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携带有胚系 PALB2 致病性变异的个体中的非典型脉络膜黑素瘤。

Atypical choroidal nevus in a subject with a germline PALB2 pathogenic variant.

机构信息

Department of Ophthalmology and Visual Sciences, Havener Eye Institute, The Ohio State University Wexner Medical Center, Columbus, OH, USA.

Department of Internal Medicine Division of Human Genetics and James Comprehensive Cancer Center, The Ohio State University Wexner Medical Center, Columbus, OH, USA.

出版信息

Fam Cancer. 2022 Jan;21(1):1-5. doi: 10.1007/s10689-020-00220-2. Epub 2021 Jan 6.

Abstract

Recent evidence suggests that PALB2 variants may increase risk for the development of uveal melanoma and uveal melanocytic neoplasms. Here we report a case of an atypical choroidal nevus in a patient with a personal history of cancer and pathogenic PALB2 germline variant. A 75-year-old white female presented with an elevated predominantly amelanotic choroidal lesion OS. On examination and ophthalmic imaging, the mass measured 8.8 mm × 6.5 mm × 1.5 mm. The mass showed predominantly medium to high reflectivity on diagnostic A-scan and acoustic hollowing on B-scan. OCT over the lesion showed no subretinal fluid. The patient has a personal history of breast cancer and gastric adenoma and a strong family history of cancer. The patient was found to have a pathogenic truncating variant in PALB2 (rs118203998 c.3549C > A, p.Y1183*). Together with our previous findings of pathogenic PALB2 variants in uveal melanoma patients, this new finding of an atypical choroidal nevus in a patient with a pathogenic PALB2 germline variant suggests that pathogenic PALB2 variants may be a risk factor for uveal melanocytic neoplasms. This finding warrants further assessment of the prevalence and progression of uveal melanocytic neoplasms in PALB2 pathogenic variant carriers.

摘要

最近的证据表明,PALB2 变体可能会增加葡萄膜黑色素瘤和葡萄膜黑色素细胞肿瘤的发病风险。在这里,我们报告了一例具有癌症个人史和致病性 PALB2 种系变异的患者的不典型脉络膜痣病例。一位 75 岁的白人女性因 OS 眼出现升高的主要无色素性脉络膜病变而就诊。通过检查和眼科成像,该肿块在 OS 眼的大小为 8.8mm×6.5mm×1.5mm。该肿块在诊断性 A 扫描上显示出中等到高反射率,在 B 扫描上显示出声影空洞。病变上的 OCT 显示没有视网膜下液。该患者有乳腺癌和胃腺瘤的个人病史,以及癌症的强烈家族史。该患者被发现存在 PALB2 中的致病性截断变异(rs118203998 c.3549C > A,p.Y1183*)。结合我们之前在葡萄膜黑色素瘤患者中发现的致病性 PALB2 变异,该新发现的具有致病性 PALB2 种系变异的不典型脉络膜痣表明,致病性 PALB2 变异可能是葡萄膜黑色素细胞肿瘤的一个风险因素。这一发现需要进一步评估 PALB2 致病性变异携带者中葡萄膜黑色素细胞肿瘤的患病率和进展情况。

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