Spontaneous Epidural Hematoma Secondary to Sphenoid Sinusitis Neurol Med Chir (Tokyo) 50, 399¿401, 2010 Spontaneous Epidural Hematoma Secondary to Sphenoid Sinusitis —Case Report— Yoshinobu TAKAHASHI, Naoya HASHIMOTO*, and Akihiko HINO** Department of Neurosurgery, Hokuto Hospital, Obihiro, Hokkaido; *Department of Neurosurgery, Osaka University Medical School, Suita, Osaka; **Department of Neurosurgery, Saiseikai Shiga Hospital, Ritto, Shiga Abstract Epidural hematoma is typically caused by direct head trauma. Spontaneous epidural hematoma can be caused by infections of adjacent regions, vascular malformations of the dura mater, and disorders of blood coagulation. A 10-year-old girl with no history of head injury presented with complaints of headache and fever. On arrival at our hospital, her Glasgow Coma Scale score was 13 and neurological examination revealed right oculomotor palsy and left hemiparesis. Computed tomography (CT) showed an epidural hematoma in the right temporal base. Preoperative angiography identified no specific vascular lesions. Removal of the hematoma was undertaken immediately. Retrospective evaluation of the preoperative CT revealed sphenoid sinusitis and a bone defect between the temporal base and the sphenoid sinus. The epidural hematoma was probably caused by the spread of inflammation from the sphenoid sinus. Sphenoid sinusitis is one of the possible causes of acute epidural hematoma, especially if no traumatic episodes or risk factors can be identified. Key words: Received sphenoid sinusitis, May 28, 2009; Accepted spontaneous epidural hematoma September 25, 2009 Neurol Med Chir (Tokyo) 50, May, 2010 399 Y. Takahashi et al. 400 Introduction Epidural hematoma is generally a direct sequela of head injury. Spontaneous epidural hematoma can arise from various other pathological conditions, including dural vascular lesions, disorders of coagulation, and infections of adjacent regions. Epidural hematoma occurring as a complication of paranasal sinusitis is very rare. We report a case of spontaneous epidural hematoma secondary to sphenoid sinusitis in a child. Case Report A 10-year-old girl with no history of head trauma suffered sudden onset of headache and high fever. She became drowsy and was brought to the hospital. Her Glasgow Coma Scale score on arrival at the hospital was 13. Neurological examination revealed oculomotor palsy on the right side and left hemiparesis. Emergent computed tomography (CT) showed an epidural hematoma at the Fig. 1 Computed tomography scan on admission showing an epidural hematoma in the right middle fossa, with marked midline shift. Fig. 2 Bone window computed tomography scan on admission showing a bone defect between the temporal base and the sphenoid sinus (arrow). right temporal base with marked midline shift (Fig. 1). Routine blood examination revealed no evidence of any coagulation disorder. Subsequent internal and external carotid angiography revealed no specific vascular lesions. Immediate surgery was undertaken to remove the epidural hematoma. The source of the bleeding could not be identified. Postoperative angiography also failed to reveal any lesions. Retrospective evaluation of the preoperative CT and postoperative magnetic resonance imaging demonstrated fluid collection in the sphenoid sinus, consistent with the diagnosis of sinusitis. The bone window CT images also showed a bone defect between the temporal base and the sphenoid sinus (Fig. 2). We suspected that the epidural hematoma was caused by spread of inflammation from the sphenoid sinus. The patient was successfully treated with antibiotics, recovered well, and was discharged with no neurological deficits. Discussion Review of 80 cases of intracranial arteriovenous malformations encountered during a 7-year period concluded that dural arteriovenous fistulas (AVFs) accounted for 6.25% of all such cases.6) In general, vascular malformations tend to bleed into the subarachnoid or subdural space. Only one case of epidural hematoma caused by dural AVF has been reported.3) In our patient, neither preor postoperative angiography detected vascular malformations of the dura mater, nor were such malformations observed during surgery. Intracranial hemorrhage may also be caused by coagulopathies. Among 124 cases of intracranial hemorrhage in patients receiving anticoagulant therapy, the bleeding was subdural in 82% and intracerebral in 18%, whereas epidural bleeding was not observed.7) In the present case, there was no evidence of bleeding diathesis. Infections of adjacent regions including otitis, orbital cellulites, and paranasal sinusitis are also associated with the development of epidural hematoma. The first case of spontaneous epidural hematoma associated with sinusitis was reported in 1968.5) Nine cases of paranasal sinusitis associated with epidural hematoma have been reported (Table 1). Some mechanisms have been proposed to explain the formation of epidural hematoma associated with infections of adjacent regions. The infection may extend from the primary site through diploic vascular channels resulting in arteritis and subsequent rupture of the meningeal vessel wall.5) Alternatively, granulation tissue may erode through bone, with spontaneous hemorrhage occurring secondary to infarction of the tissue trapped by the bone edges.3) Intraoperative findings in a case of thrombosed diploic vessels were consistent with the former mechanism.4) However, numerous studies have confirmed the presence of bone destruction during surgery consistent with the second mechanism.1,5,8) In our case, head CT had clearly delineated the sinusitis and also the bone defect between the right temporal base and the sphenoid sinus, although we failed to notice these findings preoperatively. The risk is that the dura mater may become progressively stripped from the inner table of Neurol Med Chir (Tokyo) 50, May, 2010 Spontaneous Epidural Hematoma Secondary to Sphenoid Sinusitis Table 1 401 Reported cases of epidural hematoma associated with paranasal sinusitis Author (Year) Age (yrs) Sex Sinus Location of the hematoma Surgery Outcome Kelly and Smith (1968)5) Rajput and Rzdilsky (1971)11) Marks and Shaw (1982)8) Ataya (1986)1) Sakamoto et al. (1997)12) Papadopoulos et al. (2001)10) Griffiths et al. (2002)4) Moonis et al. (2002)9) Chaiyasate et al. (2007)2) Present case 11 18 31 31 16 17 17 21 14 10 M M M M F M M M M F frontal frontal frontal frontal maxillary frontal pansinusitis sphenoid pansinusitis sphenoid lt frontal lt frontal rt frontal lt frontal lt frontal rt frontal lt frontal lt temporal rt frontal rt temporal done no done done done done done done done done good dead good good good good good good good good the skull by extension of pus, exudates, and air.1,2,4,5,8–12) In our patient, the inflammation probably spread through the bone defect to cause local arteritis and rupture of the blood vessels, and progressive detachment of the dura mater led to recurrent bleeding and the formation of the epidural hematoma. Epidural hematoma may show rapid onset in some cases, but progress more slowly in others. In our case, the epidural hematoma was formed at the temporal base, and the infection spread directly from the sphenoid sinus, which led to rapid progression and impending tentorial herniation. All cases reported until date (including the present case) have occurred in patients younger than 31 years. This may be related to the increased adherence of the dura mater to the inner surface of the skull that occurs with advancing age. The present case illustrates that paranasal sinusitis is one of the possible causes of acute epidural hematoma, particularly in younger patients, especially in the absence of history of trauma or other identifiable risk factors. References 1) 2) 3) Ataya NL: Extradural hematoma secondary to chronic sinusitis: A case report. J Laryngol Otol 100: 951–953, 1986 Chaiyasate S, Halewyck S, Van Rompaey K, Clement P: Spontaneous extradural hematoma as a presentation of sinusitis: Case report and literature review. Int J Pediatr Otorhinolaryngol 71: 827–830, 2007 Gallagher JP, Browder EJ: Extradural hematoma: Experience with 167 patients. J Neurosurg 29: 1–12, 1968 Neurol Med Chir (Tokyo) 50, May, 2010 4) Griffiths SJ, Jatavallabhula NS, Mitchell RD: Spontaneous extradural haematoma associated with craniofacial infections: case report and review of the literature. Br J Neurosurg 16: 188–191, 2002 5) Kelly DL Jr, Smith JM: Epidural hematoma secondary to frontal sinusitis: Case report. J Neurosurg 28: 67–69, 1968 6) Kune Z, Bret J: Congenital arterio-sinusal fistulae. Acta Neurochir (Wien) 20: 85–103, 1969 7) Lizuka J: Intracranial and intraspinal haematomas associated with anticoagulant therapy. Neurochirurgia (Stuttg) 15: 15–25, 1972 8) Marks SM, Shaw MDM: Spontaneous intracranial extradural hematoma. Case report. J Neurosurg 57: 708–709, 1982 9) Moonis G, Granados A, Simon SL: Epidural hematoma as a complication of sphenoid sinusitis and epidural abscess. A case report and literature review. Clin Imaging 26: 382–385, 2002 10) Papadopoulos MC, Dyer A, Hardwidge C: Spontaneous extradural hematoma with sinusitis. A case report. J R Soc Med 94: 588–589, 2001 11) Rajput AJ, Rzdilsky B: Extradural hematoma following frontal sinusitis. Report of case and review of literature. Arch Otolaryngol Head Neck Surg 94: 83–86, 1971 12) Sakamoto T, Harimoto K, Inoue S, Konishi A: Extradural hematoma following maxillary sinusitis. Case illustration. J Neurosurg 87: 132, 1997 Address reprint requests to: Yoshinobu Takahashi, M.D., Department of Neurosurgery, Hokuto Hospital, 7–5 Inada, Obihiro, Hokkaido 080–0039, Japan. e-mail: yosinobu＠hokuto7.or.jp