Duodenal obstruction: A rare complication of langerhans cell histiocytosis

Pediatric Hematology Oncology Journal Pub Date : 2024-06-24 DOI:10.1016/j.phoj.2024.06.005

Vaishnavi Sreenivasan , Anmol Aatli , Saahiti Andhavaram , Apoorva Sharma , Rashmi Dandriyal , Shyam S. Meena , Nidhi Sugandhi , Sumit Mehndiratta , Nidhi Chopra , Amitabh Singh

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Abstract

Background

Langerhans cell histiocytosis (LCH) has a variable presentation. Gastrointestinal involvement is rare and portends a poor prognosis in LCH.

Case report

Herein, we describe the presentation, progression, management, and outcome of an 11-month-old female infant with LCH. The patient presented with severe acute malnutrition, hepatosplenomegaly, and gastrointestinal involvement in the form of persistent vomiting. She was managed with chemotherapy initially and subsequently developed complete duodenal narrowing. She underwent Kimura's duodenojejunostomy and improved. Hence, chemotherapy was restarted. Two months later, she developed features of intestinal obstruction with intraoperative findings of ileal gangrene requiring adhesiolysis, ileal resection, and ileostomy. Subsequently, she succumbed to sepsis.

Conclusion

This case report aims to highlight the atypical gastrointestinal manifestations of LCH and the challenges faced in managing such a case.

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十二指肠梗阻：朗格汉斯细胞组织细胞增生症的罕见并发症

背景朗格汉斯细胞组织细胞增生症（LCH）的表现多种多样。病例报告在此，我们描述了一名11个月大的朗格汉斯细胞组织细胞增生症女婴的表现、病情发展、处理和预后。患者表现为严重急性营养不良、肝脾肿大和持续呕吐的胃肠道受累。她最初接受了化疗，随后出现十二指肠完全狭窄。她接受了木村十二指肠空肠吻合术，病情有所好转。因此，化疗重新开始。两个月后，她出现肠梗阻症状，术中发现回肠坏疽，需要进行粘连溶解、回肠切除和回肠造口术。结论本病例报告旨在强调 LCH 的非典型胃肠道表现以及处理此类病例所面临的挑战。

本文章由计算机程序翻译，如有差异，请以英文原文为准。

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来源期刊

Pediatric Hematology Oncology Journal

CiteScore

1.00

自引率

0.00%

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