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Nonimmunoglobulin Crystal-Storing Histiocytosis (CSH): Case Report and Literature Review

Nonimmunoglobulin Crystal-Storing Histiocytosis (CSH): Case Report and Literature Review

Authors :
Manuel Beltran
Sharad Khurana
Yennifer Gil
Jason T. Lewis
Rohit Kumar
James M. Foran
Source :
Case Reports in Hematology, Vol 2020 (2020)
Publication Year :
2020
Publisher :
Hindawi Limited, 2020.

Abstract

Crystal-storing histiocytosis (CSH) is an uncommon condition in which histiocytes accumulate a crystalline matter within their cytoplasm. Generally, those crystals are composed of either monoclonal or polyclonal immunoglobulin chains, which have a strong association with an underlying lymphoproliferative or plasma cell disorder (LP-PCD). Rarely, CSH has been reported as local or generalized manifestation of a variety of benign disorders. These cases are associated with crystals composed of nonimmunoglobulin substances. We are reporting an exceptional case of a local colonic CSH with Charcot–Leyden crystals. This patient underwent a screening colonoscopy that detected some polyps. The biopsy reported tubular adenomas, with a markedly dense, transmural inflammatory infiltrates, which were predominantly composed of eosinophils and crystal-storing histiocytes containing Charcot–Leyden crystals. The patient had a negative workup for LP-PCD and autoimmune conditions, including a normal skeletal survey and bone marrow aspirate/biopsy. The only positive laboratory workup was an elevated absolute eosinophil count and a positive IgG anti-Strongyloides antibody. Giving those findings, this parasitic infection is the most likely etiology of the CSH in our patient. Although there was an initial negative evaluation for LP-PCD, close monitoring of patients with either immunoglobulin or nonimmunoglobulin CSH is recommended.

Details

Language :
English
ISSN :
20906560 and 20906579
Volume :
2020
Database :
Directory of Open Access Journals
Journal :
Case Reports in Hematology
Publication Type :
Academic Journal
Accession number :
edsdoj.94b1ca1a619f43aabdadcd7779fa3e9b
Document Type :
article
Full Text :
https://doi.org/10.1155/2020/8856411