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1型神经纤维瘤病患者的妊娠情况:一项基于登记处的回顾性全人群研究。

The pregnancy in neurofibromatosis 1: A retrospective register-based total population study.

作者信息

Leppävirta Jussi, Kallionpää Roope A, Uusitalo Elina, Vahlberg Tero, Pöyhönen Minna, Timonen Susanna, Peltonen Juha, Peltonen Sirkku

机构信息

University of Turku, Turku, Finland.

Turku University Hospital, Turku, Finland.

出版信息

Am J Med Genet A. 2017 Oct;173(10):2641-2648. doi: 10.1002/ajmg.a.38372. Epub 2017 Aug 16.

DOI:10.1002/ajmg.a.38372
PMID:28815922
Abstract

The objective of this retrospective total population study was to form a view of the pregnancies of the patients with neurofibromatosis type 1 (NF1). A cohort of 1,410 Finnish patients with NF1 was acquired by searching NF1-related inpatient and outpatient hospital visits and confirming the diagnoses by reviewing the medical records. Ten matched control persons per patient with NF1 were collected from Population Register Centre. Study persons were linked to data from Medical Birth Register and Care Register for Health Care through the personal identity code. Cesarean deliveries, hypertension/preeclampsia, and placental abruptions were more common among mothers with NF1 with adjusted odds ratios of 2.24 (95%CI 1.63-3.07), 1.96 (95%CI 1.18-3.24), and 13.40 (95%CI 4.26-42.13), respectively. The adjusted mean pregnancy duration was 0.65 (95%CI 0.42-0.88) weeks shorter among the mothers with NF1 than in the control group consisting of non-NF1 mothers giving birth to a non-NF1 child. The pregnancies of non-NF1 mothers giving birth to a NF1 child were 0.43 (95%CI 0.24-0.62) weeks shorter than in the control group. In summary, NF1 of the mother was associated with a shortened pregnancy and increased pregnancy complications. Also, the NF1 of the fetus slightly shortened pregnancy. Since mothers with NF1 are at increased risk for pregnancy complications, careful evaluation of their pregnancies is warranted.

摘要

这项回顾性全人群研究的目的是了解1型神经纤维瘤病(NF1)患者的妊娠情况。通过搜索与NF1相关的住院和门诊就诊记录,并查阅病历以确认诊断,纳入了1410名芬兰NF1患者队列。从人口登记中心为每名NF1患者收集10名匹配的对照者。研究对象通过个人身份代码与医疗出生登记册和医疗保健护理登记册的数据相链接。NF1母亲的剖宫产、高血压/先兆子痫和胎盘早剥更为常见,调整后的优势比分别为2.24(95%CI 1.63 - 3.07)、1.96(95%CI 1.18 - 3.24)和13.40(95%CI 4.26 - 42.13)。与生育非NF1孩子的非NF1母亲组成的对照组相比,NF1母亲的调整后平均妊娠时长缩短了0.65(95%CI 0.42 - 0.88)周。生育NF1孩子的非NF1母亲的妊娠时长比对照组缩短了0.

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