Fermeli Dionysia D, Theofanopoulos Andreas, Papadakos Dimitrios, Boulieris Spiros, Constantoyannis Constantine
Department of Neurosurgery, University Hospital of Patras, Patras, Greece.
Am J Case Rep. 2021 May 25;22:e930437. doi: 10.12659/AJCR.930437.
BACKGROUND Cavernous malformations (CMs) or hemangiomas are benign vascular hamartomas of the central nervous system (CNS) that constitute 5-15% of all CNS vascular malformations. Most patients with brainstem CMs present with a sudden onset of seizures, intracranial hemorrhage, cranial nerve deficits, headache, or ataxia. Up to 20% to 50% of patients are asymptomatic, and their CMs are diagnosed incidentally on brain magnetic resonance imaging. CASE REPORT We present a case of a 42-year-old man with a brainstem cavernous hemangioma presenting with fever of unknown origin and mild headache without meningismus. The patient underwent a midline suboccipital craniectomy and removal of a ruptured brainstem cavernous hemangioma and the surrounding thrombus. Postoperatively, the patient developed left facial nerve palsy, left abducens nerve palsy, and xerostomia. Abducens palsy and xerostomia resolved spontaneously days after the operation. At the 6-month follow-up, the patient showed stable improvement with resolution of his neurological deficits. CONCLUSIONS To our knowledge, there is no reported case of a patient with a ruptured brainstem cavernoma presenting with fever of unknown origin as the main symptom. We assume that the minimal intraventricular hemorrhage triggered the hypothalamic thermoregulating mechanism. Thus, it would be useful for physicians to raise the suspicion of a ruptured brainstem cavernous malformation with further imaging evaluation when investigating fever of unknown origin.
背景 海绵状血管畸形（CMs）或血管瘤是中枢神经系统（CNS）的良性血管错构瘤，占所有CNS血管畸形的5% - 15%。大多数脑干CM患者表现为突然发作的癫痫、颅内出血、颅神经功能缺损、头痛或共济失调。高达20%至50%的患者无症状，其CM在脑部磁共振成像时被偶然诊断。病例报告 我们报告一例42岁男性，患有脑干海绵状血管瘤，表现为不明原因发热和轻度头痛，无颈项强直。患者接受了枕下中线开颅手术，切除破裂的脑干海绵状血管瘤及周围血栓。术后，患者出现左侧面神经麻痹、左侧展神经麻痹和口干。展神经麻痹和口干在术后数天自行缓解。在6个月的随访中，患者神经功能缺损症状缓解，病情稳定改善。结论 据我们所知，尚无破裂脑干海绵状血管瘤患者以不明原因发热为主要症状的报道病例。我们推测最小量的脑室内出血触发了下丘脑体温调节机制。因此，在调查不明原因发热时，医生通过进一步的影像学评估提高对破裂脑干海绵状畸形的怀疑是有用的。
