Journal
HUMAN PATHOLOGY
Volume 65, Issue -, Pages 180-186Publisher
W B SAUNDERS CO-ELSEVIER INC
DOI: 10.1016/j.humpath.2016.10.028
Keywords
Crystal-storing histiocytosis; Liquid chromatography-tandem mass spectrometry; MALT; Mucosa-associated lymphoid tissue lymphoma; Pulmonary mucosa-associated lymphoid tissue lymphoma
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Funding
- Ministry of Education, Culture, Sports, Science, and Technology of Japan (Chiyoda-ku, Tokyo, Japan)
- Ministry of Health, Labor, and Welfare of Japan (Chiyoda-ku, Tokyo, Japan)
- Grants-in-Aid for Scientific Research [15K19433, 17K09630] Funding Source: KAKEN
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Crystal-storing histiocytosis (CSH) is an uncommon finding in lymphoplasmacytic disorders that presents histiocytes with abnormal intralysosomal accumulations of immunoglobulin light chains as crystals of unknown etiology. A 38-year-old woman with antiphospholipid syndrome had a surgical lung biopsy because of multiple lung mass lesions. In a right middle lobe lesion, lymphoplasmacytic cells had a monocytoid appearance, destructive lymphoepithelial lesions, and positive immunoglobulin heavy chain (IGH) gene rearrangements. A right upper lobe lesion manifested proliferating rounded histiocytes with abundant, deeply eosinophilic cytoplasm and negative IGH gene rearrangements. Electron microscopy and mass spectrometry revealed a case of pulmonary CSH: abnormal proliferation of the immunoglobulin k chain of a variable region that may be crystallized within plasma cells and histiocytes. We report a rare case of localized pulmonary CSH complicating pulmonary mucosa-associated lymphoid tissue lymphoma with multiple mass lesions. We demonstrate advances in the understanding of the pathogenesis of CSH by various analyses of these lesions. (C) 2017 Elsevier Inc. All rights reserved.
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