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Cutaneous epitheloid angiomatous nodule and epitheloid hemangioma: Distinct entities on the epitheloid vascular tumors spectrum?
P1512
P1514
Localized cutaneous eruption with enlarged histiocytes induced by subcutaneous granulocyte macrophage colonyestimulating factor injections Matthew Hazey, MD, University of Louisville, Lousville, KY, United States; Jeffrey Callen, MD, University of Louisville, Louisville, KY, United States; Soon Bahrami, MD, University of Louisville, Louisville, KY, United States
Hypocellular plaque-like CD34-positive dermal fibroma (medallion-like dermal dendrocyte hamartoma) Lorea Bagazgoitia, MD, Hospital Ram on y Cajal, Madrid, Spain; Carmen Moreno, MD, Hospital Ram on y Cajal, Madrid, Spain; Pedro Jaen, MD, PhD, Hospital Ram on y Cajal, Madrid, Spain
Background: Granulocyte macrophage colonyestimulating factor (GM-CSF) and granulocyte colonyestimulating factor (G-CSF) have been associated with the development of localized and diffuse cutaneous eruptions. Previous reports of diffuse eruptions coinciding with their infusion have proposed that an increased number of enlarged dermal macrophages on histopathology suggests the etiologic role of these cytokines in the reaction. We report a 70-year-old woman who developed a localized hypersensitivity reaction at the site of subcutaneous GM-CSF injections. Biopsy specimens showed enlarged histiocytes within the dermal infiltrate, suggesting that this finding may signify etiology in both localized and diffuse eruptions caused by these agents. Observation: A 70-year-old white woman with history of T3bN1bM1a melanoma of the left lower extremity was referred for evaluation of a red, pruritic rash on her abdomen. She had recently been enrolled in a clinical trial for treatment of her melanoma, and the eruption began approximately 1 week after starting daily subcutaneous injections of GM-CSF into her abdominal wall as per the trial protocol. She denied any other new medications and was not being treated with other chemotherapy as part of the clinical trial. The physical examination revealed multiple erythematous, pseudovesiculated plaques with surrounding ecchymoses localized to the central and right abdomen. Biopsy specimens showed an eosinophilpredominant, pandermal inflammatory infiltrate and marked dermal edema, suggesting a hypersensitivity reaction. In addition, numerous plump histiocytes were present interstitially amongst the collagen bundles. Her eruption resolved with Clobetasol cream and cessation of GM-CSF injections.
Plaque-like CD34+ dermal fibroma (PDF) or (medallion-like dermal dendrocyte hamartoma) is a recently described congenital dermal neoplasm. Only 11 cases have been reported in the English literature, and its clinical and pathologic manifestations are not completely defined. We present the case of a 20-year-old man presenting with a round, erythematous, atrophic plaque on the midline of the anterior aspect of the neck. The lesion was asymptomatic and had been stable since birth. The histologic examination revealed a band-like hypocelullar fibrotic area in the superficial reticular dermis that did not spread to the deep reticular dermis and subcutaneous tissue. The fibrotic background and the scattered cells were CD34+ and S-100e. Embebed in this fibrotic area there were abundant capilar vessels without lost of elastic fibers as were demonstrated with the orcein stain. Clinical differential diagnosis includes atrophic DFSP, anetoderma, or aplasia cutis. Nevertheless, the morphologic features closely resembled those of a scar or an striae distensae. Although the histologic findings were not identical to those described recently as characteristic, the clinical features were suggestive enough to make the diagnosis of PDF. In our opinion, the PDF spectrum might include lesions with variable cellular density which can show similar clinical manifestations. Commercial support: None identified.
Comment: Because patient populations treated with GM-CSF and G-CSF are typically also being treated with various chemotherapy and antimicrobial agents, determining the etiology of a suspected drug-induced cutaneous eruption can be challenging. Our case provides further evidence that increased numbers of large, dermal histiocytes may implicate these cytokines as the inciting agent. Furthermore, the case illustrates that this histologic finding can be seen in localized reactions related to subcutaneous injections and more diffuse eruptions related to systemic infusion. Commercial support: None identified.
P1515
Pitfalls in the diagnosis of cutaneous RosaieDorfman disease: Report on two cases Vineet Mishra, MD, University of Alabama at Birmingham, Birmingham, AL, United States; Aleodor Andea, MD, University of Alabama at Birmingham, Birmingham, AL, United States; Boni Elewski, MD, University of Alabama at Birmingham, Birmingham, AL, United States; Carlo Gavino, MD, University of Alabama at Birmingham, Birmingham, AL, United States RosaieDorfman disease (RD) is a rare entity; therefore, atypical histologic presentations may pose diagnostic problems. We present two cases with a unique diagnostic challenge because of the lack of characteristic histologic features in the initial punch biopsy specimens. In both cases, the patients presented with a hyperpigmented, indurated plaque on the extremity. Punch biopsies were performed showing a mixed dermal infiltrate with abundant histiocytes that was negative for S100 and microorganisms both on special stains (GMS and AFB) and tissue cultures. Given these findings, a diagnosis of an atypical xanthogranuloma was given. Several months later, excisional biopsies were performed showing the classic histology of RD with a dense dermal infiltrate composed of large foamy cells exhibiting emperipolesis in a background of mixed acute and chronic inflammation. The foamy cells decorated with CD68 and S100 but were negative for CD1a. The diagnosis of cutaneous RD was subsequently sealed. In conclusion, our study documents a pitfall in the diagnosis of RD in small biopsies in which because of sampling errors a nonspecific histology and immunophenotype may be encountered. Awareness of this diagnostic challenge is essential in establishing the correct diagnosis.
Cutaneous epitheloid angiomatous nodule and epitheloid hemangioma: Distinct entities on the epitheloid vascular tumors spectrum? Altea Esteve Martinez, MD, Hospital General Universitario Valencia, Valencia, Spain; Ana Garcıa Rabasco, DO, Hospital General Universitario de Valencia, Valencia, Spain; Jose Luis Sanchez Carazo, DO, Hospital General Universitario de Valencia, Valencia, Spain; Ram on Garcıa Ruiz, DO, Hospital General Universitario de VAlencia, Valencia, Spain; Victor Alegre de Miquel, DO, Hospital General Universitario de VAlencia, Valencia, Spain We report a case of cutaneous epitheloid angiomatous nodule (CEAN) and a review of four cases of epitheloid hemangioma (EH), two entities sharing histologic findings on the benign pole of vascular tumours. A 56-year-old man presented with a rapidly growing asymptomatic subcutaneous firm nodule on the cubital side of the second finger of his right hand. A MRI showed an increased vascular component with tortuous features. Biopsy performed exhibited a solid proliferation of large epitheloid endothelial cells on the deep dermis with wide eosinophilic cytoplasm and enlarged vesicular nuclei with prominent nucleoli. No atypical cells were found. Disperse within the proliferation were open channels with single cell-lined epitheloid endothelium. Inmunohistochemically the tumor stained with CD34, CD31, actine, and vimentine. CK and S100 were negative. Ki67 showed a low proliferation rate. CEAN diagnosis was made, and the tumor was excised. Four cases of HE were also reviewed. All were women of middle age, with their lesions found on the face and neck. Two of them presented with multiple tumors and the other two solitary lesions. One had eosinophilia and other one presented with locoregional adenophaty. All the lesions were excised with only one recurrence after 10 years. Histologically, all the proliferations showed typical features of EH with a vascular proliferation with prominent endothelial cells protruding in to the lumina and a diffuse infiltrate of lymphocytes and eosinophils. CEAN was described by Brenn in 1984. Currently there are 30 cases in the literature. Some authors defend that because of its clinical presentation and different histologic features, it is an independent entity of EH. Other authors state that it is a rare variant of EH because they have overlapping features. Our case differs both clinically and histologically from the lesions of EH reviewed, leading us to believe they are two entities within the same epitheloid vascular benign tumours spectrum. The recognition of those lesions and distinguishing them from malignant vascular neoplasms is important.
Commercial support: None identified.
Commercial support: None identified.
P1513
AB68
J AM ACAD DERMATOL
FEBRUARY 2011
P1514
Localized cutaneous eruption with enlarged histiocytes induced by subcutaneous granulocyte macrophage colonyestimulating factor injections Matthew Hazey, MD, University of Louisville, Lousville, KY, United States; Jeffrey Callen, MD, University of Louisville, Louisville, KY, United States; Soon Bahrami, MD, University of Louisville, Louisville, KY, United States
Hypocellular plaque-like CD34-positive dermal fibroma (medallion-like dermal dendrocyte hamartoma) Lorea Bagazgoitia, MD, Hospital Ram on y Cajal, Madrid, Spain; Carmen Moreno, MD, Hospital Ram on y Cajal, Madrid, Spain; Pedro Jaen, MD, PhD, Hospital Ram on y Cajal, Madrid, Spain
Background: Granulocyte macrophage colonyestimulating factor (GM-CSF) and granulocyte colonyestimulating factor (G-CSF) have been associated with the development of localized and diffuse cutaneous eruptions. Previous reports of diffuse eruptions coinciding with their infusion have proposed that an increased number of enlarged dermal macrophages on histopathology suggests the etiologic role of these cytokines in the reaction. We report a 70-year-old woman who developed a localized hypersensitivity reaction at the site of subcutaneous GM-CSF injections. Biopsy specimens showed enlarged histiocytes within the dermal infiltrate, suggesting that this finding may signify etiology in both localized and diffuse eruptions caused by these agents. Observation: A 70-year-old white woman with history of T3bN1bM1a melanoma of the left lower extremity was referred for evaluation of a red, pruritic rash on her abdomen. She had recently been enrolled in a clinical trial for treatment of her melanoma, and the eruption began approximately 1 week after starting daily subcutaneous injections of GM-CSF into her abdominal wall as per the trial protocol. She denied any other new medications and was not being treated with other chemotherapy as part of the clinical trial. The physical examination revealed multiple erythematous, pseudovesiculated plaques with surrounding ecchymoses localized to the central and right abdomen. Biopsy specimens showed an eosinophilpredominant, pandermal inflammatory infiltrate and marked dermal edema, suggesting a hypersensitivity reaction. In addition, numerous plump histiocytes were present interstitially amongst the collagen bundles. Her eruption resolved with Clobetasol cream and cessation of GM-CSF injections.
Plaque-like CD34+ dermal fibroma (PDF) or (medallion-like dermal dendrocyte hamartoma) is a recently described congenital dermal neoplasm. Only 11 cases have been reported in the English literature, and its clinical and pathologic manifestations are not completely defined. We present the case of a 20-year-old man presenting with a round, erythematous, atrophic plaque on the midline of the anterior aspect of the neck. The lesion was asymptomatic and had been stable since birth. The histologic examination revealed a band-like hypocelullar fibrotic area in the superficial reticular dermis that did not spread to the deep reticular dermis and subcutaneous tissue. The fibrotic background and the scattered cells were CD34+ and S-100e. Embebed in this fibrotic area there were abundant capilar vessels without lost of elastic fibers as were demonstrated with the orcein stain. Clinical differential diagnosis includes atrophic DFSP, anetoderma, or aplasia cutis. Nevertheless, the morphologic features closely resembled those of a scar or an striae distensae. Although the histologic findings were not identical to those described recently as characteristic, the clinical features were suggestive enough to make the diagnosis of PDF. In our opinion, the PDF spectrum might include lesions with variable cellular density which can show similar clinical manifestations. Commercial support: None identified.
Comment: Because patient populations treated with GM-CSF and G-CSF are typically also being treated with various chemotherapy and antimicrobial agents, determining the etiology of a suspected drug-induced cutaneous eruption can be challenging. Our case provides further evidence that increased numbers of large, dermal histiocytes may implicate these cytokines as the inciting agent. Furthermore, the case illustrates that this histologic finding can be seen in localized reactions related to subcutaneous injections and more diffuse eruptions related to systemic infusion. Commercial support: None identified.
P1515
Pitfalls in the diagnosis of cutaneous RosaieDorfman disease: Report on two cases Vineet Mishra, MD, University of Alabama at Birmingham, Birmingham, AL, United States; Aleodor Andea, MD, University of Alabama at Birmingham, Birmingham, AL, United States; Boni Elewski, MD, University of Alabama at Birmingham, Birmingham, AL, United States; Carlo Gavino, MD, University of Alabama at Birmingham, Birmingham, AL, United States RosaieDorfman disease (RD) is a rare entity; therefore, atypical histologic presentations may pose diagnostic problems. We present two cases with a unique diagnostic challenge because of the lack of characteristic histologic features in the initial punch biopsy specimens. In both cases, the patients presented with a hyperpigmented, indurated plaque on the extremity. Punch biopsies were performed showing a mixed dermal infiltrate with abundant histiocytes that was negative for S100 and microorganisms both on special stains (GMS and AFB) and tissue cultures. Given these findings, a diagnosis of an atypical xanthogranuloma was given. Several months later, excisional biopsies were performed showing the classic histology of RD with a dense dermal infiltrate composed of large foamy cells exhibiting emperipolesis in a background of mixed acute and chronic inflammation. The foamy cells decorated with CD68 and S100 but were negative for CD1a. The diagnosis of cutaneous RD was subsequently sealed. In conclusion, our study documents a pitfall in the diagnosis of RD in small biopsies in which because of sampling errors a nonspecific histology and immunophenotype may be encountered. Awareness of this diagnostic challenge is essential in establishing the correct diagnosis.
Cutaneous epitheloid angiomatous nodule and epitheloid hemangioma: Distinct entities on the epitheloid vascular tumors spectrum? Altea Esteve Martinez, MD, Hospital General Universitario Valencia, Valencia, Spain; Ana Garcıa Rabasco, DO, Hospital General Universitario de Valencia, Valencia, Spain; Jose Luis Sanchez Carazo, DO, Hospital General Universitario de Valencia, Valencia, Spain; Ram on Garcıa Ruiz, DO, Hospital General Universitario de VAlencia, Valencia, Spain; Victor Alegre de Miquel, DO, Hospital General Universitario de VAlencia, Valencia, Spain We report a case of cutaneous epitheloid angiomatous nodule (CEAN) and a review of four cases of epitheloid hemangioma (EH), two entities sharing histologic findings on the benign pole of vascular tumours. A 56-year-old man presented with a rapidly growing asymptomatic subcutaneous firm nodule on the cubital side of the second finger of his right hand. A MRI showed an increased vascular component with tortuous features. Biopsy performed exhibited a solid proliferation of large epitheloid endothelial cells on the deep dermis with wide eosinophilic cytoplasm and enlarged vesicular nuclei with prominent nucleoli. No atypical cells were found. Disperse within the proliferation were open channels with single cell-lined epitheloid endothelium. Inmunohistochemically the tumor stained with CD34, CD31, actine, and vimentine. CK and S100 were negative. Ki67 showed a low proliferation rate. CEAN diagnosis was made, and the tumor was excised. Four cases of HE were also reviewed. All were women of middle age, with their lesions found on the face and neck. Two of them presented with multiple tumors and the other two solitary lesions. One had eosinophilia and other one presented with locoregional adenophaty. All the lesions were excised with only one recurrence after 10 years. Histologically, all the proliferations showed typical features of EH with a vascular proliferation with prominent endothelial cells protruding in to the lumina and a diffuse infiltrate of lymphocytes and eosinophils. CEAN was described by Brenn in 1984. Currently there are 30 cases in the literature. Some authors defend that because of its clinical presentation and different histologic features, it is an independent entity of EH. Other authors state that it is a rare variant of EH because they have overlapping features. Our case differs both clinically and histologically from the lesions of EH reviewed, leading us to believe they are two entities within the same epitheloid vascular benign tumours spectrum. The recognition of those lesions and distinguishing them from malignant vascular neoplasms is important.
Commercial support: None identified.
Commercial support: None identified.
P1513
AB68
J AM ACAD DERMATOL
FEBRUARY 2011