Copyright
©The Author(s) 2015. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Gastroenterol. Jun 14, 2015; 21(22): 7059-7064
Published online Jun 14, 2015. doi: 10.3748/wjg.v21.i22.7059
Published online Jun 14, 2015. doi: 10.3748/wjg.v21.i22.7059
Abnormal layering of muscularis propria as a cause of chronic intestinal pseudo-obstruction: A case report and literature review
Napat Angkathunyakul, Department of Pathology, Ramathibodi Hospital, Mahidol University, Bangkok 10400, Thailand
Suporn Treepongkaruna, Department of Pediatrics, Ramathibodi Hospital, Mahidol University, Bangkok 10400, Thailand
Sani Molagool, Department of Surgery, Ramathibodi Hospital, Mahidol University, Bangkok 10400, Thailand
Nichanan Ruangwattanapaisarn, Department of Diagnostic and Therapeutic Radiology, Ramathibodi Hospital, Mahidol University, Bangkok 10400, Thailand
Author contributions: Angkathunyakul N designed the report, interpreted histologic findings, and performed staining and immunohistochemistry studies; Treepongkaruna S and Molagool S collected the patient’s clinical data; Ruangwattanapaisarn N provided the radiographic data; Angkathunyakul N and Treepongkaruna S reviewed and analyzed the literature and wrote the paper.
Supported by Faculty of Medicine, Ramathibodi Hospital, Mahidol University, Thailand.
Ethics approval: The study was reviewed and approved by the Committee on Human Rights Related to Research Involving Human Subjects, based on the Declaration of Helsinki.
Informed consent: The legal guardian of the study participant provided informed written consent prior to study enrollment.
Conflict-of-interest: The authors have no conflicts of interest to declare.
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: Napat Angkathunyakul, MD, Pathologist, Department of Pathology, Ramathibodi Hospital, Mahidol University, Bangkok 10400, Thailand. napat.ang@mahidol.ac.th
Telephone: +66-2-2011667 Fax: +66-2-3547266
Received: January 7, 2015
Peer-review started: January 8, 2015
First decision: January 22, 2015
Revised: March 9, 2015
Accepted: March 30, 2015
Article in press: March 31, 2015
Published online: June 14, 2015
Peer-review started: January 8, 2015
First decision: January 22, 2015
Revised: March 9, 2015
Accepted: March 30, 2015
Article in press: March 31, 2015
Published online: June 14, 2015
Core Tip
Core tip: This report describes the case of a 9-mo-old boy who presented with chronic intestinal pseudo-obstruction. Full-thickness small bowel biopsy showed abnormal layering of muscularis propria (additional oblique layer) and serosal aberrant muscularization. There have been only eight previously reported abnormal layering cases and only one case with an additional oblique layer. The patient also had a short small bowel without malrotation, brachydactyly, and absence of the 2nd to 4th middle phalanges of both hands. The patient showed clinical improvement with medical treatment and nutritional support.