A case report on recurrent alternating Tolosa-Hunt syndrome due to bacterial sphenoid sinusitis: rediscussing the diagnostic terminology and classification

作者全名："He, Wei; Zhu, Yinglin; Zhang, Yinan; Dong, Liang; Zhou, Zefang; Zhou, Jiying"

作者地址："[He, Wei; Zhang, Yinan; Zhou, Zefang] Chongqing Med Univ, Affiliated Hosp 1, Dept Neurol, Branch 1, 191st Ren Min Rd, Yu Zhong Dist, Chongqing 400015, Peoples R China; [Zhu, Yinglin] Kansas City Univ Med & Biosci, Sch Osteopath Med, Joplin, MO 64801 USA; [Dong, Liang; Zhou, Jiying] Chongqing Med Univ, Affiliated Hosp 1, Dept Neurol, 1st You Yi Rd, Chongqing 400016, Peoples R China"

通信作者："Zhou, ZF (通讯作者)，Chongqing Med Univ, Affiliated Hosp 1, Dept Neurol, Branch 1, 191st Ren Min Rd, Yu Zhong Dist, Chongqing 400015, Peoples R China.; Zhou, JY (通讯作者)，Chongqing Med Univ, Affiliated Hosp 1, Dept Neurol, 1st You Yi Rd, Chongqing 400016, Peoples R China."

来源：BMC NEUROLOGY

ESI学科分类：NEUROSCIENCE & BEHAVIOR

WOS号：WOS:000913287300001

JCR分区：Q3

影响因子：2.2

年份：2023

卷号：23

期号：1

开始页：　

结束页：　

文献类型：Article

关键词：Tolosa-Hunt syndrome (THS); Painful ophthalmoplegia; Cavernous sinus syndrome; Diagnostic criteria; ICHD-3

摘要："BackgroundTolosa-Hunt syndrome (THS) is characterized by painful ophthalmoplegia caused by idiopathic granulomatous inflammation involving the cavernous sinus region. Patients respond well to steroid therapy. THS is included in the differential diagnosis of cavernous sinus syndrome, so it is important to fully exclude other lesions in this area before treatment, otherwise steroid treatment may lead to fatal outcomes. Here we describe a patient who initially presented with symptoms that simulated THS symptoms and developed recurrent alternating painful ophthalmoplegia during follow-up, and the patient was finally diagnosed with cavernous sinusitis caused by bacterial sphenoid sinusitis.Case presentationA 34-year-old woman presented with left painful ophthalmoplegia. Magnetic resonance imaging (MRI) revealed abnormal signals in the left cavernous sinus area, and these abnormal signals were suspected to be THS. After steroid treatment, the patient obtained pain relief and had complete recovery of her ophthalmoplegia. However, right painful ophthalmoplegia appeared during the follow-up period. MRI showed obvious inflammatory signals in the right cavernous sinus and right sphenoid sinus. Then nasal sinus puncture and aspiration culture were performed, and the results showed a coagulase-negative staphylococcus infection. After antibiotic treatment with vancomycin, the painful ophthalmoplegia completely resolved, and the neurological examination and MRI returned to normal.ConclusionSome other causes of painful ophthalmoplegia also fulfill the diagnostic criteria for THS in the International Classification of Headache Disorders third edition (ICHD-3) and respond well to steroid therapy. Early diagnosis of THS may be harmful to patients, and clinicians should exercise great caution when dealing with similar cases without a biopsy. Using ""cavernous sinus syndrome"" instead of ""Tolosa-Hunt syndrome"" as a diagnostic category may provide a better clinical thinking for etiological diagnosis."

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