Visceral leishmaniasis triggering an adult‐onset Still's disease: a unique case

Spoto, Silvia; Costantino, Sebastiano; Valeriani, Emanuele; Fogolari, Marta; Cocco, Eleonora; Dicuonzo, Giordano; Ciccozzi, Massimo; Angeletti, Silvia

doi:10.1002/ccr3.1266

Cited by 2 publications

(1 citation statement)

References 18 publications

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“…Many case reports refer to the occurrence of AOSD after viral infection or bacterial infection. Silvia et al firstly reported that visceral leishmaniasis triggered the AOSD 5 . This is the second reported case about AOSD caused by visceral leishmaniasis.…”

Section: Discussionmentioning

confidence: 84%

Death from an Adult‐onset Still’s disease triggered by visceral leishmaniasis

Wang

Huang

Wang

et al. 2020

Parasite Immunology

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A 24-year-old patient presented with a 4-month-long high-spiking fever, characteristic rash, sore throat and arthralgias. Bone marrow biopsy examination demonstrated visceral leishmaniasis with the presence of typical Leishman-Donovan bodies. During the treatment for visceral leishmaniasis with the sodium stibogluconate, he died suddenly. According to the symptoms, we infer that the patient catch the adult-onset Still's disease due to the visceral leishmaniasis.

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Section: Discussionmentioning

confidence: 84%

Death from an Adult‐onset Still’s disease triggered by visceral leishmaniasis

Wang

Huang

Wang

et al. 2020

Parasite Immunology

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Efficacy and safety of pentamidine isethionate for tegumentary and visceral human leishmaniasis: a systematic review

Piccica

Lagi

Bartoloni

et al. 2021

Journal of Travel Medicine

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Rationale for Review We performed a systematic review of the literature to investigate the efficacy and safety of pentamidine isethionate for the treatment of human tegumentary and visceral leishmaniasis. Key findings A total of 616 papers were evaluated, and 88 studies reporting data on 3108 cases of leishmaniasis (2082 patients with tegumentary leishmaniasis and 1026 with visceral leishmaniasis) were finally included. The majority of available studies were on New World cutaneous leishmaniasis and visceral leishmaniasis caused by Leishmania donovani. At the same time, few data are available for Old World cutaneous leishmaniasis, mucosal leishmaniasis, and visceral leishmaniasis caused by L. infantum. Pooled cure rate for tegumentary leishmaniasis was 78.8% (CI 95%, 76.9–80.6%) and 92.7% (CI 95%, 88.3–97.1%) according to controlled randomized trial and observational studies and case report and case series respectively. Pooled cure rate for visceral leishmaniasis was 84.8% (CI 95%, 82.6–87.1%) and 90.7% (CI 95%, 84.1–97.3%) according to controlled randomized trial and observational studies and case report and case series, respectively. Comparable cure rate was observed in recurrent and refractory cases of visceral leishmaniasis. Concerning the safety profile, among about 2000 treated subjects with some available information, the most relevant side effects were six cases of arrhythmia (including four cases of fatal ventricular fibrillation), 20 cases of irreversible diabetes, 26 cases of muscular aseptic abscess following intramuscular administration. Conclusions/recommendations Pentamidine isethionate is associated with a similar cure rate of the first-line anti-leishmanial drugs. Severe and irreversible adverse effect appear to be rare. The drug may still have a role in the treatment of any form of human leishmaniasis when the first-line option has failed or in patients who cannot tolerate other drugs also in the setting of travel medicine. In difficult cases, the drug can also be considered as a component of a combination treatment regimen.

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Visceral leishmaniasis triggering an adult‐onset Still's disease: a unique case

Abstract: Key Clinical MessageAdult‐onset Still's disease (AOSD) due to visceral leishmaniasis (VL) has not been previously reported. This case report analyzes a single episode of AOSD probably due to a visceral leishmaniasis successfully treated with pentamidine isethionate and prednisone.

Cited by 2 publications

References 18 publications

Death from an Adult‐onset Still’s disease triggered by visceral leishmaniasis

Death from an Adult‐onset Still’s disease triggered by visceral leishmaniasis

Efficacy and safety of pentamidine isethionate for tegumentary and visceral human leishmaniasis: a systematic review

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