S-100 –Negative, CD1a-Positive Histiocytosis: A New Variant With Features of Both Indeterminate Cells and Interstitial Dermal Dendrocytes

Ventarola, Daniel; Mercer, Stephen E.; Contard, Paul; Odaimi, Marcel; Emanuel, Patrick O.; Birgé, Miriam B.; Phelps, Robert

doi:10.1001/archdermatol.2011.219

Cited by 14 publications

(24 citation statements)

References 4 publications

(4 reference statements)

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“…Treatment for this S‐100 − variant of ICH depends on the organ or system involved. There have been documented cases of using UVA, phototherapy, topical 5‐fluorouracil, total skin electron beam therapy, thalidomide, and surgical excision for cutaneous lesions . Spontaneous regression of skin lesions has also been reported .…”

Section: Discussionmentioning

confidence: 80%

“…Dermal macrophages are capable of being induced to differentiate into Langerhans cells, and there is thought to be a common transitional macrophage/dendritic cell progenitor for Langerhans cells, indeterminate cells, and macrophages. The unique immunophenotype is hypothesized to be caused by arrest in the maturation process of the histiocytes …”

Section: Discussionmentioning

confidence: 99%

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A case of S‐100‐negative CD1a‐positive indeterminate cell histiocytosis

et al. 2019

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Histiocytoses are a group of rare disorders characterized by a proliferation of monocytes/macrophages and dendritic cells. We present a case of a 3‐year‐old girl with a diffuse papular eruption without systemic symptoms demonstrating a proliferation of strongly CD1a+ histiocytes, but negative for S‐100 and langerin on histopathology. Systemic work‐up including bone marrow biopsy was unremarkable, and the patient received a diagnosis of CD1a+ S− 100‐indeterminate cell histiocytosis.

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Section: Discussionmentioning

confidence: 80%

Section: Discussionmentioning

confidence: 99%

A case of S‐100‐negative CD1a‐positive indeterminate cell histiocytosis

et al. 2019

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“…Immunoreactivity for S‐100 was reported in almost all instances where it was tested (74/77, 96%), although the staining pattern and intensity varied widely. Moreover, while some authors were comfortable supporting an indeterminate dendritic cell lineage in the absence of S‐100 positivity, other reports of similar lesions were equivocal regarding lineage assignment …”

Section: Discussionmentioning

confidence: 99%

Indeterminate dendritic cell neoplasm of the skin: A 2‐case report and review of the literature

et al. 2017

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Indeterminate dendritic cell neoplasm (IDCN) is an exceedingly rare and mostly cutaneous histiocytosis, frequently associated with other hematopoietic malignancies. We report 2 cases of multilesional cutaneous IDCN. A 55-year-old male with no associated malignancy and complete response to ultraviolet phototherapy; and a 72-year-old male with chronic myelomonocytic leukemia (CMML). Both cases showed histiocytoid cytology, positivity for CD1a and no expression of langerin or BRAF . With our patients, the literature describes 79 cases of IDCNs, including 65 (82%) with only skin involvement, 7 cases (9%) with involvement of skin and a second site, 5 cases (6%) involving lymph nodes only, 1 splenic lesion and 1 systemic disease. Seventeen cases (22%) were associated with other hematopoietic malignancies, most commonly CMML (6 cases), follicular lymphoma (4 cases) and acute myeloid leukemia (3 cases). All IDCNs associated with myeloid malignancies were limited to the skin, while most cases associated with lymphoma were limited to lymph nodes. Reported responses of cutaneous lesions to ultraviolet phototherapy are encouraging, while systemic chemotherapy is appropriate for clinically aggressive cases and treatment of associated malignancies. Recognition of the clinico-morphologic spectrum of IDCNs should prevent misdiagnoses and prompt investigation of possible associated neoplasms.

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“…1 These 2 diagnoses are mainly differentiated by the expression of langerin (CD207), which is a surrogate marker for Birbeck granules. 1,3,4,7 In our patient, the lack of expression of langerin in the cutaneous infiltrate together with the expression of CD1a, raised consideration to a diagnosis of ICH over LCH. The lack of expression of S100 protein on skin biopsy is the primary factor that separates this presentation from typical ICH.…”

Section: Discussionmentioning

confidence: 73%

“…[1][2][3] Both macrophages and dendritic cells arise from common progenitor cells in the bone marrow. 2 In the skin, antigen-presenting cells are mainly represented by indeterminate cells, Langerhans cells, and interstitial dendritic cells.…”

Section: Introductionmentioning

confidence: 99%

S100-Negative, CD1a-Positive Cutaneous Histiocytosis in a Patient with S100-Positive, CD1a-Positive Pulmonary Histiocytosis

Mask-Bull

Crowson

John

et al. 2015

The American Journal of Dermatopathology

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In the diagnostic approach to histiocytic proliferations, immunohistochemistry may be a source of both confusion and clarification. We present a case of a 60-year-old man with a generalized pruritic eruption that demonstrated positive staining for CD1a, but negative staining for langerin and S100 protein. This immunophenotype is neither representative nor characteristic of any recognized dendritic cell tumor but has been previously described in 3 cases of skin-limited histiocytosis. However, our patient also demonstrated pulmonary histiocytic infiltrates that were positive for both CD1a and S100 proteins. This differing expression of S100 protein witnessed in 2 separate organ systems affords us insight into the pathophysiology of these histiocytic proliferations.

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S-100 –Negative, CD1a-Positive Histiocytosis: A New Variant With Features of Both Indeterminate Cells and Interstitial Dermal Dendrocytes

Cited by 14 publications

References 4 publications

A case of S‐100‐negative CD1a‐positive indeterminate cell histiocytosis

A case of S‐100‐negative CD1a‐positive indeterminate cell histiocytosis

Indeterminate dendritic cell neoplasm of the skin: A 2‐case report and review of the literature

S100-Negative, CD1a-Positive Cutaneous Histiocytosis in a Patient with S100-Positive, CD1a-Positive Pulmonary Histiocytosis

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