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半个世纪的隐匿:一例罕见超长潜伏期结核样型麻风。

Half a Century in Hiding: A Unique Case of Tuberculoid Leprosy with an Unprecedented Incubation Period.

机构信息

Department of Infectious Diseases, Mayo Clinic Florida, Jacksonville, FL, USA.

出版信息

Am J Case Rep. 2024 Feb 14;25:e942048. doi: 10.12659/AJCR.942048.

Abstract

BACKGROUND Leprosy, also known as Hansen's disease, is a neglected tropical disease with low prevalence in the United States. The disease's long incubation period can cause delayed presentation, and most affected individuals have a history of travel or work in leprosy-endemic regions. The immune response to Mycobacterium leprae determines the clinical characteristics of leprosy, with tuberculoid leprosy being characterized by well-defined granulomas and involvement of peripheral nerves. The recommended treatment is a combination of dapsone and rifampin for 12 months. CASE REPORT A 78-year-old man with a history of extensive travel to Africa and Asia 50 years ago, presented with a non-tender, non-pruritic, and hypopigmented skin lesion on his left knee. Biopsy results confirmed granulomatous inflammation and the presence of Mycobacterium leprae, leading to a diagnosis of tuberculoid/paucibacillary leprosy. The patient received dapsone and rifampin treatment, which resulted in symptom improvement. CONCLUSIONS The patient's long incubation period of 50 years between exposure and symptom onset is remarkable and possibly one of the longest reported for tuberculoid leprosy. It emphasizes the importance of considering leprosy in cases with an extensive travel history and long incubation periods. Our patient's case presented contradictory staining results, suggesting potential sampling variation or a rare mixed leprosy form. Based on his clinical findings, he was diagnosed with tuberculoid leprosy. Early diagnosis and treatment are crucial to prevent irreversible nerve damage and improve patient outcomes. Healthcare providers should be vigilant in acquiring a detailed travel history to facilitate early diagnosis and appropriate management of leprosy cases.

摘要

背景

麻风病,又称汉森病,是一种被忽视的热带病,在美国的发病率较低。该疾病潜伏期长,可导致就诊延迟,大多数受影响的个体都有在麻风病流行地区旅行或工作的历史。针对麻风分枝杆菌的免疫反应决定了麻风病的临床特征,结核样型麻风病的特征是定义明确的肉芽肿和外周神经受累。推荐的治疗方法是联合使用氨苯砜和利福平治疗 12 个月。

病例报告

一名 78 岁男性,50 年前曾广泛前往非洲和亚洲旅行,现左膝出现无痛、非瘙痒性、色素减退性皮肤损伤。活检结果证实为肉芽肿性炎症和麻风分枝杆菌的存在,导致结核样/少菌型麻风病的诊断。患者接受了氨苯砜和利福平治疗,症状得到改善。

结论

患者从接触到出现症状的潜伏期长达 50 年,这是非常显著的,可能是报告的最长潜伏期之一。这强调了在有广泛旅行史和长潜伏期的病例中考虑麻风病的重要性。我们患者的病例呈现出相互矛盾的染色结果,提示可能存在采样变异或罕见的混合麻风病形式。根据他的临床发现,他被诊断为结核样麻风病。早期诊断和治疗对于预防不可逆的神经损伤和改善患者预后至关重要。医疗保健提供者应警惕获取详细的旅行史,以促进麻风病病例的早期诊断和适当管理。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8d9a/10877640/c090f1a593ec/amjcaserep-25-e942048-g001.jpg

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