Takeshi Miyazaki1, Fumihito Ohta, Mitsuhiro Daisu, Yoshinobu Hoshii. 1. Department of Neurosurgery, Masuda Red Cross Hospital, and Department of Neurosurgery, Shimane Medical University, Izumoshi, Shimane, Japan. takeshi@med.shimane-u.ac.jp
Abstract
OBJECTIVE AND IMPORTANCE: We are sometimes involved in the care of patients with neurofibromatosis Type 1 because of the associated disorders of cervicocerebral vessels. However, extracranial vertebral artery aneurysm in neurofibromatosis Type 1 is very rare. We present the first reported case of a rupture of an extracranial vertebral artery aneurysm into the thoracic cavity in a patient with neurofibromatosis Type 1. CLINICAL PRESENTATION: A 52-year-old woman who presented with a decrease in left-sided grip and numbness of the left upper limb was admitted. During history taking, she developed shock. Radiological examination revealed that a left extracranial vertebral artery aneurysm had ruptured into the thoracic cavity. With consciousness decreasing gradually because of hemorrhagic shock, the patient became comatose. INTERVENTION: Balloon occlusion of the vertebral artery proximal to the aneurysm was performed and surgical ligation was attempted, but cardiac arrest occurred immediately after the beginning of surgery, and the patient died. The vertebral artery proximal to the aneurysm was removed for pathological examination. CONCLUSION: In this case, the changes noted were interpreted as changes showing fragility of the vascular wall secondary to neurofibromatosis Type 1. Patients with neurofibromatosis Type 1 exhibit disorders of cervicocerebral vessels, and in some cases progression may follow a violent course. Periodic follow-up of such patients and early diagnosis are important.
OBJECTIVE AND IMPORTANCE: We are sometimes involved in the care of patients with neurofibromatosis Type 1 because of the associated disorders of cervicocerebral vessels. However, extracranial vertebral artery aneurysm in neurofibromatosis Type 1 is very rare. We present the first reported case of a rupture of an extracranial vertebral artery aneurysm into the thoracic cavity in a patient with neurofibromatosis Type 1. CLINICAL PRESENTATION: A 52-year-old woman who presented with a decrease in left-sided grip and numbness of the left upper limb was admitted. During history taking, she developed shock. Radiological examination revealed that a left extracranial vertebral artery aneurysm had ruptured into the thoracic cavity. With consciousness decreasing gradually because of hemorrhagic shock, the patient became comatose. INTERVENTION: Balloon occlusion of the vertebral artery proximal to the aneurysm was performed and surgical ligation was attempted, but cardiac arrest occurred immediately after the beginning of surgery, and the patient died. The vertebral artery proximal to the aneurysm was removed for pathological examination. CONCLUSION: In this case, the changes noted were interpreted as changes showing fragility of the vascular wall secondary to neurofibromatosis Type 1. Patients with neurofibromatosis Type 1 exhibit disorders of cervicocerebral vessels, and in some cases progression may follow a violent course. Periodic follow-up of such patients and early diagnosis are important.
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