Familial perimesencephalic subarachnoid hemorrhage: two case reports.

Ulaş Cıkla,Guner Menekse,Mustafa K Başkaya,Patrick A Turski,David B Niemann,Beverly Aagaard-Kienitz

doi:10.1186/1752-1947-8-380

Abstract

IntroductionNon-aneurysmal spontaneous subarachnoid hemorrhage is characterized by an accumulation of a limited amount of subarachnoid hemorrhage, predominantly around the midbrain, and a lack of blood in the brain parenchyma or ventricular system. It represents 5% of all spontaneous subarachnoid hemorrhage cases. In spite of extensive investigation, understanding of the mechanisms leading to perimesencephalic non-aneurysmal subarachnoid hemorrhage remains incompletely defined. A growing body of evidence has supported a familial predisposition for non-aneurysmal spontaneous subarachnoid hemorrhage.Case presentationA 39-year-old Caucasian man presented with sudden onset headache associated with diplopia. His computed tomography scan revealed perimesencephalic subarachnoid hemorrhage. A cerebral angiogram showed no apparent source of bleeding. He was treated conservatively and discharged after 1 week without any neurological deficits. The older brother of the first case, a 44-year-old Caucasian man, presented 1.5 years later with acute onset of headache and his computed tomography scan also showed perimesencephalic non-aneurysmal subarachnoid hemorrhage. He was discharged home with normal neurological examination 1 week later. Follow-up angiograms did not reveal any source of bleeding in either patient.ConclusionsWe report the cases of two siblings with perimesencephalic non-aneurysmal subarachnoid hemorrhage, which may further suggest a familial predisposition of non-aneurysmal spontaneous subarachnoid hemorrhage and may also point out the possible higher risk of perimesencephalic non-aneurysmal subarachnoid hemorrhage in the first-degree relatives of patients with perimesencephalic non-aneurysmal subarachnoid hemorrhage.

Highlights

Non-aneurysmal spontaneous subarachnoid hemorrhage is characterized by an accumulation of a limited amount of subarachnoid hemorrhage, predominantly around the midbrain, and a lack of blood in the brain parenchyma or ventricular system
We report the cases of two siblings with perimesencephalic non-aneurysmal subarachnoid hemorrhage, which may further suggest a familial predisposition of non-aneurysmal spontaneous subarachnoid hemorrhage and may point out the possible higher risk of perimesencephalic non-aneurysmal subarachnoid hemorrhage in the first-degree relatives of patients with perimesencephalic non-aneurysmal subarachnoid hemorrhage
P-non-aneurysmal subarachnoid hemorrhage (NSAH) has generally been considered a distinct entity after the identification of different prognoses from the other NSAH by van Gijn et al [2]

Summary

Conclusions

Our report together with the previous reports [6,7] suggest that a positive family history in firstdegree relatives of P-NSAH accounts for higher risk of P-NSAH. Further investigation is necessary to illuminate the precise role of familial inheritance in pathogenesis. Reporting new aspects of such rare associations would help physicians to better understand the underlying pathology in these types of inadequately explained clinical entities. Consent Written informed consent was obtained from the patients for publication of this case report and any accompanying images. Copies of the written consents are available for review by the Editor-in-Chief of this journal. Competing interests The authors declare that they have no competing interests. All authors read and approved the final manuscript

Introduction

Findings

Discussion