Mankuzhy Nikhil P, Anderson Bailey, Heider Amer, Michniacki Thomas F, Kumar-Sinha Chandan, Mody Rajen
Oakland University William Beaumont School of Medicine, Rochester, MI, USA.
Department of Pediatrics, University of Michigan, Ann Arbor, MI, USA.
Transl Pediatr. 2019 Dec;8(5):449-454. doi: 10.21037/tp.2019.11.05.
Tenosynovial giant cell tumors (TSGCT) are a group of rare, benign soft tissue tumors with common histologic and cytogenetic features, with a median age of diagnosis being 47 years. Generally divided into localized and diffuse subtypes, TSGCTs are typically driven by overexpression of macrophage colony stimulating factor receptor-1 (CSF1R). Treatment of TSGCT is tumor resection, followed by radiation therapy in cases of incomplete resection. Even when the tumor is completely removed, recurrence rates can be as high as 30% in some anatomical locations. Here we report the identification of a previously undescribed KRAS p.G12D activating mutation within a pediatric TSGCT patient, who clinically presented with an enlarging right lower extremity mass pathologically consistent with TSGCT. The patient continues to be in remission three years after complete surgical removal. KRAS mutations are usually found in adult cancers, such as lung and pancreatic, as well as giant cell lesion of the jaw. This case demonstrates the utility of integrative clinical sequencing in identifying lesions with aggressive potential and aiding in complex diagnoses.
腱鞘巨细胞瘤(TSGCT)是一组罕见的良性软组织肿瘤,具有常见的组织学和细胞遗传学特征,诊断的中位年龄为47岁。TSGCT通常分为局限性和弥漫性亚型,通常由巨噬细胞集落刺激因子受体-1(CSF1R)的过表达驱动。TSGCT的治疗方法是肿瘤切除,不完全切除的病例随后进行放射治疗。即使肿瘤被完全切除,在某些解剖部位的复发率仍可高达30%。在此,我们报告在一名儿科TSGCT患者中鉴定出一种先前未描述的KRAS p.G12D激活突变,该患者临床上表现为右下肢肿块增大,病理检查与TSGCT一致。在完全手术切除三年后,该患者仍处于缓解期。KRAS突变通常见于成人癌症,如肺癌和胰腺癌,以及颌骨巨细胞病变。本病例证明了综合临床测序在识别具有侵袭性潜力的病变及辅助复杂诊断方面的实用性。