Sharapova Svetlana O, Golovataya Elena I, Shepelevich Elena V, Mareika Yuliya E, Guryanova Irina E, Stegantseva Maria V, Aleinikova Olga V
Research Department, Belarusian Research Center for Pediatric Oncology, Hematology and Immunology, Minsk Region, Belarus.
Prenatal Center, Belarusian Research and Practical Center "Mother and Child", Minsk, Belarus.
Cent Eur J Immunol. 2020;45(4):507-510. doi: 10.5114/ceji.2020.103387. Epub 2021 Jan 30.
Nijmegen breakage syndrome (NBS) is a rare autosomal recessive disorder, characterized by spontaneous chromosomal instability with predisposition to immunodeficiency and cancer. We present a repeated NBS in two sons from one woman after two marriages. We describe the clinical data, cytogenetic, and molecular findings of a prenatally diagnosed fetus, and his brothers with NBS. The first patient developed peripheral T-cell lymphoma at the age of 16 years and died 5 months after the protocol start. The diagnosis of NBS was established after his death. The second patient was born after the fifth pregnancy, third delivery in the second marriage; he developed cortical T-cell leukemia at the age of 3 years, received hematopoietic stem cells transplantation (HSCT) and he is alive now. In a year after repeated NBS case in this family, mother became pregnant again and the mutation was detected in the male fetus after the prenatal diagnosis; the pregnancy was aborted. At the age of 41 years, mother's seventh pregnancy finished by miscarriage. In three months, she was pregnant again, only one mutation in NBN gene was detected during the prenatal diagnostics in the female fetus; healthy female was born at term. To our knowledge, this is the first time to describe the repeated cases of two patients born with Nijmegen breakage syndrome from one mother and two different fathers. This case highlights the value of checking NBN carrier in Belarusian families during genetic counselling.
奈梅亨断裂综合征(NBS)是一种罕见的常染色体隐性疾病,其特征是自发染色体不稳定,易患免疫缺陷和癌症。我们报告了一名女性在两段婚姻后所生的两个儿子均患NBS的病例。我们描述了一名产前诊断胎儿及其患NBS的兄弟的临床数据、细胞遗传学和分子学发现。第一名患者在16岁时患上外周T细胞淋巴瘤,在方案开始后5个月死亡。在他死后确诊为NBS。第二名患者是在第五次怀孕后出生的,是第二次婚姻中的第三次分娩;他在3岁时患上皮质T细胞白血病,接受了造血干细胞移植(HSCT),目前还活着。在这个家庭出现重复的NBS病例一年后,母亲再次怀孕,产前诊断在男性胎儿中检测到突变;此次怀孕终止。母亲41岁时,第七次怀孕以流产告终。三个月后,她再次怀孕,产前诊断在女性胎儿中仅检测到NBN基因的一个突变;足月时生下一名健康女婴。据我们所知,这是首次描述一名母亲与两名不同父亲所生的两名患有奈梅亨断裂综合征患者的重复病例。该病例凸显了在白俄罗斯家庭进行遗传咨询时检查NBN携带者的价值。