关键词: CSF leak Gorham stout disease Multifocal low flow vascular anomalies Temporal bone

Mesh : Humans Cerebrospinal Fluid Rhinorrhea / surgery diagnosis etiology Child Retrospective Studies Female Male Temporal Bone / abnormalities diagnostic imaging Child, Preschool Vascular Malformations / complications diagnosis Treatment Outcome Adolescent

来  源:   DOI:10.1016/j.ijporl.2024.111999

Abstract:
OBJECTIVE: Atraumatic cerebrospinal fluid (CSF) rhinorrhea is uncommon in children and necessitates a multi-disciplinary evaluation for an etiology. Underlying osseous abnormality due to extensive or multifocal low flow vascular anomaly should be considered as a potential cause of spontaneous CSF leak. Treatment of multifocal low flow vascular anomalies may include medical and surgical approaches. In this series, we seek to determine the presenting signs and symptoms and medical and surgical treatment options for multifocal or extensive low flow vascular anomalies.
METHODS: A retrospective case series at a quaternary care children\'s hospital was compiled. All children with CSF rhinorrhea diagnosed and treated for multifocal low flow vascular anomalies at our institution were included. A total of four patients were identified.
RESULTS: All four patients had delay in initial diagnosis of underlying cause of meningitis and CSF rhinorrhea. Average age at diagnosis of multifocal low flow vascular anomaly was 7 years. This was on average 4 years after initial presentation for medical attention. Treatment approach was multidisciplinary and included medical management with sirolimus and bisphosphonates as well as surgical approaches to the skull base (lateral and anterior) to prevent CSF egress.
CONCLUSIONS: Consideration of multifocal low flow vascular anomaly should be included in any pediatric patient presenting with CSF rhinorrhea.
摘要:
目的:非创伤性脑脊液(CSF)鼻漏在儿童中并不常见,需要对病因进行多学科评估。由于广泛或多灶性低流量血管异常引起的潜在骨异常应被视为自发性CSF泄漏的潜在原因。多灶性低流量血管异常的治疗可包括医学和手术方法。在这个系列中,我们寻求确定多灶性或广泛性低流量血管异常的体征和症状以及药物和手术治疗方案.
方法:编制了一个第四纪儿童护理医院的回顾性病例系列。包括在我们机构诊断和治疗多灶性低流量血管异常的所有CSF鼻漏儿童。总共确定了4名患者。
结果:所有4例患者对脑膜炎和脑脊液鼻漏的潜在病因的初步诊断均有延迟。诊断多灶性低流量血管异常的平均年龄为7岁。这平均是在初次就诊后4年。治疗方法是多学科的,包括西罗莫司和双膦酸盐的医疗管理以及颅底(外侧和前部)的手术方法,以防止CSF流出。
结论:任何表现为CSF鼻漏的儿科患者应考虑多灶性低流量血管异常。
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