Alotaibi Naif H, Abaalkhail Mashal, Almusa Hala, Alshenaifi Lama A, Alomairin Ahmed
Department of Otolaryngology-Head & Neck, King Faisal Specialist Hospital and Research Center (KFSH&RC), Riyadh, Saudi Arabia; Department of Surgery, College of Medicine, Alfaisal University, Riyadh, Saudi Arabia.
College of Medicine, King Saud University, Riyadh, Saudi Arabia.
Int J Surg Case Rep. 2025 Jan;126:110642. doi: 10.1016/j.ijscr.2024.110642. Epub 2024 Nov 23.
Allergic fungal rhinosinusitis (AFRS) is a distinct subset of chronic rhinosinusitis characterized by a type I hypersensitivity to fungi. Immunocompromised patients are at increased risk for fungal infections. This case highlights the complexities of managing AFRS in patients not eligible for surgery.
We present a 32-year-old patient with a history of Wilson's disease and a previous liver transplant on immunosuppressive medications. The patient presented to our tertiary hospital with a year-long history of bilateral progressive nasal obstruction, along with diplopia, anterior and posterior rhinorrhea, intermittent numbness on the left side of her face, tinnitus and nausea. Initial imaging revealed extensive sinonasal disease with hyperdense components. FESS was performed, and biopsy and culture confirmed the diagnosis of AFRS. Notably, gross dehiscence of the carotid arteries was observed during surgery. Three and a half years after surgery, the patient experienced a relapse of symptoms after COVID-19 infection. Due to the risk of inadvertent damage to the carotid artery during surgery, the patient was offered a trial of dupilumab instead. The patient reported complete resolution of symptoms 6 months after starting dupilumab.
Given the significant surgical risks associated with carotid artery dehiscence, a trial of dupilumab was initiated. This biologic therapy, known for its efficacy in treating type 2 inflammatory conditions, resulted in a remarkable response, with complete resolution of symptoms within six months. Previous reports of dupilumab use in AFRS have yielded similar outcomes.
The case underscores the potential of dupilumab as a safe and effective alternative for managing recalcitrant AFRS, particularly in patients at high risk for surgical complications.
变应性真菌性鼻-鼻窦炎(AFRS)是慢性鼻-鼻窦炎的一个独特亚组,其特征为对真菌的I型超敏反应。免疫功能低下的患者发生真菌感染的风险增加。本病例突出了在不符合手术条件的患者中管理AFRS 的复杂性。
我们报告一名32岁有威尔逊病病史且曾接受肝移植并服用免疫抑制药物的患者。该患者到我们的三级医院就诊,有长达一年的双侧进行性鼻塞病史,伴有复视、鼻前和鼻后流涕(鼻漏)、左侧面部间歇性麻木、耳鸣和恶心。初始影像学检查显示鼻窦疾病广泛且有高密度成分。进行了功能性内镜鼻窦手术(FESS),活检和培养确诊为AFRS。值得注意的是,手术中观察到颈动脉明显裂开。手术后三年半,患者在感染新冠病毒后症状复发。由于手术中有意外损伤颈动脉的风险,改为给予患者度普利尤单抗试验性治疗。患者报告在开始使用度普利尤单抗6个月后症状完全缓解。
鉴于与颈动脉裂开相关的重大手术风险,启动了度普利尤单抗试验性治疗。这种生物疗法以其在治疗2型炎症性疾病方面的疗效而闻名,产生了显著反应,症状在6个月内完全缓解。先前关于度普利尤单抗用于AFRS的报道也有类似结果。
该病例强调了度普利尤单抗作为治疗难治性AFRS的安全有效替代方案的潜力,特别是在有手术并发症高风险的患者中。