Epidemiological and clinical trends of visceral leishmaniasis in Portugal: retrospective analysis of cases diagnosed in public hospitals between 2010 and 2020
Authors: Rocha Rafael , Conceição Cláudia , Gonçalves Luzia ,   LeishPT group, Carvalho Ana Cláudia , Maia André , Martins André , Carujo António , Maio António , Forra Catarina , Melita Catarina , Couto Daniela , Fernandes Diana , Pereira Dulce , Leal Ema , Sarmento Helena , Sousa Inês , Gonçalves Jean-Pierre , Marinho Joana , Vasconcelos Joana , Cunha João , Rodrigues João , Silva José Miguel , Caley Lídia , Malheiro Luís , Santos Luís , Garcia Margarida , Prata Margarida , Cunha Maria , Lima Maria , Andrade Maria Margarida , Marques Marta , Alpalhão Miguel , Silva Mónica , Ferraz Rita , Soares Rui , Fernandes Salomão , Llobet Samuel , Cruz Sofia , Guimarães Teresa , Branco Tiago , Robalo-Nunes Tomás , Almeida Vasco , Maia Carla 
Published: June 2024

Abstract
Background
Leishmania infantum is endemic in the Mediterranean region, presenting mostly as visceral leishmaniasis (VL). In Portugal, reporting of VL cases to public health authorities is mandatory, but significant underreporting is likely. This study aimed to describe the epidemiological and clinical aspects of the VL cases diagnosed in hospitals of the Portuguese National Health Service (NHS), between 2010 and 2020.

Methods
Collaboration was requested to every hospital of the Portuguese NHS in Mainland Portugal. Cases were screened through a search of diagnostic discharge codes or, if not available, by a search of positive laboratory results for Leishmania infection. Sociodemographic and clinical data was retrieved from medical records. Simultaneously, the National Health authority was contacted to request access to data of notified cases of VL between 2010 and 2020. Descriptive, hypothesis testing and multiple binary logistic regression models were performed.

Results
A total of 221 VL cases were identified. A significant increase in estimated national incidence was seen in the years after 2016 (P = 0.030). VL was predominantly diagnosed in people living with HIV (PLWH) and in children (representing around 60% of the new cases), but the outcome was generally poorer in non-HIV patients with associated immunosuppression, with significantly lower rates of clinical improvement at 7 (P = 0.003) and 30 days (P = 0.008) after treatment. Atypical presentations, with gastrointestinal and/or respiratory involvement, were seen in 8.5% of VL cases. Hemophagocytic lymphohistiocytosis was diagnosed in 40.0% of children under 5 years of age. Only 49.7% of incident VL cases were reported. Simultaneous involvement of the skin was confirmed in 5.9% of patients.

Conclusions
VL presents a continuing threat in Portugal, especially to PLWH and children, and an increasing threat to other immunosuppressed groups. Recent increases in incidence should be closely monitored to allow prompt interventions. Programs to control the disease should focus on providing tools for earlier diagnosis and on reducing underreporting and promoting an integrated surveillance of human and animal disease. These data should be combined with asymptomatic infection and vector information, following a One Health approach.

Graphical Abstract