Abstract
Background
There is a strong correlation between the level of circulating female sex hormones and the parturient growth of meningiomas. As a result, rapid changes in meningioma size occur during pregnancy, putting both the mother and fetus at risk. Large, symptomatic meningiomas require surgical resection, regardless of the status of pregnancy. However, the preferred timing of such complex intervention is a matter of debate. The rarity of this clinical scenario and the absence of prospective trials make it difficult to reach evidence-based conclusions. The aim of this study was to create evidence-based management guidelines for timing of surgery for pregnancy-related intracranial meningiomas.
Method
The English literature from 1990 to 2016 was systematically reviewed according to PRISMA guidelines for all surgical cases of pregnancy–related intracranial meningiomas. Cases were divided into two groups: patients who have had surgery during pregnancy and delivered thereafter (group A) and patients who delivered first (group B). Groups were compared for demographic, clinical and radiological features, as well as for neurosurgical, obstetrical and neonatological outcomes. Statistical analysis was performed to assess differences.
Results
A total of 104 surgical cases were identified and reviewed, of which 86 were suitable for comparison and statistical analysis. Thirty-five patients (40%) underwent craniotomy for resection during pregnancy or at delivery (group A) and 51 patients (60%) underwent surgery after delivery (group B). Groups showed no significant differences in characteristics such as age at diagnosis, number of gestations, presenting symptoms, tumor site and tumor size. Despite a comparable distribution over the gestational trimesters, group A had significantly more patients diagnosed prior to the 27th gestational week (46 vs 17.5%, p = 0.0075). Group A was also associated with a significantly higher rate of both emergent craniotomies (40 vs 19.6%, p = 0.0048) and emergent Caesarian deliveries (47 vs 17.8%, p = 0.00481). The time from diagnosis to surgery was significantly longer in group B (11 weeks vs 1 week in group A, p = 0.0013). The rate of premature delivery was high but similar in both groups (∼70%). Risks of maternal mortality or fetal mortality were associated with group A (odds ratio = 14.7), but did not reach statistical significance.
Conclusions
While surgical resection of meningioma during pregnancy may be associated with increased maternal and fetal mortalities, the overall neurosurgical, obstetrical and neonatological outcomes, as well as many clinical characteristics, are similar to patients undergoing resection postpartum. We believe that fetal survival chances have a significant impact on decision-making, as patients diagnosed at a later stage in pregnancy (≥27th week of gestation) were more likely to undergo delivery first. This complicated clinical scenario requires the close cooperation of multiple disciplines. While the mother’s health and well-being should always be paramount in guiding management, we hope that the overall good outcomes observed by this systematic review will encourage colleagues to aim for term pregnancies whenever possible in order to reduce prematurity-related problems.
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The authors aimed to perform a meta-analysis of the literature on surgery for intracranial meningiomas during pregnancy, with the ultimate objective of suggesting guidelines for the management of patients in this situation. This is an admirable objective, given the relatively limited data published and complexity of decision-making in this situation. Unfortunately, the published reports are widely diverse, complicating the task. In particular, they lack almost any information about late maternal-child outcomes, the ultimate determinant of whether a given management approach has been successful. The authors nevertheless present a management algorithm for intracranial meningioma during pregnancy (Fig. 1) that looks reasonable, with the caveat that the range between gestational week (GW) 27 and 36 is too wide. There is a vast difference in neonatal outcomes at the two ends of this spectrum, and there is also a fairly rapid increase in risk to the mother for surgery after about GW 28–30. We addressed these issues in some depth in our paper, and they are reviewed elsewhere [1–3]. Based on these issues, we do not feel the current literature is sufficiently robust or homogenous to support the development of evidence-based guidelines.
The central issue in this clinical situation is balancing the well-being of mother and fetus. The authors encourage delaying surgery until after delivery as a general rule, raising the point that Caesarian section is also associated with some risk. In their conclusion, they urge their colleagues to aim for late or term deliveries whenever possible to reduce problems from prematurity. We wish to question this broad recommendation. First, while it is well known that later delivery improves neonatal outcomes, we lack data in this specific situation to support substantial morbidity or mortality for these children. More importantly, this recommendation obviously contradicts our current ethical premise that the mother’s well-being takes first place for caregivers [1, 2].
We are concerned that following a general approach of delaying surgery may in fact increase risks for both mother and baby. In fact, the authors found significant differences in the rates or urgent/emergent craniotomy as well as Caesarian section in cases where craniotomy was performed during Pregnancy. In addition, the two maternal deaths occurred in cases where surgery had been delayed; they were not a direct result of the neurosurgical intervention itself. It is certainly possible that in some cases when tumor growth increases pressure on critical neuroanatomy, delaying surgery can lead to irreparable neurological damage or even herniation. For this reason, we have found that issues relating to the risks of craniotomy during pregnancy versus delay, and decisions regarding how long to allow the pregnancy to continue must be carefully examined in each situation, and we shy away from any broad rule of thumb. Of course we want to do what is possible to allow normal fetal/child development. Surely when possible we should try to get past the gestational week GW 27 hurdle, and then the week GW 30 hurdle, and further, when we feel it can be done safely for the mother.
Another important issue is the central role of the patient. It is not enough to present information to the patient. In fact, the ultimate decision rests with her. In our experience, these women are concerned not only for themselves but also for one or more living children. Given the current ethical premise that protecting the life and well-being of the mother is our highest priority, we feel that only she and her family have the right to choose a significant delay in surgery when she faces the risk of serious impairment or even death.
Sergey Spektor
Jerusalem, Israel
References
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Laviv, Y., Bayoumi, A., Mahadevan, A. et al. Meningiomas in pregnancy: timing of surgery and clinical outcomes as observed in 104 cases and establishment of a best management strategy. Acta Neurochir 160, 1521–1529 (2018). https://doi.org/10.1007/s00701-017-3146-8
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DOI: https://doi.org/10.1007/s00701-017-3146-8