Korean J Gastroenterol.  2022 Sep;80(3):149-153. 10.4166/kjg.2022.047.

Langerhans Cell Histiocytosis with the Synchronous Invasion of Stomach and Colon in an Adult Patient: A Case Report

• Kim SJ
• Hah SI
• Kwak JY
• Choi JW
• Cho HC
• Ha CY
• Jung WT
• Lee OJ
• Lee CM

• KMID: 2533318
• DOI: http://doi.org/10.4166/kjg.2022.047

Abstract

Langerhans cell histiocytosis (LCH) is reported less frequently in adults than in children. The most common site of involvement in adults is the bone, accounting for 30-50% of cases. The gastrointestinal tract is very rare, accounting for approximately 2%. We present a case of Langerhans cell histiocytosis that simultaneously invaded multiple organs, including the stomach and colon, in an adult. A 37-year-old woman with no underlying disease complained of chest discomfort and a palpable right submandibular mass. A right Level II neck mass and mediastinal LN enlargement were confirmed on the pharynx and chest CT scan. Multiple subepithelial masses with central ulceration and erosion were observed in the corpus and fundus on the esophagogastroduodenoscopy and in the right colon on the colonoscopy. The histopathology findings were the same in each tissue biopsied from the stomach, colon, and right neck lymph nodes. Langerhans cells with classical reniform nuclei and prominent eosinophils invaded the normal glands, and S100 and CD1a were positive in the immunohistochemical stain. Gastrointestinal involvement of LCH in adults is rare, asymptomatic,and can involve multiple digestive organs simultaneously, so upper endoscopy and colonoscopy should be considered for a diagnosis.

Keyword

Case reports; Histiocytosis; Langerhans-cell; Gastrointestinal tract; S100; CD1a

Figure

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  Fig. 1 Computed tomography (CT) findings of the right neck lymph node and mediastinal lymph node (black triangular arrows). (A) 3 cm-sized lymph node at the right level II neck on the axial view of the pharyngeal CT. Axial view. (B) Coronal view of the pharyngeal CT. (C) Conglomerated multiple small to enlarged lymph nodes in right upper paratracheal and prevascular area on the axial view of the chest CT. (D) Coronal view of the chest CT.

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  Fig. 2 Endoscopic findings of multiple involvement in the stomach and colon in adult Langerhans cell histiocytosis patients. (A) Multiple subepithelial tumors with central erosion on fundus. (B) 1.2 cm sized ulcerative mass with elevated margin on the great curvature of the body. (C) Subepithelial tumor with central hyperemic erosion on the ascending colon. (D) Subepithelial tumor with central umbilicus on the transverse colon.

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  Fig. 3 Pathology findings of the right neck lymph node and gastric ulcerative lesion. (A) Pathologic findings of the right neck lymph node (hematoxylin and eosin stain [H&E], ×100). (B) Pathologic findings of the gastric ulcerative lesion (H&E, ×100). (C) CD1a staining was positive (×100). (D) S-100 staining was positive (×100).

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