University of Ottawa, Ottawa, ON, Canada.
Division of Anatomical Pathology, The Ottawa Hospital, Ottawa, ON, Canada.
BMC Infect Dis. 2023 Nov 21;23(1):815. doi: 10.1186/s12879-023-08811-x.
Leprosy is rare within non-endemic countries such as Canada, where cases are almost exclusively imported from endemic regions, often presenting after an incubation period of as many as 20 years. Due to its rarity and prolonged incubation period, diagnosis is often delayed, which may result in neurologic impairment prior to the initiation of treatment. In this report we describe a case that is novel in its incubation period, which is the longest reported to-date and may have contributed to diagnostic delay. The case also uniquely demonstrates the challenges of distinguishing leprosy reactions from new rheumatologic manifestations in a patient with established autoimmune disease.
We describe an 84-year-old male patient with rheumatoid arthritis on methotrexate and hydroxychloroquine, with no travel history outside Canada for 56 years, who presented in 2019 with new-onset paresthesias and rash. His paresthesias persisted despite a short course of prednisone, and his rash recurred after initial improvement. He underwent skin biopsy in May 2021, which eventually led to the diagnosis of leprosy. He was diagnosed with type 1 reaction and was started on rifampin, dapsone, clofazimine and prednisone, with which his rash resolved but his neurologic impairment remained.
This case report serves to highlight the potential for leprosy to present after markedly prolonged incubation periods. This is especially relevant in non-endemic countries that is home to an aging demographic of individuals who migrated decades ago from endemic countries. The importance of this concept is emphasized by the persistent neurologic impairment suffered by our case due to untreated type 1 reaction. We also demonstrate the necessity of skin biopsy in distinguishing this diagnosis from other autoimmune mimics in a patient with known autoimmune disease.
麻风病在加拿大等非流行国家很少见，这些国家的病例几乎都是从流行地区输入的，潜伏期通常长达 20 年。由于其罕见性和潜伏期长，诊断常常被延误，这可能导致在开始治疗之前出现神经损伤。在本报告中，我们描述了一个潜伏期很长的病例，这是迄今为止报道的最长潜伏期病例，可能导致了诊断的延误。该病例还独特地展示了在患有已确诊自身免疫性疾病的患者中，区分麻风病反应与新的风湿表现的挑战。
我们描述了一位 84 岁的男性患者，患有类风湿关节炎，正在服用甲氨蝶呤和羟氯喹，56 年来没有在加拿大境外旅行的经历，于 2019 年出现新发感觉异常和皮疹。他的感觉异常在短期使用泼尼松治疗后仍然存在，皮疹在最初改善后再次出现。他于 2021 年 5 月接受了皮肤活检，最终导致了麻风病的诊断。他被诊断为 1 型反应，并开始服用利福平、氨苯砜、氯法齐明和泼尼松，皮疹消退，但神经损伤仍然存在。
本病例报告强调了麻风病在明显延长的潜伏期后出现的可能性。这在那些有几十年前从流行地区移民而来的、人口老龄化的非流行国家尤其相关。由于我们的病例因未治疗的 1 型反应而持续存在神经损伤，因此这个概念的重要性得到了强调。我们还展示了在患有已知自身免疫性疾病的患者中，皮肤活检在区分这种诊断与其他自身免疫性疾病模拟物的必要性。
