Crystal-Storing Histiocytosis: The Iceberg of More Serious Conditions

Mobarki, Mousa; Papoudou‐Bai, Alexandra; Dumollard, Jean Marc; Musawi, Shaqraa; Madkhali, Mohammed Ali; Muqri, Khalid Y.; Péoc’h, Michel; Karpathiou, Georgia

doi:10.3390/diagnostics13020271

Cited by 6 publications

(4 citation statements)

References 140 publications

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“…14 organ involvement. Multiorgan involvement in CSH was identified in 28.9% of all CSH cases, 15 which is comparable to our findings in cutaneous CSH (30%). The bone marrow, liver, and lymph nodes appear to be the most commonly involved organs in patients with cutaneous CSH.…”

Section: Review Of Literaturesupporting

confidence: 90%

“…All described cutaneous CSH cases were associated with an underlying LP‐PCD, with MM being the most common. Approximately 10% of CSH cases, however, do not have an identifiable underlying LP‐PCD 2,15 . In such instances, CSH may be the sole pathologic finding or may be related to an infection or a benign disease with an inflammatory/autoimmune component, like Crohn's disease or rheumatoid arthritis 1,13,15 .…”

Section: Discussionmentioning

confidence: 99%

“…Approximately 10% of CSH cases, however, do not have an identifiable underlying LP-PCD. 2,15 In such instances, CSH may be the sole pathologic finding or may be related to an infection or a benign disease with an inflammatory/autoimmune component, like Crohn's disease or rheumatoid arthritis. 1,13,15 CSH was also reported as one of the possible cutaneous manifestations of IgG4-related disease.…”

Section: Review Of Literaturementioning

confidence: 99%

“…2,15 In such instances, CSH may be the sole pathologic finding or may be related to an infection or a benign disease with an inflammatory/autoimmune component, like Crohn's disease or rheumatoid arthritis. 1,13,15 CSH was also reported as one of the possible cutaneous manifestations of IgG4-related disease. 3 In addition, CSH can precede LP-PCD with a varying duration of months to years before diagnosis of LP-PCD.…”

Section: Review Of Literaturementioning

confidence: 99%

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Cutaneous crystal storing histiocytosis: A case series with review of literature

Homsi,

Knapp,

Agrawal

et al. 2024

J Cutan Pathol

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Crystal‐storing histiocytosis (CSH) is a rare condition in which crystals accumulate in the cytoplasm of histiocytes and is usually associated with a lymphoplasmacytic neoplasm. Cutaneous CSH is extraordinarily rare and limited to case reports in the literature. We report two cases of this disease with cutaneous involvement. Case 1 was a 65‐year‐old male with a 4‐month history of a pruritic eruption that started as a solitary pink to skin‐colored indurated plaque on the anterior neck before progressing to involve the whole neck, chest wall, and face. Case 2 was a 54‐year‐old woman with a history of unspecified “lymphoma” who presented with a soft nodule on the forearm. Biopsies from both cases had similar findings and showed a proliferation of epithelioid cells with pink cytoplasm and intracellular crystalline structures infiltrating the dermis and subcutaneous fat. In the first case, the cells were positive for CD43, CD45, CD68, and IgG kappa, and in the second case, the crystals were positive for IgG lambda. Based on these findings, the patients were diagnosed with cutaneous CSH. We highlight this rare diagnosis and the importance of investigating an underlying lymphoplasmacytic neoplasm.

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Section: Review Of Literaturesupporting

confidence: 90%

Section: Discussionmentioning

confidence: 99%