Shahini Endrit, Marziliano Donatello, Losito Francesco, Zappimbulso Marianna, Cavalcanti Elisabetta, Armentano Raffaele, Fucilli Fabio, Cozzolongo Raffaele, Ingravallo Giuseppe, Pappagallo Fabrizio, Iatta Roberta, Desantis Vanessa, Saracino Annalisa, Vacca Angelo, Solimando Antonio Giovanni
Gastroenterology Unit, National Institute of Gastroenterology-IRCCS "Saverio de Bellis", Castellana Grotte, Bari, Italy.
Guido Baccelli Unit of Internal Medicine, Department of Precision and Regenerative Medicine and Ionian Area-(DiMePRe-J), School of Medicine, Aldo Moro University of Bari, Bari, Italy.
Front Immunol. 2025 Jun 5;16:1599381. doi: 10.3389/fimmu.2025.1599381. eCollection 2025.
Leishmaniasis is a potentially life-threatening protozoan infection that presents with many clinical manifestations, including cutaneous, mucocutaneus and visceral forms. In patients with rheumatoid arthritis (RA), cutaneous leishmaniasis can persist or re-emerge due to treatment-induced immunosuppression. However, it remains unclear whether this severe opportunistic infection is primarily driven by medication-induced immunosuppression or other poorly understood immune-mediated mechanisms that increase susceptibility.
We describe an unusual case of disseminated leishmaniasis in a 50-year-old Italian man from Apulia, diagnosed with RA two years earlier. Following 15 months of unsuccessful immunosuppressive therapies, he developed severe multilineage pancytopenia, moderate hypertransaminasemia, elevated inflammatory markers, monoclonal gammopathy, clinically significant hepatosplenomegaly, and an ulcerated skin lesion. Initial diagnostic efforts excluded common infectious agents, primary hematological disorders, Felty syndrome, and amyloidosis. The non-specific histopathological findings from the pyoderma gangrenosum-like lesion and the transient clinical response to empirical steroids, broad-spectrum antibiotics, and granulocyte colony-stimulating factors further complicated the diagnostic process. The breakthrough came when a liver biopsy, performed to investigate persistent hypertransaminasemia, revealed amastigotes within macrophages. This finding triggered a re-evaluation of the ulcerated skin lesion, and histological analysis confirmed concurrent cutaneous leishmaniasis. Subsequent bone marrow biopsy also identified amastigotes, clinching the diagnosis of disseminated leishmaniasis. A holistic re-assessment of the patient's clinical presentation, developmental history, and laboratory, radiologic, and pathological data led to the definitive diagnosis. Treatment with standard intravenous amphotericin B resulted in clinical resolution. A follow-up bone marrow biopsy a few weeks later confirmed the infection had been completely eradicated.
In patients with rheumatological conditions, the overlapping symptoms of systemic diseases and infections like leishmaniasis can lead to significant diagnostic delays. This case underscores the importance of comprehensive and meticulous diagnostic evaluations in immunosuppressed individuals to prevent potentially fatal outcomes.
利什曼病是一种潜在的危及生命的原生动物感染，有多种临床表现，包括皮肤型、黏膜皮肤型和内脏型。在类风湿关节炎（RA）患者中，由于治疗引起的免疫抑制，皮肤利什曼病可能持续存在或再次出现。然而，尚不清楚这种严重的机会性感染主要是由药物引起的免疫抑制还是其他尚不清楚的免疫介导机制导致易感性增加所驱动。
我们描述了一例来自普利亚的50岁意大利男性播散性利什曼病的不寻常病例，该患者两年前被诊断为RA。在15个月的免疫抑制治疗失败后，他出现了严重的多系血细胞减少、中度高转氨酶血症、炎症标志物升高、单克隆丙种球蛋白病、具有临床意义的肝脾肿大以及皮肤溃疡病变。最初的诊断努力排除了常见的感染病原体、原发性血液系统疾病、费尔蒂综合征和淀粉样变性。坏疽性脓皮病样病变的非特异性组织病理学发现以及对经验性使用类固醇、广谱抗生素和粒细胞集落刺激因子的短暂临床反应使诊断过程更加复杂。当为调查持续的高转氨酶血症而进行的肝脏活检显示巨噬细胞内有无鞭毛体时，取得了突破。这一发现促使对皮肤溃疡病变进行重新评估，组织学分析证实并发皮肤利什曼病。随后的骨髓活检也发现了无鞭毛体，确诊为播散性利什曼病。对患者的临床表现、发育史以及实验室、放射学和病理学数据进行全面重新评估后得出了明确诊断。采用标准静脉注射两性霉素B治疗后临床症状缓解。几周后的随访骨髓活检证实感染已被完全根除。
在患有风湿病的患者中，系统性疾病和利什曼病等感染的重叠症状可能导致显著的诊断延迟。该病例强调了对免疫抑制个体进行全面细致的诊断评估以预防潜在致命后果的重要性。
