Cargando…

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...

Descripción completa

Detalles Bibliográficos
Autores principales: Kong, Linghong, Xue, Liyan, Zhong, Yanfeng, Wang, Shenglan, Zheng, Danfeng, Wang, Lining, Jiao, Yang, Zhang, Xinpeng, Xue, Huizhong, Liu, Xiaogang
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9798227/
https://www.ncbi.nlm.nih.gov/pubmed/36591494
http://dx.doi.org/10.3389/fonc.2022.1024971
_version_ 1784860863158550528
author Kong, Linghong
Xue, Liyan
Zhong, Yanfeng
Wang, Shenglan
Zheng, Danfeng
Wang, Lining
Jiao, Yang
Zhang, Xinpeng
Xue, Huizhong
Liu, Xiaogang
author_facet Kong, Linghong
Xue, Liyan
Zhong, Yanfeng
Wang, Shenglan
Zheng, Danfeng
Wang, Lining
Jiao, Yang
Zhang, Xinpeng
Xue, Huizhong
Liu, Xiaogang
author_sort Kong, Linghong
collection PubMed
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.
format Online
Article
Text
id pubmed-9798227
institution National Center for Biotechnology Information
language English
publishDate 2022
publisher Frontiers Media S.A.
record_format MEDLINE/PubMed
spelling pubmed-97982272022-12-30 Crystal-storing histiocytosis in the stomach: A case report and review of the literature Kong, Linghong Xue, Liyan Zhong, Yanfeng Wang, Shenglan Zheng, Danfeng Wang, Lining Jiao, Yang Zhang, Xinpeng Xue, Huizhong Liu, Xiaogang Front Oncol Oncology 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. Frontiers Media S.A. 2022-12-15 /pmc/articles/PMC9798227/ /pubmed/36591494 http://dx.doi.org/10.3389/fonc.2022.1024971 Text en Copyright © 2022 Kong, Xue, Zhong, Wang, Zheng, Wang, Jiao, Zhang, Xue and Liu https://creativecommons.org/licenses/by/4.0/This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.
spellingShingle Oncology
Kong, Linghong
Xue, Liyan
Zhong, Yanfeng
Wang, Shenglan
Zheng, Danfeng
Wang, Lining
Jiao, Yang
Zhang, Xinpeng
Xue, Huizhong
Liu, Xiaogang
Crystal-storing histiocytosis in the stomach: A case report and review of the literature
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 Oncology
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9798227/
https://www.ncbi.nlm.nih.gov/pubmed/36591494
http://dx.doi.org/10.3389/fonc.2022.1024971
work_keys_str_mv AT konglinghong crystalstoringhistiocytosisinthestomachacasereportandreviewoftheliterature
AT xueliyan crystalstoringhistiocytosisinthestomachacasereportandreviewoftheliterature
AT zhongyanfeng crystalstoringhistiocytosisinthestomachacasereportandreviewoftheliterature
AT wangshenglan crystalstoringhistiocytosisinthestomachacasereportandreviewoftheliterature
AT zhengdanfeng crystalstoringhistiocytosisinthestomachacasereportandreviewoftheliterature
AT wanglining crystalstoringhistiocytosisinthestomachacasereportandreviewoftheliterature
AT jiaoyang crystalstoringhistiocytosisinthestomachacasereportandreviewoftheliterature
AT zhangxinpeng crystalstoringhistiocytosisinthestomachacasereportandreviewoftheliterature
AT xuehuizhong crystalstoringhistiocytosisinthestomachacasereportandreviewoftheliterature
AT liuxiaogang crystalstoringhistiocytosisinthestomachacasereportandreviewoftheliterature