Biallelic NF1 inactivation in high grade serous ovarian cancers from patients with neurofibromatosis type 1

被引:0
作者
Eliza Courtney
Sock Hoai Chan
Shao Tzu Li
Diana Ishak
Khurshid Merchant
Tarryn Shaw
Wen Yee Chay
Felicia Hui Xian Chin
Wai Loong Wong
Adele Wong
Joanne Ngeow
机构
[1] National Cancer Centre Singapore,Cancer Genetics Service, Division of Medical Oncology
[2] KK Women’s and Children’s Hospital,Department of Pathology and Laboratory Medicine
[3] KK Women’s and Children’s Hospital,KK Gynaecological Cancer Centre
[4] Nanyang Technological University,Lee Kong Chian School of Medicine
来源
Familial Cancer | 2020年 / 19卷
关键词
Neurofibromatosis; NF1; Neurofibromin; Ovarian; Cancer;
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摘要
Neurofibromatosis type 1 (NF1) is a multisystem disorder caused by germline heterozygous NF1 loss-of-function variants. The NF1 gene encodes neurofibromin, a RAS GTPase-activating protein, which functions by down-regulating RAS/RAF/MAPK-signalling pathways. Somatic NF1 aberrations frequently occur in sporadic ovarian cancer (OC), but the incidence of OC in NF1 patients is rare. Here we report the germline and somatic findings for two unrelated patients with NF1 and high-grade serous OC. Germline testing revealed a heterozygous NF1 pathogenic variant in each patient, c.7096_7101del (p.Asn2366_Phe2367del) and c.964delA (p.Ile322Leufs*54), respectively. No germline variants in well-established OC predisposition genes were detected, including BRCA1 and BRCA2. Tumor loss-of-heterozygosity analysis demonstrated loss of the wild type NF1 allele for both patients. Biallelic NF1 inactivation occurs as part of OC pathogenesis in NF1 patients. Although the penetrance of NF1-associated OC is insufficient to warrant risk-reducing interventions, our findings highlight the potential for therapies targeting the RAS/RAF/MAPK-signalling pathway for these cases.
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页码:353 / 358
页数:5
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[1]  
Evans DG(2010)Birth incidence and prevalence of tumor-prone syndromes: estimates from a UK family genetic register service Am J Med Genet Part A 152A 327-332
[2]  
Howard E(2015)Incidence and mortality of neurofibromatosis: a total population study in Finland J Invest Dermatol 135 904-906
[3]  
Giblin C(2013)Neurofibromatosis type 1 (NF1): a diagnosis and management Handb Clin Neurol 115 939-955
[4]  
Uusitalo E(1988)Von recklinghausen neurofibromatosis: a clinical and population study in south-east Wales Brain 111 1355-1381
[5]  
Leppävirta J(2007)Neurofibromatosis 1 and neurofibromatosis 2: a twenty first century perspective Lancet Neurol 6 340-351
[6]  
Koffert A(2012)Pediatric plexiform neurofibromas: impact on morbidity and mortality in neurofibromatosis type 1 J Pediatr 160 461-467
[7]  
Ferner RE(2007)Prevalência de neurofibromas plexiformes em crianças e adolescentes com neurofibromatose tipo 1 J Pediatr (Rio J) 83 571-573
[8]  
Gutmann DH(1997)Type 1 neurofibromatosis: a descriptive analysis of the disorder in 1,728 patients Am J Med Genet 70 138-143
[9]  
Huson SM(1993)Neurofibromatosis type 1: Review of the first 200 patients in an Australian clinic J Child Neurol 8 395-402
[10]  
Harper PS(2012)Quantitative assessment of whole-body tumor burden in adult patients with neurofibromatosis PLoS One 7 e35711-655