Histiocytoid Sweet syndrome harboring an isocitrate dehydrogenase 1 mutation: A case report and retrospective analysis of 29 cases of histiocytoid Sweet syndrome

Libby, Tiffany J.; Fleming, Kirsten E.; Amin, Bijal

doi:10.1111/cup.13418

Cited by 5 publications

(8 citation statements)

References 7 publications

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“…In contrast to histiocytoid Sweet syndrome, in overt leukemia cutis, immature myeloblasts are present and can stain positive for CD34 and CD117 7 . Interestingly, histiocytoid Sweet syndrome harboring an IDH1 mutation has been previously reported, demonstrating a neutrophil‐rich infiltrate 8 . In contrast to this previously reported case, the infiltrate in our case was composed primarily of immature mononuclear cells compatible with blasts.…”

Section: Discussioncontrasting

confidence: 79%

Differentiation syndrome during ivosidenib treatment with immunohistochemistry showing isocitrate dehydrogenase R132H mutation

et al. 2020

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We report a case of differentiation syndrome in a patient receiving the IDH1 inhibitor ivosidenib, with skin biopsy showing isocitrate dehydrogenase (IDH) R132Hmutated leukemia cutis. A 72-year-old man with IDH1-mutated acute myeloid leukemia (AML), status-post allogeneic cell transplantation, on ivosidenib for 6 months, was admitted for culture-negative neutropenic fever, pink and purpuric plaques and patches on the legs, abdomen and back, edema, hypotension, and shortness of breath. Skin biopsy revealed an infiltrate of atypical, immature, myeloperoxidase-positive mononuclear cells compatible with leukemia cutis or Sweet syndrome. Although dermal edema and interstitial neutrophilic infiltrate with

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Section: Discussioncontrasting

confidence: 79%

Differentiation syndrome during ivosidenib treatment with immunohistochemistry showing isocitrate dehydrogenase R132H mutation

et al. 2020

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“…The biopsy findings were also unique and demonstrated morphologic features of both variants of Sweet syndrome. Histopathologic variants of Sweet syndrome include the subcutaneous form with deeper involvement and histiocytoid Sweet syndrome that includes the presence of neutrophilic precursors with kidney‐shaped elongated nuclei resembling histiocytes 7 . Histiocytoid Sweet may also have the infiltrate extending into the subcutis, 8 similar to that seen in our case.…”

Section: Discussionsupporting

confidence: 70%

Neoplastic or inflammatory? A case report of Sweet syndrome with CD30+ cells in a patient with B‐lymphoblastic leukemia

Ehyaee,

Reddy,

Ahmed

2023

J Cutan Pathol

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CD30+ cells are typically part of lymphoproliferative disorders but can also be seen in inflammatory dermatoses. We present a case of 47‐year‐old man with a history of B‐lymphoblastic leukemia (B‐ALL) who presented with fever, leukocytosis, and papulonodular skin lesions, involving the extremities and trunk. A punch biopsy specimen demonstrated papillary dermal edema with a neutrophilic and histiocytic infiltrate extending into the subcutis. The infiltrate also harbored scattered large cells that were positive for CD30 and demonstrated the immunohistochemical profile of monocytes. A diagnosis of histiocytoid Sweet syndrome with CD30+ cells was made. The case is unique, demonstrating a combination of Sweet syndrome variants with subcutis involvement, histiocytoid morphology, and large CD30+ cells. A prior history of B‐ALL and immunohistochemical profile of monocytes with immature morphology broadened the differential diagnosis and added to the diagnostic challenge.

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“…No biopsies of H-SS demonstrated purely subcutaneous infiltrates, although some cases did have an extension of the dermal infiltrate into the subcutis. Similarly, some of the conventional Sweet syndrome cases had at least minor involvement of the subcutis in addition to the dermis, but no cases had purely subcutaneous involvement [ 53 ].…”

Section: Resultsmentioning

confidence: 99%

“…It has been hypothesized that these lesions begin with the release of immature myeloid cells from the bone marrow in early stages, and these cells mature to neutrophils in later stages of the disease [ 53 ]. Histopathological differential diagnosis should be established with leukemia cutis [ 63 ] and other inflammatory conditions characterized by histiocytes interstitially arranged between collagen bundles of the dermis [ 52 , 54 , 61 ].…”

Section: Resultsmentioning

confidence: 99%

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Sweet Syndrome Associated with Myelodysplastic Syndrome—A Review of a Multidisciplinary Approach

Ferea

Mihai

Bălan

et al. 2023

Life

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Sweet syndrome (SS) is a rare disease described as a febrile neutrophilic dermatosis with acute onset, the pathogenesis of which has not yet been elucidated. The syndrome is characterized by the sudden onset of erythematous infiltrated papules or plaques located on the upper body and is associated with fever, leukocytosis and neutrophilia. The lesions show a dense dermal infiltration with mature neutrophils. The condition is responsive to systemic steroids. The central nervous system, bones, muscles, eyes, ears, mouth, heart, lung, liver, kidneys, intestines, and spleen may be affected by SS as extracutaneous manifestations. More and more cases have been found to be associated with malignancies, particularly myelodysplastic syndrome, and, less frequently, other hematologic malignancies or solid tumors. Approximately 21% of patients with SS have an associated malignancy and up to 80% of MASS cases are associated with hematological diseases, predominantly myelodysplastic syndrome (MDS) or acute myeloid leukemia (AML). Myelodysplastic syndrome is a clonal disease of the bone marrow characterized by inefficient hematopoiesis, dysplasia of the bone marrow and peripheral cytopenias. Affected patients have a high risk of leukemic transformation. After analyzing later studies and current practical aspects regarding MDS-related SS, we suggest an algorithm for evaluating these patients.

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Histiocytoid Sweet syndrome harboring an isocitrate dehydrogenase 1 mutation: A case report and retrospective analysis of 29 cases of histiocytoid Sweet syndrome

Cited by 5 publications

References 7 publications

Differentiation syndrome during ivosidenib treatment with immunohistochemistry showing isocitrate dehydrogenase R132H mutation

Differentiation syndrome during ivosidenib treatment with immunohistochemistry showing isocitrate dehydrogenase R132H mutation

Neoplastic or inflammatory? A case report of Sweet syndrome with CD30+ cells in a patient with B‐lymphoblastic leukemia

Sweet Syndrome Associated with Myelodysplastic Syndrome—A Review of a Multidisciplinary Approach

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