1 who presented with 3 months of hematochezia and loose stools. A colonoscopy revealed pancolitis and histology demonstrating acute cryptitis, focal crypt abscesses, and architectural distortion consistent with UC. Due to the paucity of reported cases, the findings of both diseases in the same individual could reasonably be discounted as coincidence. However, in light of increasing reports of concurrent NF1 and UC, advances in characterizing the microenvironment within neurofibromas, and recent findings regarding potential shared genetic susceptibility, it is increasingly possible that the proposed common pathway is accurate. Our case adds to the literature and underscores the need for further investigation.
Copyright © 2016 by the American Academy of Pediatrics.

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Year:  2016        PMID: 26823541     DOI: 10.1542/peds.2015-0973

Source DB:  PubMed          Journal:  Pediatrics        ISSN: 0031-4005            Impact factor:   7.124


  2 in total

1.  Inflammatory Bowel Disease and Primary Sclerosing Cholangitis in a Pediatric Patient With Neurofibromatosis Type 1.

Authors:  Emily Lehan; Tao Wang; Don Soboleski; Amy Acker; Mohit Kehar
Journal:  ACG Case Rep J       Date:  2021-05-14

2.  Mendelian Disease Associations Reveal Novel Insights into Inflammatory Bowel Disease.

Authors:  Lichy Han; Mateusz Maciejewski; Christoph Brockel; Lovisa Afzelius; Russ B Altman
Journal:  Inflamm Bowel Dis       Date:  2018-02-15       Impact factor: 5.325

  2 in total

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