Beatson Meghan E, Smithy James W, Domenico Dylan, Gundem Gunes, Papaemmanouil Elli, Moy Andrea, Chen Joyce M, Pulitzer Melissa P, Ariyan Charlotte, Busam Klaus J
Department of Pathology and Laboratory Medicine, Memorial Sloan Kettering Cancer Center, New York, NY, USA.
Department of Medicine, Memorial Sloan Kettering Cancer Center, New York, NY, USA.
J Cutan Pathol. 2025 Aug;52(8):528-532. doi: 10.1111/cup.14824. Epub 2025 Jun 2.
While early reports of CRTC1::TRIM11 cutaneous tumors suggested a predominantly indolent clinical course, a few cases have been documented with lymph node and distant metastases. We report herein a metastasizing tumor with a lethal outcome. It presented as a subcutaneous mass on the back of a 17-year-old teenager. Histopathologically, the tumor was subepidermal and composed of amelanotic spindle and epithelioid cells with nuclear pleomorphism. The tumor cells were immunoreactive for Sox10 and S100, focally positive for Melan-A, and negative for PRAME. Whole genome and whole transcriptome sequencing revealed a CRTC1::TRIM11 fusion as well as a number of additional genetic and genomic aberrations. No TERT promoter gene mutation was identified. The patient developed metastases to the lung, lymph nodes, and soft tissue. The tumor was refractory to various treatments. The patient died of widely metastatic disease 18 months after clinical presentation. This case expands our knowledge of the clinical, pathologic, and molecular findings of CRTC1::TRIM11 cutaneous tumors. In contrast to other reported cases, the tumor of our patient contained additional genomic aberrations. More cases are needed to assess what factors are relevant for the prognosis of CRTC1::TRIM11 cutaneous tumors to help distinguish those with indolent from those with aggressive behavior.
虽然早期关于CRTC1::TRIM11皮肤肿瘤的报告表明其临床病程主要呈惰性,但已有少数病例记录出现淋巴结转移和远处转移。我们在此报告一例发生转移且导致致命后果的肿瘤。该肿瘤表现为一名17岁青少年背部的皮下肿块。组织病理学检查显示,肿瘤位于表皮下,由无黑色素的梭形细胞和上皮样细胞组成,细胞核具有多形性。肿瘤细胞对Sox10和S100呈免疫反应性,对Melan-A呈局灶性阳性,对PRAME呈阴性。全基因组和全转录组测序揭示了CRTC1::TRIM11融合以及一些其他的基因和基因组畸变。未发现TERT启动子基因突变。患者出现了肺、淋巴结和软组织转移。该肿瘤对各种治疗均耐药。患者在临床表现18个月后死于广泛转移的疾病。本病例扩展了我们对CRTC1::TRIM11皮肤肿瘤的临床、病理和分子学发现的认识。与其他报告的病例不同,我们患者的肿瘤包含额外的基因组畸变。需要更多病例来评估哪些因素与CRTC1::TRIM11皮肤肿瘤的预后相关,以帮助区分惰性肿瘤和侵袭性肿瘤。