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      The disease burden of human cystic echinococcosis based on HDRs from 2001 to 2014 in Italy

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          Abstract

          Background

          Cystic echinococcosis (CE) is an important neglected zoonotic parasitic infection belonging to the subgroup of seven Neglected Zoonotic Disease (NZDs) included in the World Health Organization’s official list of 18 Neglected Tropical Diseases (NTDs). CE causes serious global human health concerns and leads to significant economic losses arising from the costs of medical treatment, morbidity, life impairments and fatality rates in human cases. Moreover, CE is endemic in several Italian Regions. The aim of this study is to perform a detailed analysis of the economic burden of hospitalization and treatment costs and to estimate the Disability Adjusted Life Years (DALYs) of CE in Italy.

          Methods and findings

          In the period from 2001 to 2014, the direct costs of 21,050 Hospital Discharge Records (HDRs) belonging to 12,619 patients with at least one CE-related diagnosis codes were analyzed in order to quantify the economic burden of CE. CE cases average per annum are 901 (min—max = 480–1,583). Direct costs include expenses for hospitalizations, medical and surgical treatment incurred by public and private hospitals and were computed on an individual basis according to Italian Health Ministry legislation. Moreover, we estimated the DALYs for each patient. The Italian financial burden of CE is around € 53 million; the national average economic burden per annum is around € 4 million; the DALYs of the population from 2001 to 2014 are 223.35 annually and 5.26 DALYs per 10 5 inhabitants.

          Conclusion

          In Italy, human CE is responsible for significant economic losses in the public health sector. In humans, costs associated with CE have been shown to have a great impact on affected individuals, their families and the community as a whole. This study could be used as a tool to prioritize and make decisions with regard to a surveillance system for this largely preventable yet neglected disease. It demonstrates the need of implementing a CE control program aimed at preventing the considerable economic and social losses it causes in high incidence areas.

          Author summary

          CE is a neglected tropical disease that remains a considerable health problem in endemic regions and which leads to substantial economic losses for agriculture sectors and public health systems. The parasitic cycle requires a definitive host (canidae) and an intermediate host (livestock species), while humans are only occasionally hosts. CE is more prevalent in areas where extensive or semi-extensive farming of livestock (mostly sheep) is common. The study of prevalence and health expenditure at the national macro-regional and regional level is a prerequisite for creating awareness of the importance of this disease. The Italian Ministry of Health provides remuneration to its regions’ hospitals for treatment of illnesses. In this study 21,050 hospital discharge records with CE diagnosis from 2001 to 2014 related to 12,619 patients were analyzed. The median of CE hospitalizations per annum was 848. The direct costs of HDRs for echinococcosis were estimated to be € 53,175,934.34. The assessment of economic remuneration by Italian Regions grouped by Nomenclature of Territorial Units 1 (NUTS1) shows that Southern Italy and its major islands spent €13,523,999 and €15,171,739 respectively. The national annual average direct cost for 10 5 inhabitants was € 6,398. The study of the National DALYs, a measure of social burden of disease, shows that 41% was correlated to Island patients DALYs per annum are 223.4. An analysis of direct costs and an estimate of DALYs are key factors for establishing how to prioritize control measures for CE, which is essentially a preventable disease.

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          Most cited references28

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          Biological, epidemiological, and clinical aspects of echinococcosis, a zoonosis of increasing concern.

          Echinococcosis in humans is a zoonotic infection caused by larval stages (metacestodes) of cestode species of the genus Echinococcus. Cystic echinococcosis (CE) is caused by Echinococcus granulosus, alveolar echinococcosis (AE) is caused by E. multilocularis, and polycystic forms are caused by either E. vogeli or E. oligarthrus. In untreated cases, AE has a high mortality rate. Although control is essentially feasible, CE remains a considerable health problem in many regions of the northern and southern hemispheres. AE is restricted to the northern hemisphere regions of North America and Eurasia. Recent studies have shown that E. multilocularis, the causative agent of AE, is more widely distributed than previously thought. There are also some hints of an increasing significance of polycystic forms of the disease, which are restricted to Central and South America. Various aspects of human echinococcosis are discussed in this review, including data on the infectivity of genetic variants of E. granulosus to humans, the increasing invasion of cities in Europe and Japan by red foxes, the main definitive hosts of E. multilocularis, and the first demonstration of urban cycles of the parasite. Examples of emergence or reemergence of CE are presented, and the question of potential spreading of E. multilocularis is critically assessed. Furthermore, information is presented on new and improved tools for diagnosing the infection in final hosts (dogs, foxes, and cats) by coproantigen or DNA detection and the application of molecular techniques to epidemiological studies. In the clinical field, the available methods for diagnosing human CE and AE are described and the treatment options are summarized. The development of new chemotherapeutic options for all forms of human echinococcosis remains an urgent requirement. A new option for the control of E. granulosus in the intermediate host population (mainly sheep and cattle) is vaccination. Attempts are made to reduce the prevalence of E. multilocualaris in fox populations by regular baiting with an anthelmintic (praziquantel). Recent data have shown that this control option may be used in restricted areas, for example in cities, with the aim of reducing the infection risk for humans.
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            World Health Organization Estimates of the Global and Regional Disease Burden of 11 Foodborne Parasitic Diseases, 2010: A Data Synthesis

            Background Foodborne diseases are globally important, resulting in considerable morbidity and mortality. Parasitic diseases often result in high burdens of disease in low and middle income countries and are frequently transmitted to humans via contaminated food. This study presents the first estimates of the global and regional human disease burden of 10 helminth diseases and toxoplasmosis that may be attributed to contaminated food. Methods and Findings Data were abstracted from 16 systematic reviews or similar studies published between 2010 and 2015; from 5 disease data bases accessed in 2015; and from 79 reports, 73 of which have been published since 2000, 4 published between 1995 and 2000 and 2 published in 1986 and 1981. These included reports from national surveillance systems, journal articles, and national estimates of foodborne diseases. These data were used to estimate the number of infections, sequelae, deaths, and Disability Adjusted Life Years (DALYs), by age and region for 2010. These parasitic diseases, resulted in 48.4 million cases (95% Uncertainty intervals [UI] of 43.4–79.0 million) and 59,724 (95% UI 48,017–83,616) deaths annually resulting in 8.78 million (95% UI 7.62–12.51 million) DALYs. We estimated that 48% (95% UI 38%-56%) of cases of these parasitic diseases were foodborne, resulting in 76% (95% UI 65%-81%) of the DALYs attributable to these diseases. Overall, foodborne parasitic disease, excluding enteric protozoa, caused an estimated 23.2 million (95% UI 18.2–38.1 million) cases and 45,927 (95% UI 34,763–59,933) deaths annually resulting in an estimated 6.64 million (95% UI 5.61–8.41 million) DALYs. Foodborne Ascaris infection (12.3 million cases, 95% UI 8.29–22.0 million) and foodborne toxoplasmosis (10.3 million cases, 95% UI 7.40–14.9 million) were the most common foodborne parasitic diseases. Human cysticercosis with 2.78 million DALYs (95% UI 2.14–3.61 million), foodborne trematodosis with 2.02 million DALYs (95% UI 1.65–2.48 million) and foodborne toxoplasmosis with 825,000 DALYs (95% UI 561,000–1.26 million) resulted in the highest burdens in terms of DALYs, mainly due to years lived with disability. Foodborne enteric protozoa, reported elsewhere, resulted in an additional 67.2 million illnesses or 492,000 DALYs. Major limitations of our study include often substantial data gaps that had to be filled by imputation and suffer from the uncertainties that surround such models. Due to resource limitations it was also not possible to consider all potentially foodborne parasites (for example Trypanosoma cruzi). Conclusions Parasites are frequently transmitted to humans through contaminated food. These estimates represent an important step forward in understanding the impact of foodborne diseases globally and regionally. The disease burden due to most foodborne parasites is highly focal and results in significant morbidity and mortality among vulnerable populations.
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              Validity of bipolar disorder hospital discharge diagnoses: file review and multiple register linkage in Sweden.

              Hospital discharge registers (HDRs) are frequently used in epidemiological research. However, the validity of several important psychiatric diagnostic entities, including bipolar disorder, remains uncertain. Hence, we aimed to develop an optimal algorithm for register-based identification of DSM-IV-TR bipolar disorder. We identified potential cases in the Swedish national HDR using two separate discharge diagnoses of bipolar disorder according to ICD versions 8-10 during January 1, 1973 to December 31, 2004. In a randomly selected subsample of 135 cases from the county of Sörmland, two senior psychiatrists reassessed the diagnostic status based on patients' medical records. We scrutinized false-positive cases and modified the initial algorithm to improve positive predictive value while minimizing false negatives. Finally, we externally validated resulting caseness algorithms by linking HDR diagnostic data with best-estimate clinical diagnoses from the National Quality Assurance Register for Bipolar Disorder (BipoläR), dispensed lithium prescriptions from the National Prescribed Drug Register, and the ICD-10 diagnoses from the National Outpatient Register respectively. The algorithm with two discharge diagnoses of bipolar disorder yielded a positive predictive value of 0.81. Modification by excluding individuals diagnosed with ICD-8 296.20 (manic-depressive psychosis, depressed type), and/or ICD-9 296.B (unipolar affective psychosis, melancholic form), gave a positive positive predictive value of 0.92. The modified algorithm also had statistically superior external validity compared with the original algorithm. Our findings suggest that DSM-IV-TR bipolar disorder caseness based on two inpatient episodes with a bipolar disorder diagnosis is sufficiently sensitive and specific to be used in further epidemiological study of bipolar disorder. © 2011 John Wiley & Sons A/S.
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                Author and article information

                Contributors
                Role: ConceptualizationRole: Data curationRole: InvestigationRole: MethodologyRole: Project administrationRole: ResourcesRole: SupervisionRole: ValidationRole: VisualizationRole: Writing – original draftRole: Writing – review & editing
                Role: ConceptualizationRole: Data curationRole: Formal analysisRole: InvestigationRole: ResourcesRole: ValidationRole: Writing – original draftRole: Writing – review & editing
                Role: Data curationRole: Formal analysisRole: InvestigationRole: MethodologyRole: SoftwareRole: ValidationRole: Writing – original draftRole: Writing – review & editing
                Role: Data curationRole: MethodologyRole: ResourcesRole: SoftwareRole: Writing – review & editing
                Role: SupervisionRole: Validation
                Role: VisualizationRole: Writing – original draftRole: Writing – review & editing
                Role: Writing – review & editing
                Role: ConceptualizationRole: Project administrationRole: SupervisionRole: ValidationRole: VisualizationRole: Writing – original draftRole: Writing – review & editing
                Role: Editor
                Journal
                PLoS Negl Trop Dis
                PLoS Negl Trop Dis
                plos
                plosntds
                PLoS Neglected Tropical Diseases
                Public Library of Science (San Francisco, CA USA )
                1935-2727
                1935-2735
                26 July 2017
                July 2017
                : 11
                : 7
                : e0005771
                Affiliations
                [1 ] National Reference Laboratory of Cystic Echinococcosis, Istituto zooprofilattico sperimentale della Sardegna, Sassari, Italy
                [2 ] Territorial Department of Nuoro, Istituto zooprofilattico sperimentale della Sardegna, Sassari, Italy
                [3 ] Department of Political Science, Communication Sciences and Information Engineering, University of Sassari, Sassari, Italy
                [4 ] Epidemiological Veterinary Regional Observatory, Istituto zooprofilattico sperimentale della Sardegna, Sassari, Italy
                Texas A&M University College Station, UNITED STATES
                Author notes

                The authors have declared that no competing interests exist.

                Author information
                http://orcid.org/0000-0002-1024-5250
                Article
                PNTD-D-16-02158
                10.1371/journal.pntd.0005771
                5546721
                28746395
                40f2a92e-1999-4f34-8ea0-b8c7772fd23f
                © 2017 Piseddu et al

                This is an open access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.

                History
                : 12 January 2017
                : 3 July 2017
                Page count
                Figures: 7, Tables: 3, Pages: 18
                Funding
                Funded by: Istituto Zooprofilattico Sperimentale della Sardegna
                The authors received no specific funding for this work.
                Categories
                Research Article
                Medicine and Health Sciences
                Parasitic Diseases
                Helminth Infections
                Echinococcosis
                Medicine and Health Sciences
                Tropical Diseases
                Neglected Tropical Diseases
                Echinococcosis
                People and Places
                Population Groupings
                Ethnicities
                Italian People
                People and Places
                Geographical Locations
                Europe
                Italy
                Social Sciences
                Economics
                Health Economics
                Medicine and Health Sciences
                Health Care
                Health Economics
                Medicine and Health Sciences
                Surgical and Invasive Medical Procedures
                Social Sciences
                Economics
                Medicine and Health Sciences
                Public and Occupational Health
                Medicine and Health Sciences
                Parasitic Diseases
                Custom metadata
                vor-update-to-uncorrected-proof
                2017-08-07
                Data are available from the Ministry of Health Institutional Data Access for researchers who meet the criteria for access to confidential data. In compliance with current Italian legislation on privacy, it should be noted that the publication and/or dissemination of data and their processing must take place only in aggregate form. Contact email for the Ministry of Health Institutional Data Acces: -Direzione Generale della Programmazione Sanitaria- email: dp.segreteria@ 123456sanita.it .

                Infectious disease & Microbiology
                Infectious disease & Microbiology

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