Histiocytoid Sweet syndrome harboring an isocitrate dehydrogenase 1 mutation

A case report and retrospective analysis of 29 cases of histiocytoid Sweet syndrome

Tiffany J. Libby, Kirsten Fleming, Bijal D. Amin

Research output: Contribution to journalArticle

Abstract

Histiocytoid Sweet syndrome (HSS) is a rare histopathologic variant of Sweet syndrome that demonstrates dermal and/or subcutaneous infiltrate with a prominent component of myeloid cells resembling histiocytes. It has been known to occur in association with hematologic neoplasms, including myelodysplastic syndrome (MDS) and acute myelogenous leukemia, but whether it confers an increased risk of such neoplasms is controversial. Here, we describe a case of a HSS that led to the diagnosis of MDS with an isocitrate dehydrogenase 1 (IDH-1) mutation and a corresponding study looking for additional cases of IDH-1 mutations in biopsies of histiocytoid and conventional Sweet syndrome.

Original languageEnglish (US)
JournalJournal of Cutaneous Pathology
DOIs
StatePublished - Jan 1 2019

Fingerprint

Sweet Syndrome
Isocitrate Dehydrogenase
Mutation
Myelodysplastic Syndromes
Histiocytes
Hematologic Neoplasms
Myeloid Cells
Acute Myeloid Leukemia
Biopsy
Skin
Neoplasms

Keywords

  • histiocytoid sweet syndrome
  • myelodysplastic syndrome
  • subcutaneous sweet syndrome

ASJC Scopus subject areas

  • Pathology and Forensic Medicine
  • Histology
  • Dermatology

Cite this

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abstract = "Histiocytoid Sweet syndrome (HSS) is a rare histopathologic variant of Sweet syndrome that demonstrates dermal and/or subcutaneous infiltrate with a prominent component of myeloid cells resembling histiocytes. It has been known to occur in association with hematologic neoplasms, including myelodysplastic syndrome (MDS) and acute myelogenous leukemia, but whether it confers an increased risk of such neoplasms is controversial. Here, we describe a case of a HSS that led to the diagnosis of MDS with an isocitrate dehydrogenase 1 (IDH-1) mutation and a corresponding study looking for additional cases of IDH-1 mutations in biopsies of histiocytoid and conventional Sweet syndrome.",
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AU - Fleming, Kirsten

AU - Amin, Bijal D.

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AB - Histiocytoid Sweet syndrome (HSS) is a rare histopathologic variant of Sweet syndrome that demonstrates dermal and/or subcutaneous infiltrate with a prominent component of myeloid cells resembling histiocytes. It has been known to occur in association with hematologic neoplasms, including myelodysplastic syndrome (MDS) and acute myelogenous leukemia, but whether it confers an increased risk of such neoplasms is controversial. Here, we describe a case of a HSS that led to the diagnosis of MDS with an isocitrate dehydrogenase 1 (IDH-1) mutation and a corresponding study looking for additional cases of IDH-1 mutations in biopsies of histiocytoid and conventional Sweet syndrome.

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