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Remote cerebellar hemorrhage after supratentorial procedures (part 1): a systematic review

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Abstract

A remote cerebellar hemorrhage (RCH) is a spontaneous bleeding in the posterior fossa, which may rarely occurs as a complication of supratentorial procedures, and it shows a typical bleeding pattern defined “the zebra sign.” However, its pathophysiology still remains unknown. We performed a comprehensive review collecting all cases of RCH after supratentorial craniotomies reported in literature in order to identify the most frequently associated procedures and the possible risk factors. We assessed percentages of incidence and 95 % confidence intervals of all demographic, neuroradiological, and clinical features of the patients. Univariate and multivariate analyses were used to evaluate their association with outcome. We included 49 articles reporting 209 patients with a mean age of 49.09 ± 17.07 years and a male/female ratio 130/77. A RCH was more frequently reported as a complication of supratentorial craniotomies for intracranial aneurysms, tumors debulking, and lobectomies. In the majority of cases, RCH occurrence was associated with impairment of consciousness, although some patients remained asymptomatic or showed only slight cerebellar signs. Coagulation disorders, perioperative cerebrospinal fluid drainage, hypertension, and seizures were the most frequently reported risk factors. Zebra sign was the most common bleeding pattern, being observed in about 65 % out of the cases, followed by parenchymal hematoma and mixed hemorrhage in similar percentages. A multivariate analysis showed that symptomatic onset and intake of antiplatelets/anticoagulants within a week from surgery were independent predictors of poor outcome. However, about 75 % out of patients showed a good outcome and a RCH often appeared as a benign and self-limiting condition, which usually did not require surgical treatment, but only prolonged clinical surveillance, unless in the event of the occurrence of complications.

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Correspondence to Carmelo Lucio Sturiale.

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Adam Tucker, Osaka, Japan

The authors are to be commended for conducting perhaps the largest review to date including an analysis of 209 patients in 49 articles published during 1977 to 2013. Although many of the findings served to confirm results from prior reviews, this study was unique in that it provided a precise statistical analysis of patient characteristics, risk factors, and outcomes. Of note, better outcomes were observed both in studies published after 2001, probably reflecting recent advances in management, and with the zebra pattern (1.5 % mortality), which was usually bilateral. In contrast, there was an overall 11.7 % mortality, with 80.9 % due to brainstem compression from intracerebellar hemorrhage (ICH). Multivariate logistic regression analysis showed that symptomatic onset, antiplatelet/anticoagulation medication, and older age were independently associated with poor outcome, while in univariate analysis timing of remote cerebellar hemorrhage (RCH) appearance, bilateral localization, and sex were unrelated. Because the respective incidences of coagulation disorders, perioperative CSF drainage, and hypertension did not exceed one third of the cases, these risk factors were considered to play a relatively limited etiological role.

RCH is rare postoperative neurosurgical and orthopedic complication with as yet unknown etiology, however, possibly because of recent advances in both neuroimaging and interventional therapy, this entity is beginning to confront neurologists and stroke specialists as well. Aside from a report of two cases of remote cerebellar hemorrhage from the site of craniotomy published in 2015, which found multiple foci of hemorrhage as an additional adverse prognostic factor, there have been few new studies, especially ones pertaining to the etiology of remote cerebellar hemorrhage. However, recent indirect investigations on post-stroke remote cerebral hemorrhage, in particular a 2014 large prospective cohort, found that previous stroke and higher age were independently associated with remote parenchymal hemorrhage, which occurred in 2.2 % (970) of patients from a total group of 43,494 patients treated with intravenous recombinant tissue-type plasminogen activator. Thus, in addition to classical mechanical factors related to CSF over-drainage, cerebellar sag and venous bleeding or hypertensive parenchymal bleeding factors, these data, as well as neuroimaging diagnostic advances in microbleeds, tend to suggest the possibility of previously unconsidered multiple histopathological conditions such as cerebral amyloid angiopathy (CAA), small vessel microangiopathic processes, and other age-related hemorrhagic and or ischemic contributing factors. Prospective studies with incorporation of susceptibility-weighted imaging or other modalities including tissue diagnosis would be useful for clarifying issues of causative mechanisms, prognosis, and could possibly lead to new treatment avenues.

Currently, there are no established guidelines for management of post-procedural remote cerebellar hemorrhage, though recent reports have recommended preventative measures, such as perioperative blood pressure control, management of coagulopathies, avoidance of excessive CSF drainage and inadvertent durotomies, as well as adequate CSF replacement with watertight closure. Finally, although the authors took the ambitious task of dividing remote hemorrhage into a two part review (remote hemorrhage after supratentorial and spinal procedures), it may be constructive to address the relations between the two phenomena within the second part of their review.

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Jack Jallo, Philadelphia, USA

Dr. Sturiale and his group have written an interesting review paper regarding remote cerebellar hemorrhage after supratentorial procedures, describing the incidence of a relatively under recognized complication in neurosurgery. We hope that this paper will help to shed light on this unpredictable complication and further the community’s understanding for the sake of future treatment and prevention.

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Sturiale, C.L., Rossetto, M., Ermani, M. et al. Remote cerebellar hemorrhage after supratentorial procedures (part 1): a systematic review. Neurosurg Rev 39, 565–573 (2016). https://doi.org/10.1007/s10143-015-0691-6

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