Department of Otolaryngology-Head and Neck Surgery, Institute of Biomedical Sciences, Tokushima University Graduate School, Tokushima, Japan.
Department of Otolaryngology, Yoshinogawa Medical Center, Tokushima, Japan.
J Med Invest. 2024;71(3.4):310-313. doi: 10.2152/jmi.71.310.
Orbital apex syndrome (OAS) is a complex condition characterized by visual loss, diplopia, and eye pain that occurs secondary to several pathological processes involving the orbital apex. We report a case of acute invasive fungal rhinosinusitis (AIFRS) associated with OAS. A 76-year-old man with left-sided visual loss, diplopia, palpebral ptosis, and headache was diagnosed with OAS secondary to Tolosa-Hunt syndrome and received systemic corticosteroid therapy from his neurologist. Owing to persistent symptoms, we opened the optic canal using a transnasal endoscopic approach for a surgical biopsy of the orbital apex lesions. Histopathological evaluation revealed numerous Aspergillus organisms in the biopsied granuloma. After surgical debridement, he received a 12-month course of voriconazole, and no recurrence of AIFRS occurred during 8-year follow-up. Patients with OAS may occasionally be prescribed corticosteroids because the clinical manifestations of AIFRS-induced OAS are similar to those observed in OAS secondary to Tolosa-Hunt syndrome, especially no nasal symptoms which is known to respond to corticosteroid therapy. Because both AIFRS-induced OAS and OAS secondary to Tolosa-Hunt syndrome induce ophthalmoplegia, proptosis, eye pain, it is sometimes difficult to differentiate these two diseases in early stage. However, corticosteroid therapy causes exacerbation of fungal infection in patients with AIFRS-induced OAS resulting in delayed accurate diagnosis and poor prognosis. AIFRS is associated with a high mortality rate ; therefore, transnasal endoscopic biopsy of orbital apex lesions before corticosteroid administration is recommended in patients with OAS. J. Med. Invest. 71 : 310-313, August, 2024.
眶尖综合征（OAS）是一种复杂的疾病，其特征是视力丧失、复视和眼部疼痛，这些症状继发于涉及眶尖的几种病理过程。我们报告一例与眶尖综合征相关的急性侵袭性真菌性鼻-鼻窦炎（AIFRS）病例。一名 76 岁男性因左眼视力丧失、复视、眼睑下垂和头痛而被诊断为托洛萨-亨特综合征继发的 OAS，并接受了神经内科医生的全身皮质类固醇治疗。由于症状持续存在，我们采用经鼻内镜入路开放视神经管，对眶尖病变进行手术活检。组织病理学评估显示活检肉芽肿中有许多曲霉属真菌。在手术清创后，他接受了为期 12 个月的伏立康唑治疗，在 8 年的随访期间未发生 AIFRS 复发。OAS 患者偶尔会开皮质类固醇，因为 AIFRS 引起的 OAS 的临床表现与托洛萨-亨特综合征继发的 OAS 相似，尤其是没有已知对皮质类固醇治疗有反应的鼻部症状。由于 AIFRS 引起的 OAS 和托洛萨-亨特综合征继发的 OAS 均引起眼肌麻痹、眼球突出、眼痛，因此有时难以在早期区分这两种疾病。然而，皮质类固醇治疗会导致 AIFRS 引起的 OAS 患者的真菌感染恶化，导致诊断延迟和预后不良。AIFRS 死亡率高；因此，建议在 OAS 患者中在皮质类固醇给药前对眶尖病变进行经鼻内镜活检。J. Med. Invest. 71 : 310-313, August, 2024.
