Tufts University School of Medicine, Boston, Massachusetts, USA.
Department of Dermatology, Boston Children's Hospital, Harvard Medical School, Boston, Massachusetts, USA.
Pediatr Dermatol. 2024 Jan-Feb;41(1):76-79. doi: 10.1111/pde.15353. Epub 2023 Jul 24.
Encephalocraniocutaneous lipomatosis (ECCL) is a rare neurocutaneous disorder caused by somatic FGFR1 and KRAS variants. It shares significant phenotypic overlap with several closely related disorders caused by mutations in the RAS-MAPK pathway (mosaic RASopathies). We report a diagnostically challenging case of ECCL in which next-generation sequencing of affected tissue identified a pathologic FGFR1 p.K656E variant, thereby establishing a molecular diagnosis. Patients with FGFR1-associated ECCL carry a risk of developing malignant brain tumors; thus, genetic testing of patients with suspected ECCL has important management implications.
脑-颅-皮脂肪增多症(ECCL)是一种由体细胞 FGFR1 和 KRAS 变异引起的罕见神经皮肤疾病。它与 RAS-MAPK 通路(嵌合 RAS 病)突变引起的几种密切相关的疾病有显著的表型重叠。我们报告了一例诊断具有挑战性的 ECCL 病例,受影响组织的下一代测序确定了病理性 FGFR1 p.K656E 变异,从而建立了分子诊断。携带 FGFR1 相关 ECCL 的患者有发展恶性脑肿瘤的风险;因此,对疑似 ECCL 的患者进行基因检测具有重要的管理意义。