Moran Jakob M T, Hoang Mai P, Mariño-Enríquez Adrian, DeSimone Mia S
Department of Pathology, Massachusetts General Hospital, Harvard Medical School, Boston, MA; and.
Department of Pathology, Brigham and Women's Hospital, Harvard Medical School, Boston, MA.
Am J Dermatopathol. 2025 Jan 1;47(1):65-69. doi: 10.1097/DAD.0000000000002831. Epub 2024 Aug 14.
Malignant proliferating pilar tumors (MPPTs) are rare, unique cutaneous adnexal tumors. Sarcomatous transformation in MPPTs is even rarer (4 previous cases reported). Here, we report an extraordinary case of a MPPT with sarcomatous transformation occurring on the scalp of a 63-year-old man with an in-depth molecular profile along with histologic, immunohistochemical, and follow-up data. Shared mutations in the epithelial and sarcomatous components included a loss-of-function TP53 mutation. An inactivating TP53 mutation was only identified in the epithelial component, and an inactivating CDKN2A mutation was only identified in the sarcomatous component. Copy number variations previously reported in MPPT were also identified, including 6p21.1 loss, 6q arm loss, and 15q21.1-q26.3 gain [epithelial], and 6p22.2-p22.3 loss [sarcoma]. Histologically, the tumor demonstrated juxtaposed areas of proliferating pilar tumor, carcinoma with clear cell change, and sarcomatous areas that did not stain for AE1/AE3, p40, CD34, S100 protein, and smooth muscle actin by immunohistochemistry. The patient is alive at 2 years without evidence of recurrence or metastasis.
恶性增殖性毛根鞘瘤(MPPTs)是一种罕见的、独特的皮肤附属器肿瘤。MPPTs发生肉瘤样变更是罕见(此前报道过4例)。在此,我们报告了一例非同寻常的病例,一名63岁男性头皮上发生了伴有肉瘤样变的MPPT,并提供了详细的分子特征以及组织学、免疫组化和随访数据。上皮成分和肉瘤成分中的共同突变包括功能丧失性TP53突变。失活性TP53突变仅在上皮成分中被鉴定出,失活性CDKN2A突变仅在肉瘤成分中被鉴定出。此前在MPPT中报道的拷贝数变异也被识别出来,包括6p21.1缺失、6q臂缺失和15q21.1-q26.3增益[上皮成分],以及6p22.2-p22.3缺失[肉瘤成分]。组织学上,肿瘤表现为增殖性毛根鞘瘤、透明细胞改变的癌和肉瘤样区域并列,免疫组化显示这些肉瘤样区域对AE1/AE3、p40、CD34、S100蛋白和平滑肌肌动蛋白均无染色。该患者存活2年,无复发或转移迹象。