| Literature DB >> 35796998 |
Laura Bannach Jardim1,2, Ali Hasan3, Sheng-Han Kuo4, Jonathan Javier Magaña5, Marcondes França6, Wilson Marques7, Claudia Camejo8, Luiz Carlos Santana-da-Silva9, Emília Embiruçu Leão10, Gisele Espíndola11, Francisca Canals12, Marcelo Miranda13, Igor Salvatierra14, Mario Cornejo-Olivas15, Juan Fernandez-Ruiz16, Pedro Braga-Neto17, David José Dávila-Ortiz de Montellano18, Luis Leonardo Flores-Lagunes19, Nicolas Dupré20, Bernard Brais21, Fernando Regla Vargas22, Clécio Godeiro23, Léo Coutinho24, Helio G Teive24, Marcelo Kaufmann25, Paula Saffie26, Gabriel Vasata Furtado27, Maria Luiza Saraiva-Pereira27,28, Orlando Barsottini29, José Luiz Pedroso29, Roberto Rodríguez-Labrada30,31, Luis Velázquez-Pérez31,32, Christopher Gomez33.
Abstract
Little is known about access of rare disease carriers to health care. To increase this knowledge, the Pan American Hereditary Ataxia Network (PAHAN) conducted an exploratory survey about care for hereditary ataxias in American continents and the Caribbean. A questionnaire was sent to health professionals about the hereditary ataxias identified; access to care; and local teaching and research. The number of ataxics under current care per 100,000 inhabitants was subtracted from the expected overall prevalence of 6/100,000, to estimate the prevalence of uncovered ataxic patients. Local Human Development Indexes (HDI) were used to measure socio-economic factors. Twenty-six sites participated. Twelve sites had very high, 13 had high, and one site had medium HDI. Participants reported on 2239 and 602 patients with spinocerebellar ataxias and recessive forms under current care. The number of patients under current care per inhabitants varied between 0.14 and 12/100,000. The estimated prevalence of uncovered ataxic patients was inversely proportional to HDIs (rho = 0.665, p = 0.003). Access to diagnosis, pre-symptomatic tests, and rehabilitation were associated with HDIs. More and better molecular diagnostic tools, protocols and guidelines, and professional training for ataxia care were the top priorities common to all respondents. Evidence of inequalities was confirmed. Lower HDIs were associated with high potential numbers of uncovered ataxic subjects, and with lack of molecular diagnosis, pre-symptomatic testing, and rehabilitation. More and better diagnostic tools, guidelines, and professional training were priorities to all sites. PAHAN consortium might help with the last two tasks.Entities:
Keywords: Access to health care; American continents; Inherited ataxias; The Caribbean
Year: 2022 PMID: 35796998 DOI: 10.1007/s12311-022-01442-z
Source DB: PubMed Journal: Cerebellum ISSN: 1473-4222 Impact factor: 3.847