Crystal-storing histiocytosis in the stomach: A case report and review of the literature

Crystal-storing histiocytosis (CSH) is a rare disorder characterized by the accumulation of non-neoplastic histiocytes that contain intracytoplasmic crystallized immunoglobulins. Although CSH can occur in various organs, gastric CSH is very rare. Therefore, diagnosing gastric CSH remains a challenge...

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Main Authors: Linghong Kong, Liyan Xue, Yanfeng Zhong, Shenglan Wang, Danfeng Zheng, Lining Wang, Yang Jiao, Xinpeng Zhang, Huizhong Xue, Xiaogang Liu
Format: Article
Language:English
Published: Frontiers Media S.A. 2022-12-01
Series:Frontiers in Oncology
Subjects:
Online Access:https://www.frontiersin.org/articles/10.3389/fonc.2022.1024971/full
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author Linghong Kong
Liyan Xue
Yanfeng Zhong
Shenglan Wang
Danfeng Zheng
Lining Wang
Yang Jiao
Xinpeng Zhang
Huizhong Xue
Xiaogang Liu
author_facet Linghong Kong
Liyan Xue
Yanfeng Zhong
Shenglan Wang
Danfeng Zheng
Lining Wang
Yang Jiao
Xinpeng Zhang
Huizhong Xue
Xiaogang Liu
author_sort Linghong Kong
collection DOAJ
description Crystal-storing histiocytosis (CSH) is a rare disorder characterized by the accumulation of non-neoplastic histiocytes that contain intracytoplasmic crystallized immunoglobulins. Although CSH can occur in various organs, gastric CSH is very rare. Therefore, diagnosing gastric CSH remains a challenge. Here, we present the case of a 69-year-old man with localized gastric CSH who presented with positive fecal occult blood for 2 days. Gastroscopy showed that there was a piece of irregular whitish focus in the big bend of the gastric antrum, which was soft and elastic. Histologically, the biopsied gastric mucosa showed chronic inflammation, mild activity with erosion, and numerous eosinophilic mononuclear cells containing fibrillary crystalloid inclusions in the lamina propria. Immunohistochemically, these crystal-containing cells were positive for CD68/PGM1 and Igk, which revealed that the cells were histiocytes harboring kappa light chain-restricted immunoglobulin crystals. Electron microscopic examination showed numerous high-electron-density particles in the cytoplasm of cells, with crystal structures of different sizes and shapes. This case highlights how immunohistochemistry can help with differential diagnosis and classification.
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spelling doaj.art-a7667b5c237840a1a10d54d5021136142022-12-22T04:22:30ZengFrontiers Media S.A.Frontiers in Oncology2234-943X2022-12-011210.3389/fonc.2022.10249711024971Crystal-storing histiocytosis in the stomach: A case report and review of the literatureLinghong Kong0Liyan Xue1Yanfeng Zhong2Shenglan Wang3Danfeng Zheng4Lining Wang5Yang Jiao6Xinpeng Zhang7Huizhong Xue8Xiaogang Liu9Department of Pathology, Beijing Chuiyangliu hospital, Beijing, ChinaDepartment of Pathology, National Cancer Center/National Clinical Research Center for Cancer/Cancer Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing, ChinaDepartment of Pathology, School of Basic Medical Sciences, Peking University Health Science Center, Beijing, ChinaDepartment of Pathology, School of Basic Medical Sciences, Peking University Health Science Center, Beijing, ChinaDepartment of Pathology, School of Basic Medical Sciences, Peking University Health Science Center, Beijing, ChinaDepartment of Pathology, Beijing Chuiyangliu hospital, Beijing, ChinaDepartment of Pathology, Beijing Chuiyangliu hospital, Beijing, ChinaDepartment of Pathology, Beijing Chuiyangliu hospital, Beijing, ChinaDepartment of Pathology, Beijing Chuiyangliu hospital, Beijing, ChinaDepartment of Pathology, Beijing Chuiyangliu hospital, Beijing, ChinaCrystal-storing histiocytosis (CSH) is a rare disorder characterized by the accumulation of non-neoplastic histiocytes that contain intracytoplasmic crystallized immunoglobulins. Although CSH can occur in various organs, gastric CSH is very rare. Therefore, diagnosing gastric CSH remains a challenge. Here, we present the case of a 69-year-old man with localized gastric CSH who presented with positive fecal occult blood for 2 days. Gastroscopy showed that there was a piece of irregular whitish focus in the big bend of the gastric antrum, which was soft and elastic. Histologically, the biopsied gastric mucosa showed chronic inflammation, mild activity with erosion, and numerous eosinophilic mononuclear cells containing fibrillary crystalloid inclusions in the lamina propria. Immunohistochemically, these crystal-containing cells were positive for CD68/PGM1 and Igk, which revealed that the cells were histiocytes harboring kappa light chain-restricted immunoglobulin crystals. Electron microscopic examination showed numerous high-electron-density particles in the cytoplasm of cells, with crystal structures of different sizes and shapes. This case highlights how immunohistochemistry can help with differential diagnosis and classification.https://www.frontiersin.org/articles/10.3389/fonc.2022.1024971/fullcrystal-storing histiocytosisstomachhistopathologyimmunoglobulinelectron microscopy
spellingShingle Linghong Kong
Liyan Xue
Yanfeng Zhong
Shenglan Wang
Danfeng Zheng
Lining Wang
Yang Jiao
Xinpeng Zhang
Huizhong Xue
Xiaogang Liu
Crystal-storing histiocytosis in the stomach: A case report and review of the literature
Frontiers in Oncology
crystal-storing histiocytosis
stomach
histopathology
immunoglobulin
electron microscopy
title Crystal-storing histiocytosis in the stomach: A case report and review of the literature
title_full Crystal-storing histiocytosis in the stomach: A case report and review of the literature
title_fullStr Crystal-storing histiocytosis in the stomach: A case report and review of the literature
title_full_unstemmed Crystal-storing histiocytosis in the stomach: A case report and review of the literature
title_short Crystal-storing histiocytosis in the stomach: A case report and review of the literature
title_sort crystal storing histiocytosis in the stomach a case report and review of the literature
topic crystal-storing histiocytosis
stomach
histopathology
immunoglobulin
electron microscopy
url https://www.frontiersin.org/articles/10.3389/fonc.2022.1024971/full
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