Brainstem gliomas in pregnancy: a systematic review†

Introduction: Although brainstem gliomas are a rare group of neoplasias, when they affect pregnant women, there can be challenges with diagnosis and management. This study describes a case of brainstem glioma diagnosed in pregnancy and systematically reviews the literature on brainstem gliomas in pregnancy to provide guidance for management.Material and methods: We searched five databases from inception until October 2016 using subject headings and keywords related to pregnancy and brainstem glioma, and included original research articles that described pregnancy outcomes in women with brainstem glioma. Data extraction and quality assessment using the Joanna Briggs Institute Critical Appraisal Checklist for case reports were performed in duplicate. Outcomes were reported as proportions. The study protocol was registered with the Prospero International Prospective Register of Systematic Reviews (CRD42017060196).Results: We screened 2737 titles and abstracts, and 89 full-texts. Twelve articles describing 17 pregnancies in 16 women were included in the analysis. The median gestational age at presentation was 23 weeks. All but one case presented with neurologic deficit. Magnetic resonance (MRI) imaging conclusively diagnosed all cases. Surgical tumor resection (n = 4) and radiation therapy (n = 3) were successfully undertaken during pregnancy. There were no reported sequelae of maternal oncological management on neonatal wellbeing. Maternal mortality was high (8/16, 50%) both during (n = 5) and within 4 weeks (n = 3) of pregnancy. Pregnancy losses included one pregnancy termination and four miscarriages (associated with maternal mortality). Of the 12 live-born babies, five were premature. Two of these were the result of spontaneous preterm labor and three were delivered prematurely to facilitate glioma management. There was one case of fetal growth restriction.Conclusions: Although the symptoms of brainstem gliomas often mimic those commonly encountered in pregnancy, neurologic deficits warrant urgent investigation. MRI is the diagnostic modality of choice in pregnancy. Brainstem gliomas are associated with high maternal mortality and appropriate management, including surgical tumor resection and radiation therapy, should not be delayed on account of pregnancy. Pregnancy outcomes are favorable although there is a risk of preterm birth.Key messageBrainstem gliomas are associated with high maternal mortality and timely diagnosis using magnetic resonance imaging and treatment including surgical resection and radiation therapy should not be delayed during pregnancy. Pregnancy outcomes are generally favorable except for risk of preterm birth.