Nonimmunoglobulin Crystal-Storing Histiocytosis (CSH): Case Report and Literature Review
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 lymphoprolifer...
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| Format: | Article |
| Language: | English |
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Wiley
2020-01-01
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| Series: | Case Reports in Hematology |
| Online Access: | http://dx.doi.org/10.1155/2020/8856411 |
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| author | Manuel Beltran Sharad Khurana Yennifer Gil Jason T. Lewis Rohit Kumar James M. Foran |
| author_facet | Manuel Beltran Sharad Khurana Yennifer Gil Jason T. Lewis Rohit Kumar James M. Foran |
| author_sort | Manuel Beltran |
| collection | DOAJ |
| description | 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. |
| format | Article |
| id | doaj-art-94b1ca1a619f43aabdadcd7779fa3e9b |
| institution | OA Journals |
| issn | 2090-6560 2090-6579 |
| language | English |
| publishDate | 2020-01-01 |
| publisher | Wiley |
| record_format | Article |
| series | Case Reports in Hematology |
| spelling | doaj-art-94b1ca1a619f43aabdadcd7779fa3e9b2025-08-20T02:09:48ZengWileyCase Reports in Hematology2090-65602090-65792020-01-01202010.1155/2020/88564118856411Nonimmunoglobulin Crystal-Storing Histiocytosis (CSH): Case Report and Literature ReviewManuel Beltran0Sharad Khurana1Yennifer Gil2Jason T. Lewis3Rohit Kumar4James M. Foran5Division of General Internal Medicine, Mayo Clinic, Jacksonville, FL, USADivision of Hematology & Oncology, University of Arizona Cancer Center, Tucson, AZ, USADepartment of Anesthesia and Preoperative Medicine, Mayo Clinic, Jacksonville, FL, USADepartment of Pathology, Mayo Clinic, Jacksonville, FL, USADivision of Medical Oncology and Hematology, University of Louisville, Louisville, KY, USADepartment of Hematology & Oncology, Mayo Clinic, Jacksonville, FL, USACrystal-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.http://dx.doi.org/10.1155/2020/8856411 |
| spellingShingle | Manuel Beltran Sharad Khurana Yennifer Gil Jason T. Lewis Rohit Kumar James M. Foran Nonimmunoglobulin Crystal-Storing Histiocytosis (CSH): Case Report and Literature Review Case Reports in Hematology |
| title | Nonimmunoglobulin Crystal-Storing Histiocytosis (CSH): Case Report and Literature Review |
| title_full | Nonimmunoglobulin Crystal-Storing Histiocytosis (CSH): Case Report and Literature Review |
| title_fullStr | Nonimmunoglobulin Crystal-Storing Histiocytosis (CSH): Case Report and Literature Review |
| title_full_unstemmed | Nonimmunoglobulin Crystal-Storing Histiocytosis (CSH): Case Report and Literature Review |
| title_short | Nonimmunoglobulin Crystal-Storing Histiocytosis (CSH): Case Report and Literature Review |
| title_sort | nonimmunoglobulin crystal storing histiocytosis csh case report and literature review |
| url | http://dx.doi.org/10.1155/2020/8856411 |
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