Hussein Abdirashid Abdirahman, Mahamed Mahamed Mahamoud, Dildil Habtamu, Abdulahi Ismail, Osmael Abdulaziz Ibrahim, Admas Sebsbie, Abdi Mohamoud Hashi, Ali Abdirahman Omer
Department of Pediatrics and Child Health, Jigjiga University Sh. Hassan Yabare Comprehensive Specialized Hospital, Jigjiga, Ethiopia.
ENT Department, Jigjiga University Sh. Hassan Yabare Comprehensive Specialized Hospital, Jigjiga, Ethiopia.
Int Med Case Rep J. 2025 Jun 25;18:769-776. doi: 10.2147/IMCRJ.S524811. eCollection 2025.
Rhino-orbital-cerebral mucormycosis (ROCM) is a rare, aggressive fungal infection with high mortality, primarily affecting immunocompromised individuals, especially those with uncontrolled diabetes mellitus, though it can occur in immunocompetent hosts. Cerebral involvement carries a near 100% fatality rate if untreated, and overall mortality remains high (>30%) even with therapy. We present a pediatric ROCM case from the resource-limited Somali region of Ethiopia.
A 12-year-old female with type 1 diabetes mellitus presented with a 10-day history rapidly progressing from headache and fever to right-sided periorbital swelling, proptosis, vision loss, and a necrotic eschar. She had uncontrolled hyperglycemia and diabetic ketoacidosis (DKA) upon arrival. Imaging via CT and MRI revealed findings highly suggestive of ROCM, demonstrating right orbital involvement, cavernous sinus thrombosis, and further intracranial extension including cerebral abscesses/cerebritis and meningeal enhancement. Microbiological confirmation was unavailable due to resource limitations. Treatment included intravenous liposomal amphotericin B (requiring dose adjustment for transient nephrotoxicity), extensive endoscopic nasal debridement, right eye exenteration, and intensive glycemic control.
This case highlights the diagnostic and management challenges of ROCM in resource-limited settings. Despite the high mortality associated with ROCM, particularly in children, the patient improved significantly and was discharged in good health, albeit with persistent visual impairment. Successful management involved prompt recognition, aggressive multimodal therapy (medical and surgical), and close multidisciplinary collaboration (Pediatrics, Ophthalmology, ENT). This outcome demonstrates the feasibility of successful treatment even in resource-constrained environments and underscores the critical importance of managing underlying conditions like diabetes to prevent opportunistic infections.
鼻眶脑毛霉菌病(ROCM)是一种罕见的侵袭性真菌感染,死亡率高,主要影响免疫功能低下的个体,尤其是那些糖尿病控制不佳的患者,不过也可发生于免疫功能正常的宿主。若不治疗,脑部受累的死亡率接近100%,即使接受治疗,总体死亡率仍很高(>30%)。我们报告一例来自埃塞俄比亚资源有限的索马里地区的儿童ROCM病例。
一名12岁1型糖尿病女性患者,有10天病史,症状迅速从头痛和发热发展为右侧眶周肿胀、眼球突出、视力丧失和坏死性焦痂。入院时她患有未控制的高血糖和糖尿病酮症酸中毒(DKA)。CT和MRI成像显示高度提示ROCM的表现,显示右侧眼眶受累、海绵窦血栓形成以及进一步的颅内扩展,包括脑脓肿/脑脊髓炎和脑膜强化。由于资源限制,无法进行微生物学确诊。治疗包括静脉注射脂质体两性霉素B(因短暂性肾毒性需要调整剂量)、广泛的内镜下鼻腔清创、右眼摘除以及强化血糖控制。
本病例突出了资源有限环境中ROCM的诊断和管理挑战。尽管ROCM相关死亡率高,尤其是在儿童中,但患者显著改善并健康出院,尽管仍有持续性视力损害。成功的管理包括及时识别、积极的多模式治疗(药物和手术)以及密切的多学科协作(儿科、眼科、耳鼻喉科)。这一结果表明即使在资源受限的环境中成功治疗也是可行的,并强调了控制糖尿病等基础疾病以预防机会性感染的至关重要性。