Objective
Neurofibromatosis type 2 (NF2), tuberous sclerosis (TS), and von Hippel-Lindau disease (VHL) are tumor suppressor syndromes characterized by multiple benign tumors of the peripheral and central nervous system. These tumors may lead to an enhanced obstetric risk in female patients, but it is currently unknown whether women with NF2, TS, or VHL experience increased rates of adverse pregnancy outcomes. Current data consist primarily of case series, even the largest of which may lack power because of the small sample sizes. Use of large administrative databases can facilitate studies of pregnancy in rare disorders, as shown by our prior work with neurofibromatosis 1, another neurogenetic tumor suppressor syndrome. Using a population-based database, we found that pregnant women with NF1 experienced increased maternal morbidity, including higher rates of hypertensive and cerebrovascular complications, than the general population. In the current study, we apply a similar methodology to determine the association between NF2, TS, and VHL and adverse pregnancy outcomes.
Study Design
We performed a retrospective cohort study using hospital discharge data from the US Nationwide Inpatient Sample, 1998–2008. International Classification of Diseases , ninth revision, Clinical Modification, codes were used to identify pregnancy-related hospitalizations (codes 630–679) for women with NF2 (237.72), TS (759.5), or VHL (759.6). Patients were compared with a control group of all pregnancy-related hospitalizations not associated with NF1, NF2, TS, or VHL in the same time period. Admissions for women aged younger than 15 or older than 50 years were excluded in both groups. International Classification of Diseases , ninth revision, codes were used to identify preeclampsia, ischemic and hemorrhagic cerebrovascular disease, preterm labor, and cesarean delivery during hospitalization, as previously described. Multivariable logistic regression was used to investigate the risk of adverse outcomes in each patient population, adjusting for age, preexisting comorbidities, race, socioeconomic status, and hospital and insurance characteristics. Statistical analyses were performed using SAS version 9.1 (SAS Institute Inc, Cary, NC). This study was approved by the Partners Institutional Review Board.
Study Design
We performed a retrospective cohort study using hospital discharge data from the US Nationwide Inpatient Sample, 1998–2008. International Classification of Diseases , ninth revision, Clinical Modification, codes were used to identify pregnancy-related hospitalizations (codes 630–679) for women with NF2 (237.72), TS (759.5), or VHL (759.6). Patients were compared with a control group of all pregnancy-related hospitalizations not associated with NF1, NF2, TS, or VHL in the same time period. Admissions for women aged younger than 15 or older than 50 years were excluded in both groups. International Classification of Diseases , ninth revision, codes were used to identify preeclampsia, ischemic and hemorrhagic cerebrovascular disease, preterm labor, and cesarean delivery during hospitalization, as previously described. Multivariable logistic regression was used to investigate the risk of adverse outcomes in each patient population, adjusting for age, preexisting comorbidities, race, socioeconomic status, and hospital and insurance characteristics. Statistical analyses were performed using SAS version 9.1 (SAS Institute Inc, Cary, NC). This study was approved by the Partners Institutional Review Board.
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