Karimzada MM, Matthews MN, French SW, DeUgarte D, Kim DY. Langerhans cell histiocytosis masquerading as acute appendicitis: Case report and review. World J Gastrointest Endosc 2017; 9(3): 139-144 [PMID: 28360976 DOI: 10.4253/wjge.v9.i3.139]
Corresponding Author of This Article
Dennis Y Kim, Assistant Professor of Surgery, Department of Surgery, Harbor-UCLA Medical Center, 1000 West Carson Street, Torrance, CA 90502, United States. dekim@dhs.lacounty.gov
Research Domain of This Article
Surgery
Article-Type of This Article
Case Report
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This article is an open-access article which was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/
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Karimzada MM, Matthews MN, French SW, DeUgarte D, Kim DY. Langerhans cell histiocytosis masquerading as acute appendicitis: Case report and review. World J Gastrointest Endosc 2017; 9(3): 139-144 [PMID: 28360976 DOI: 10.4253/wjge.v9.i3.139]
World J Gastrointest Endosc. Mar 16, 2017; 9(3): 139-144 Published online Mar 16, 2017. doi: 10.4253/wjge.v9.i3.139
Langerhans cell histiocytosis masquerading as acute appendicitis: Case report and review
Mohammad M Karimzada, Michele N Matthews, Samuel W French, Daniel DeUgarte, Dennis Y Kim
Mohammad M Karimzada, Daniel DeUgarte, Dennis Y Kim, Department of Surgery, Harbor-UCLA Medical Center, Torrance, CA 90502, United States
Michele N Matthews, Samuel W French, Department of Pathology, Harbor-UCLA Medical Center, Torrance, CA 90053, United States
Author contributions: Karimzada MM, Matthews MN, French SW, DeUgarte D and Kim DY designed the case study and review; Matthews MN and French SW performed microscopic and immunohistochemical evaluations of surgical specimens; DeUgarte D provided descriptive impressions of differential diagnosis and intraoperative findings; Karimzada MM and Matthews MN wrote the paper; final revision approved by Kim DY.
Institutional review board statement: Case study was prepared at Harbor-UCLA Medical Center in affiliation with the Institutional Review Board at the LA BioMed Research Institute in Torrance, CA.
Informed consent statement: Patient consented to use of medical information pertaining to this case for teaching and research purposes.
Conflict-of-interest statement: The authors have no conflicts of interest to disclose regarding this case report and review.
Open-Access: This article is an open-access article which was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/
Correspondence to: Dennis Y Kim, Assistant Professor of Surgery, Department of Surgery, Harbor-UCLA Medical Center, 1000 West Carson Street, Torrance, CA 90502, United States. dekim@dhs.lacounty.gov
Telephone: +1-310-2222700 Fax: +1-310-5331841
Received: April 29, 2016 Peer-review started: May 3, 2016 First decision: May 17, 2016 Revised: December 6, 2016 Accepted: December 27, 2016 Article in press: December 28, 2016 Published online: March 16, 2017 Processing time: 319 Days and 21.8 Hours
Abstract
Langerhans cell histiocytosis (LCH) is a rare syndrome characterized by unifocal, multifocal unisystem, or disseminated/multi-system disease that commonly involves the bone, skin, lymph nodes, pituitary, or sometimes lung (almost exclusively in smokers) causing a variety of symptoms from rashes and bone lesions to diabetes insipidus or pulmonary infiltrates. We present a previously unreported case of gastrointestinal LCH as well as a novel characteristic lesion affecting the colon of a young woman who presented with signs and symptoms mimicking acute on chronic appendicitis. Immunohistochemical analysis of appendectomy specimen and nodular specimens on colonoscopy demonstrated S-100, CD1a, and langerin reactivity. The patient underwent systemic chemotherapy with cytarabine and demonstrated excellent response to therapy.
Core tip: Langerhans cell histiocytosis (LCH) of the adult gastrointestinal tract can affect the appendix and present as mild inflammatory findings on exploratory laparascopy, or white-yellowish nodularities and polyps on lower endoscopy. Immunohistochemical staining of surgical specimens for CD1a, S-100, and langerin should be considered in gastrointestinal lesions that demonstrate histology concerning for histiocytosis in an attempt to decrease morbidity related to undiagnosed LCH.
