Department of Experimental and Clinical Medicine, University of Florence, Florence, Italy.
Otorhinolaryngology Unit, Careggi University Hospital, Florence, Italy.
Int J Infect Dis. 2020 Aug;97:204-207. doi: 10.1016/j.ijid.2020.06.003. Epub 2020 Jun 4.
Mucocutaneous leishmaniasis (MCL) is a complication of tegumentary leishmaniasis, causing potentially life-threatening lesions in the ear, nose, and throat (ENT) region, and most commonly due to Leishmania (Viannia) braziliensis. We report a case of relapsing MCL in an Italian traveler returning from Argentina.
A 65-year-old Italian male patient with chronic kidney disease, arterial hypertension, prostatic hypertrophy, and type-2 diabetes mellitus was referred for severe relapsing MCL acquired in Argentina. ENT examination showed severe diffuse pharyngolaryngeal edema and erythema, partially obstructing the airways. A nasopharyngeal biopsy revealed a lymphoplasmacytic inflammation and presence of Leishmania amastigotes, subsequently identified as L. (V.) braziliensis by hsp70 PCR-RFLP analysis and sequencing. Despite receiving four courses of liposomal amphotericine B (L-AmB) and two courses of miltefosine over a 2-year period, the patient presented recurrence of symptoms a few months after the end of each course. After the patient was referred to us, a combined treatment was started with intravenous pentamidine 4 mg/kg on alternate days for 10 doses, followed by one dose per week for an additional seven doses, intralesional meglumine antimoniate on the nasal lesion once per week for six doses, oral azoles for three months, and aerosolized L-AmB on alternate days for three months. The treatment led to regression of mucosal lesions and respiratory symptoms. Renal function temporarily worsened, and the addition of insulin was required to maintain glycemic compensation after pentamidine discontinuation.
This case highlights the difficulties in managing a life-threatening refractory case of MCL in an Italian traveler with multiple comorbidities. Even though parenteral antimonial derivatives are traditionally considered the treatment of choice for MCL, they are relatively contraindicated in cases of chronic kidney disease.The required dose adjustment in cases of impaired renal function is unknown, therefore the use of alternative drugs is recommended. This case was resolved with combination treatment, including aerosolized L-AmB, which had never been used before for MCL.
黏膜皮肤利什曼病（MCL）是皮肤利什曼病的一种并发症，可导致耳、鼻、喉（ENT）区域危及生命的潜在病变，最常见的病因是巴西利什曼原虫（Viannia）。我们报告了一例来自阿根廷的意大利旅行者复发性 MCL 病例。
一名 65 岁的意大利男性患者，患有慢性肾脏病、动脉高血压、前列腺增生和 2 型糖尿病，因在阿根廷获得的严重复发性 MCL 而被转介。耳鼻喉科检查显示严重弥漫性咽扁桃体肿胀和红斑，部分阻塞气道。鼻咽喉活检显示淋巴浆细胞炎症和利什曼原虫无鞭毛体存在，随后通过 hsp70 PCR-RFLP 分析和测序鉴定为 L.（V.）braziliensis。尽管在 2 年内接受了 4 个疗程的脂质体两性霉素 B（L-AmB）和 2 个疗程的米替福新治疗，但在每个疗程结束后数月，患者的症状仍会复发。在患者转至我们处后，开始采用联合治疗，用 4 毫克/千克的戊烷脒静脉注射，隔日 1 次，共 10 剂，随后每周 1 次，再用 7 剂；每周 1 次，用 6 剂对鼻病变进行病灶内氨苯砜注射；口服唑类药物 3 个月；隔日 1 次，用 3 个月的 aerosolized L-AmB。治疗导致黏膜病变和呼吸症状消退。肾功能暂时恶化，戊烷脒停药后需要添加胰岛素以维持血糖代偿。
本例突出了在患有多种合并症的意大利旅行者中，管理危及生命的难治性 MCL 病例所面临的困难。尽管传统上认为亲脂性锑衍生物是 MCL 的首选治疗方法，但在慢性肾脏病的情况下，它们相对禁忌使用。在肾功能受损的情况下，所需的剂量调整尚不清楚，因此建议使用替代药物。该病例通过联合治疗得到解决，包括以前从未用于治疗 MCL 的 aerosolized L-AmB。
