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The papule had appeared 2 years earlier on the right flank and was slightly erythematous and painful&#46; It was not associated with signs of bleeding or growth&#46; A clinical diagnosis of angioleiomyoma and angiolipoma was considered and an excision biopsy of the lesion was performed&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">Histology revealed an elevated sessile lesion due to the presence of a solid&#44; well circumscribed&#44; nodular tumor with lobular edges&#44; located on the superficial third of the reticular dermis &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>A&#41;&#46; The lesion consisted of fusiform and epithelioid cells with an oval or rounded nucleus&#44; with no significant atypia&#44; variable quantities of cytoplasm&#44; poorly defined edges&#44; and practically no interstitial component &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Figs&#46; 1</a>B-D&#41;&#46; A maximum of 4 mitotic figures for every 10 fields at a magnification of 40&#215;&#46; No tumor necrosis was observed&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">The cancerous population showed diffuse and generalized positive immune staining for S100 and EMA &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41; and was focal for smooth muscle actin &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Figs&#46; 3</a>C and <a class="elsevierStyleCrossRef" href="#fig0015">3</a>D&#41; and caldesmon&#46; No immune staining was observed for cytokeratins AE1-AE3 &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Figs&#46; 3</a>A and <a class="elsevierStyleCrossRef" href="#fig0015">3</a>B&#41;&#44; Melan-A&#44; desmin&#44; glial fibrillary acidic protein&#44; p63&#44; or claudin-1&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">An RT-PCR and EWSR1 rearrangement &#40;EWSR1-POUF5F1&#44; EWSR1-ZNF444 y EWSR1-PBX1&#41; sequencing study was performed<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> and the results were negative&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">Syncytial myoepithelioma is a rare tumor that presents clinically as a papule or nodule on the limbs of middle-aged men&#46; Histologically&#44; it is a solid tumor with fusiform cells or histiocytes with a pale eosinophilic cytoplasm and vesicular nucleus&#44; with sparse stroma&#46;<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5&#44;6</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">It does not usually present mitosis&#44; necrosis or lymphovascular invasion&#44; but in rare cases&#44; up to 4 mitotic figures per 10 fields at 40&#215; have been reported&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> The diagnostic criteria for cutaneous myoepithelial carcinoma are not well established&#44; but tumors with marked cytologic atypia&#44; a high mitotic index&#44; and necrosis show more aggressive behavior&#44; with increased probability of recurrence and distant metastasis&#46;<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5&#44;7&#8211;9</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">Fifty percent of cutaneous syncytial myoepitheliomas present rearrangement of the EWSR1 gene&#46;<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4&#44;5&#44;7</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Myoepithelial tumors usually express cytokeratins and the S100 protein&#46; Myoepithelioma&#44; however&#44; presents positive immune staining for EMA and S100 protein&#44; and most cases are negative for cytokeratins&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">The differential diagnosis includes epithelioid benign fibrous histiocytoma&#44; juvenile xanthogranuloma&#44; melanocytic lesions&#44; and epithelioid sarcoma&#46;<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5&#44;9</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">Epithelioid benign fibrous histiocytoma presents as a dermal nodule of epithelioid cells&#44; frequently with binucleation&#44; positive for EMA&#44; with a fibrovascular stroma&#46; This lesion&#44; however&#44; does not present a syncytial architecture or positive immune staining for S100 protein&#44; or for GFAP or p63&#44; as is the case with syncytial myoepithelioma&#46;<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4&#44;7&#44;9</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">In the early stages&#44; juvenile xanthogranuloma presents as an exophytic lesion with eosinophilic histiocytes that does not usually present mononucleated or multinucleated lipidized cells &#40;Touton cells&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> However&#44; it tends to affect children&#44; immune staining is positive for CD163&#44; CD68&#44; and is negative for EMA and S100 proteins&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0065" class="elsevierStylePara elsevierViewall">Cutaneous syncytial myoepithelioma consists of a combination of epithelioid cells&#44; histiocytes&#44; and fusiform cells&#46; Histology of Spitz nevus shows nests of melanocytic cells with frequent maturation with descent and no syncytial architecture&#46; Both tumors are positive for S100&#44; but positivity for Melan A&#44; HMB-45&#44; and MiTF&#44; and negativity for EMA and GFAP favor a diagnosis of Spitz nevus&#46;<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4&#44;5&#44;7&#44;9</span></a></p><p id="par0070" class="elsevierStylePara elsevierViewall">Epithelioid sarcoma usually presents a combination of epithelioid and fusiform cells with cellular atypia and infiltrative growth with frequent satellite nodules&#46; In some cases&#44; however&#44; it presents with a pattern of mild atypia&#46; Both lesions are positive for EMA&#44; but epithelioid sarcoma is positive for cytokeratins and CD34&#44; negative for myoepithelial markers such as S100 proteins and GFAP&#44; and often presents loss of immune staining for INI-1&#46;<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5&#44;7&#44;9</span></a></p><p id="par0075" class="elsevierStylePara elsevierViewall">The differential diagnosis of a painful skin lesion includes angiolipoma&#44; neuroma&#44; glomic tumor&#44; schwannoma&#44; leiomyoma&#44; eccrine spiradenoma&#44; and dermatofibroma&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a></p><p id="par0080" class="elsevierStylePara elsevierViewall">Treatment of cutaneous syncytial myoepithelioma involves wide resection&#46; Our patient presented a good clinical course with no evidence of local recurrence or metastasis in follow-up examinations&#44; in line with the information reported in the literature&#46;</p><p id="par0085" class="elsevierStylePara elsevierViewall">In conclusion&#44; we report a case of painful cutaneous syncytial myoepithelioma&#46; This is a very rare benign skin tumor&#46; Its clinical presentation is nonspecific and diagnosis requires a histopathology examination and a complete immunohistochemistry panel to differentiate it from other lesions associated with a worse outcome&#46; The presence of rearrangement of the EWSR1 gene may aid the diagnosis&#46; Treatment involves wide resection of the lesion&#44; with an excellent prognosis&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflicts of Interest</span><p id="par0090" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest&#46;</p></span></span>"
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Case and Research Letters
Painful Cutaneous Syncytial Myoepithelioma: From Nonspecific Symptoms to Histopathologic Diagnosis
Mioepitelioma sincitial cutáneo doloroso: desde la clínica inespecífica al diagnóstico histopatológico
S. Guillen-Climenta,
Corresponding author
santigc85@gmail.com

Corresponding author.
, B. Vazqueza, M.I. Pinazoa, C. Monteagudob,c
a Servicio de Dermatología, Hospital Clínico Universitario, Valencia, Spain
b Servicio de Anatomía Patológica, Hospital Clínico Universitario, Valencia, Spain
c Departamento de Patología, Facultad de Medicina, Universidad de Valencia, Valencia, Spain
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The papule had appeared 2 years earlier on the right flank and was slightly erythematous and painful&#46; It was not associated with signs of bleeding or growth&#46; A clinical diagnosis of angioleiomyoma and angiolipoma was considered and an excision biopsy of the lesion was performed&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">Histology revealed an elevated sessile lesion due to the presence of a solid&#44; well circumscribed&#44; nodular tumor with lobular edges&#44; located on the superficial third of the reticular dermis &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>A&#41;&#46; The lesion consisted of fusiform and epithelioid cells with an oval or rounded nucleus&#44; with no significant atypia&#44; variable quantities of cytoplasm&#44; poorly defined edges&#44; and practically no interstitial component &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Figs&#46; 1</a>B-D&#41;&#46; A maximum of 4 mitotic figures for every 10 fields at a magnification of 40&#215;&#46; No tumor necrosis was observed&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">The cancerous population showed diffuse and generalized positive immune staining for S100 and EMA &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41; and was focal for smooth muscle actin &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Figs&#46; 3</a>C and <a class="elsevierStyleCrossRef" href="#fig0015">3</a>D&#41; and caldesmon&#46; No immune staining was observed for cytokeratins AE1-AE3 &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Figs&#46; 3</a>A and <a class="elsevierStyleCrossRef" href="#fig0015">3</a>B&#41;&#44; Melan-A&#44; desmin&#44; glial fibrillary acidic protein&#44; p63&#44; or claudin-1&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">An RT-PCR and EWSR1 rearrangement &#40;EWSR1-POUF5F1&#44; EWSR1-ZNF444 y EWSR1-PBX1&#41; sequencing study was performed<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> and the results were negative&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">Syncytial myoepithelioma is a rare tumor that presents clinically as a papule or nodule on the limbs of middle-aged men&#46; Histologically&#44; it is a solid tumor with fusiform cells or histiocytes with a pale eosinophilic cytoplasm and vesicular nucleus&#44; with sparse stroma&#46;<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5&#44;6</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">It does not usually present mitosis&#44; necrosis or lymphovascular invasion&#44; but in rare cases&#44; up to 4 mitotic figures per 10 fields at 40&#215; have been reported&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> The diagnostic criteria for cutaneous myoepithelial carcinoma are not well established&#44; but tumors with marked cytologic atypia&#44; a high mitotic index&#44; and necrosis show more aggressive behavior&#44; with increased probability of recurrence and distant metastasis&#46;<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5&#44;7&#8211;9</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">Fifty percent of cutaneous syncytial myoepitheliomas present rearrangement of the EWSR1 gene&#46;<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4&#44;5&#44;7</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Myoepithelial tumors usually express cytokeratins and the S100 protein&#46; Myoepithelioma&#44; however&#44; presents positive immune staining for EMA and S100 protein&#44; and most cases are negative for cytokeratins&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">The differential diagnosis includes epithelioid benign fibrous histiocytoma&#44; juvenile xanthogranuloma&#44; melanocytic lesions&#44; and epithelioid sarcoma&#46;<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5&#44;9</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">Epithelioid benign fibrous histiocytoma presents as a dermal nodule of epithelioid cells&#44; frequently with binucleation&#44; positive for EMA&#44; with a fibrovascular stroma&#46; This lesion&#44; however&#44; does not present a syncytial architecture or positive immune staining for S100 protein&#44; or for GFAP or p63&#44; as is the case with syncytial myoepithelioma&#46;<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4&#44;7&#44;9</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">In the early stages&#44; juvenile xanthogranuloma presents as an exophytic lesion with eosinophilic histiocytes that does not usually present mononucleated or multinucleated lipidized cells &#40;Touton cells&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> However&#44; it tends to affect children&#44; immune staining is positive for CD163&#44; CD68&#44; and is negative for EMA and S100 proteins&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0065" class="elsevierStylePara elsevierViewall">Cutaneous syncytial myoepithelioma consists of a combination of epithelioid cells&#44; histiocytes&#44; and fusiform cells&#46; Histology of Spitz nevus shows nests of melanocytic cells with frequent maturation with descent and no syncytial architecture&#46; Both tumors are positive for S100&#44; but positivity for Melan A&#44; HMB-45&#44; and MiTF&#44; and negativity for EMA and GFAP favor a diagnosis of Spitz nevus&#46;<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4&#44;5&#44;7&#44;9</span></a></p><p id="par0070" class="elsevierStylePara elsevierViewall">Epithelioid sarcoma usually presents a combination of epithelioid and fusiform cells with cellular atypia and infiltrative growth with frequent satellite nodules&#46; In some cases&#44; however&#44; it presents with a pattern of mild atypia&#46; Both lesions are positive for EMA&#44; but epithelioid sarcoma is positive for cytokeratins and CD34&#44; negative for myoepithelial markers such as S100 proteins and GFAP&#44; and often presents loss of immune staining for INI-1&#46;<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5&#44;7&#44;9</span></a></p><p id="par0075" class="elsevierStylePara elsevierViewall">The differential diagnosis of a painful skin lesion includes angiolipoma&#44; neuroma&#44; glomic tumor&#44; schwannoma&#44; leiomyoma&#44; eccrine spiradenoma&#44; and dermatofibroma&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a></p><p id="par0080" class="elsevierStylePara elsevierViewall">Treatment of cutaneous syncytial myoepithelioma involves wide resection&#46; Our patient presented a good clinical course with no evidence of local recurrence or metastasis in follow-up examinations&#44; in line with the information reported in the literature&#46;</p><p id="par0085" class="elsevierStylePara elsevierViewall">In conclusion&#44; we report a case of painful cutaneous syncytial myoepithelioma&#46; This is a very rare benign skin tumor&#46; Its clinical presentation is nonspecific and diagnosis requires a histopathology examination and a complete immunohistochemistry panel to differentiate it from other lesions associated with a worse outcome&#46; The presence of rearrangement of the EWSR1 gene may aid the diagnosis&#46; Treatment involves wide resection of the lesion&#44; with an excellent prognosis&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflicts of Interest</span><p id="par0090" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest&#46;</p></span></span>"
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Article information
ISSN: 15782190
Original language: English
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2024 January 62 35 97
2023 December 49 18 67
2023 November 71 27 98
2023 October 53 24 77
2023 September 68 30 98
2023 August 39 9 48
2023 July 51 34 85
2023 June 48 21 69
2023 May 58 36 94
2023 April 44 15 59
2023 March 58 29 87
2023 February 49 25 74
2023 January 41 26 67
2022 December 64 46 110
2022 November 34 39 73
2022 October 32 22 54
2022 September 30 36 66
2022 August 30 23 53
2022 July 17 34 51
2022 June 18 25 43
2022 May 55 41 96
2022 April 47 25 72
2022 March 43 49 92
2022 February 50 30 80
2022 January 51 61 112
2021 December 46 35 81
2021 November 44 46 90
2021 October 57 55 112
2021 September 38 40 78
2021 August 42 32 74
2021 July 63 33 96
2021 June 33 26 59
2021 May 47 35 82
2021 April 84 61 145
2021 March 60 35 95
2021 February 55 27 82
2021 January 40 16 56
2020 December 28 19 47
2020 November 21 15 36
2020 October 32 4 36
2020 September 29 13 42
2020 August 32 17 49
2020 July 20 10 30
2020 June 21 18 39
2020 May 29 17 46
2020 April 53 17 70
2020 March 9 11 20
2020 February 2 3 5
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¿Es usted profesional sanitario apto para prescribir o dispensar medicamentos?

Are you a health professional able to prescribe or dispense drugs?