A rare condition: Spontaneous subarachnoid haemorrhage due to spinal hemangioblastoma: Report of 2 cases and review of the literature - 23/10/20

Doi : 10.1016/j.neuchi.2020.05.008

C. Tuleasca ^a,^b,^c,^d,^e,^⁎ , S. Knafo ^e, P. David ^e,ⁱ, S. Richard ^f,^g,ⁱ, C. Adam ^h,ⁱ, N. Aghakhani ^e,ⁱ, F. Parker ^e,ⁱ
^a Department of clinical neurosciences, Neurosurgery service and Gamma knife centre, Lausanne university hospital (CHUV), Lausanne, Switzerland
^b Faculty of biology and medicine (FBM), university of Lausanne (UniL), Lausanne, Switzerland
^c Signal processing laboratory (LTS 5), École polytechnique fédérale de Lausanne (EPFL), Lausanne, Switzerland
^d Faculté de médecine, Sorbonne université, Paris, France
^e Service de neurochirurgie, Assistance publique–Hôpitaux de Paris, hôpitaux universitaires Paris-Sud, centre hospitalier universitaire de Bicêtre, Paris, France
^f Génétique oncologique EPHE, PSL research university, Paris, France
^g Faculté de médecine Paris-Sud, université Paris-Saclay, Inserm UMR 1186, Gustave-Roussy, Villejuif, France
^h Laboratoire de neuropathologie, GHU Paris-Sud – hôpital Bicêtre, Le Kremlin-Bicêtre, France
ⁱ PREDIR National rare adult cancer reference network: INCa, Assistance publique–Hôpitaux de Paris, GHU Paris-Sud – hôpital Bicêtre, Le Kremlin-Bicêtre, France

^⁎Corresponding author at: Centre hospitalier universitaire de Lille, hôpital Roger-Salengro; centre hospitalier universitaire de Vaudois, Neurosurgery Service and Gamma Knife Centre, rue du Bugnon 44–46, BH-08, CH-1011 Lausanne, Switzerland.Centre hospitalier universitaire de Lille, hôpital Roger-Salengro; centre hospitalier universitaire de Vaudois, Neurosurgery Service and Gamma Knife Centrerue du Bugnon 44–46, BH-08LausanneCH-1011Switzerland

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Abstract

Introduction

Subarachnoid haemorrhage (SAH), secondary to spinal hemangioblastoma (HBL), is extremely rare, with only a few case reports to date. We report the experience of our reference centre for spinal tumours and Von Hippel–Lindau (VHL) disease in patients with spinal HBL presenting with SAH. We further performed a systematic review of the literature.

Methods

We report two cases. A systematic search was performed using the PubMed, Embase and Cochrane databases, with no limit for publication date. Inclusion criteria were: patients with HBL presenting with SAH, with or without VHL. The systematic review retrieved only 10 studies, including 16 patients.

Results

In our centre, the first case concerned radicular HBL at D12 level, presenting with spinal and brain SAH. The patient underwent uneventful microsurgical en bloc resection. Postoperative course was normal. The second case concerned HBL with SAH at the cervico-medullary junction, with rapidly fatal course. The systematic review revealed female predominance, at a median age of 40 years, with HBL predominantly located at cervical level, common preoperative symptoms being headache and signs of meningeal irritation.

Conclusions

In conclusion, spinal HBL is an extremely rare cause of SAH. The systematic review found putative risk factors: female gender, age 40–50 years, cervical location, and median size 2cm. Diagnosis can be difficult when presentation mimics intracerebral SAH. We advocate early surgical removal. The risk of rapidly fatal course, in case of major haemorrhage, needs to be borne in mind.

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Keywords : Hemangioblastomas, Subarachnoid haemorrhage, Spinal, Von Hippel–Lindau

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Introduction

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Vol 66 - N° 5

P. 359-364 - novembre 2020 Retour au numéro

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A rare condition: Spontaneous subarachnoid haemorrhage due to spinal hemangioblastoma: Report of 2 cases and review of the literature - 23/10/20

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