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Crystal-storing histiocytosis: a clinicopathological study of 13 cases.

Publication ,  Journal Article
Kanagal-Shamanna, R; Xu-Monette, ZY; Miranda, RN; Dogan, A; Zou, D; Luthra, R; Weber, DM; O'Malley, DP; Jorgensen, JL; Khoury, JD; Orlowski, RZ ...
Published in: Histopathology
March 2016

Crystal-storing histiocytosis (CSH) is a rare lesion composed of histiocytes with abnormal intralysosomal accumulation of immunoglobulin (Ig) as crystals, reported in patients with plasmacytic/lymphoplasmacytic neoplasms. The aims of this study were to report the clinicopathological features of 13 patients with CSH, and to describe the proteomic composition of the crystals in three cases analysed by mass spectrometry (MS).There were seven men and six women, with a median age of 60 years (range, 33-79 years). CSH was generalized in one patient (8%) and localized in 12 (92%) patients, involving various sites. CSH was associated with a low-grade B-cell lymphoma with plasmacytoid differentiation or a plasma cell neoplasm in all cases. In 10 (77%) cases, CSH represented >50% of the neoplastic infiltrate. According to immunohistochemical studies, histiocytes were positive for monotypic kappa in 5 (50%) cases, and for monotypic lambda in 4 (40%) cases; in 1 (10%) case, the results were equivocal. MS analysis of the histiocyte contents in all three tested cases showed a predominance of variable-region fragments of Ig light and/or heavy chains.CSH is frequently associated with an underlying lymphoplasmacytic neoplasm. MS findings suggest that Ig alterations and/or possibly defects in the ability of histiocytes to process Ig play a role in pathogenesis.

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Published In

Histopathology

DOI

EISSN

1365-2559

ISSN

0309-0167

Publication Date

March 2016

Volume

68

Issue

4

Start / End Page

482 / 491

Related Subject Headings

  • Polymerase Chain Reaction
  • Pathology
  • Neoplasms, Plasma Cell
  • Middle Aged
  • Mass Spectrometry
  • Male
  • Lymphoma, B-Cell
  • Laser Capture Microdissection
  • Inclusion Bodies
  • Immunohistochemistry
 

Citation

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Kanagal-Shamanna, R., Xu-Monette, Z. Y., Miranda, R. N., Dogan, A., Zou, D., Luthra, R., … Young, K. H. (2016). Crystal-storing histiocytosis: a clinicopathological study of 13 cases. Histopathology, 68(4), 482–491. https://doi.org/10.1111/his.12768
Kanagal-Shamanna, Rashmi, Zijun Y. Xu-Monette, Roberto N. Miranda, Ahmet Dogan, Dehui Zou, Rajyalakshmi Luthra, Donna M. Weber, et al. “Crystal-storing histiocytosis: a clinicopathological study of 13 cases.Histopathology 68, no. 4 (March 2016): 482–91. https://doi.org/10.1111/his.12768.
Kanagal-Shamanna R, Xu-Monette ZY, Miranda RN, Dogan A, Zou D, Luthra R, et al. Crystal-storing histiocytosis: a clinicopathological study of 13 cases. Histopathology. 2016 Mar;68(4):482–91.
Kanagal-Shamanna, Rashmi, et al. “Crystal-storing histiocytosis: a clinicopathological study of 13 cases.Histopathology, vol. 68, no. 4, Mar. 2016, pp. 482–91. Epmc, doi:10.1111/his.12768.
Kanagal-Shamanna R, Xu-Monette ZY, Miranda RN, Dogan A, Zou D, Luthra R, Weber DM, O’Malley DP, Jorgensen JL, Khoury JD, Bueso-Ramos CE, Orlowski RZ, Medeiros LJ, Young KH. Crystal-storing histiocytosis: a clinicopathological study of 13 cases. Histopathology. 2016 Mar;68(4):482–491.
Journal cover image

Published In

Histopathology

DOI

EISSN

1365-2559

ISSN

0309-0167

Publication Date

March 2016

Volume

68

Issue

4

Start / End Page

482 / 491

Related Subject Headings

  • Polymerase Chain Reaction
  • Pathology
  • Neoplasms, Plasma Cell
  • Middle Aged
  • Mass Spectrometry
  • Male
  • Lymphoma, B-Cell
  • Laser Capture Microdissection
  • Inclusion Bodies
  • Immunohistochemistry