{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,7]],"date-time":"2026-07-07T03:00:12Z","timestamp":1783393212123,"version":"3.54.6"},"reference-count":39,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2024,8,23]],"date-time":"2024-08-23T00:00:00Z","timestamp":1724371200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["frontiersin.org"],"crossmark-restriction":true},"short-container-title":["Front. Med."],"abstract":"<jats:sec><jats:title>Background<\/jats:title><jats:p>Reports on the association between HTLV-2 infection and the development of diseases in the human host are rare, which has led the scientific community to believe that HTLV-2 is not an important etiological agent of lymphoproliferative or neurodegenerative disorders, which is the case for HTLV-1. In the present study, we demonstrated cases of fibromyalgia in HTLV-1 carriers and, in an unprecedented finding, in two patients with confirmed HTLV-2 infection.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>A total of 957 individuals visited the Virology Laboratory at the Federal University of Par\u00e1 for screening and confirmation tests for HTLV-1\/2 infection. Individuals with confirmed HTLV-1 and HTLV-2 infection were clinically evaluated for signs and symptoms associated with infection.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Sixty-nine individuals (7.2%) were identified as positive for HTLV infection, with 56 confirmed cases of HTLV-1 infection (5.9%), 12 cases of HTLV-2 infection (1.2%) and one case classified as undetermined (0.1%). Sixteen (23.2%) of these patients presented with rheumatological signs and complained of diffuse pain throughout the body; 12 of whom were infected by HTLV-1 (75%) and 4 were infected by HTLV-2 (25%). After anamnesis and careful evaluation, four patients were diagnosed with fibromyalgia, two of whom were infected by HTLV-1 (16.7%; 2\/12) and two by HTLV-2 (50%; 2\/4). The clinical follow-up and laboratory analysis results are reported in detail in this paper.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>Considering the clinical cases presented herein as the first reports of patients with HTLV-2 infection with clinical symptoms of fibromyalgia, the importance of further studies on the pathogenicity of HTLV-2, similar to what have already been performed for HTLV-1, is highlighted. Our results also confirm previous evidence of an association between HTLV-1 infection and fibromyalgia.<\/jats:p><\/jats:sec>","DOI":"10.3389\/fmed.2024.1419801","type":"journal-article","created":{"date-parts":[[2024,8,23]],"date-time":"2024-08-23T04:33:19Z","timestamp":1724387599000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":5,"title":["Fibromyalgia in patients infected with HTLV-1 and HTLV-2"],"prefix":"10.3389","volume":"11","author":[{"given":"Bianca Lumi","family":"Inomata Silva","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Francisco Erivan","family":"da Cunha Rodrigues","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"M\u00e1rcio Yutaka","family":"Tsukimata","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Bruno Jos\u00e9 Sarmento","family":"Botelho","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Luciana Cristina Coelho","family":"Santos","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gabriel","family":"dos Santos Pereira Neto","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Aline Cecy Rocha","family":"Lima","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nat\u00e1lia Pinheiro","family":"Andr\u00e9","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sarah Marques","family":"Galdino","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Danniele Chagas","family":"Monteiro","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gilberto Toshimitsu","family":"Yoshikawa","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Leonardo Teixeira","family":"Mendon\u00e7a","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Juliana Lasmar Ayres","family":"do Amaral","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rosana","family":"de Britto Pereira Cruz","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"D\u00e9bora Oliveira","family":"Onuma","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Antonio Carlos Ros\u00e1rio","family":"Vallinoto","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"B\u00e1rbara Nascimento","family":"de Carvalho Klemz","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Izaura Maria Vieira Cayres","family":"Vallinoto","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1965","published-online":{"date-parts":[[2024,8,23]]},"reference":[{"key":"ref1","volume-title":"Human T-lymphotropic virus type 1: technical report. Geneva: Licence: CC BY-NC-SA 3.0 IGO","year":"2021"},{"key":"ref2","doi-asserted-by":"publisher","first-page":"7415","DOI":"10.1073\/pnas.77.12.7415","article-title":"Detection and isolation of type C retrovirus particles from fresh and cultured lymphocytes of a patient with cutaneous T-cell lymphoma","volume":"77","author":"Poiesz","year":"1980","journal-title":"Proc Natl Acad Sci USA"},{"key":"ref3","doi-asserted-by":"publisher","first-page":"571","DOI":"10.1126\/science.6981847","article-title":"A new subtype of human T-cell leukemia virus (HTLV-II) associated with a T-cell variant of hairy cell leukemia","volume":"218","author":"Kalyanaraman","year":"1982","journal-title":"Science"},{"key":"ref4","doi-asserted-by":"publisher","first-page":"133","DOI":"10.1016\/S1473-3099(19)30402-5","article-title":"Association between HTLV-1 infection and adverse health outcomes: a systematic review and meta-analysis of epidemiological studies","volume":"20","author":"Schierhout","year":"2020","journal-title":"Lancet Infect Dis"},{"key":"ref5","doi-asserted-by":"publisher","first-page":"10","DOI":"10.1002\/ana.20126","article-title":"Human T-lymphotropic virus type II and neurological disease","volume":"56","author":"Araujo","year":"2004","journal-title":"Ann Neurol"},{"key":"ref6","first-page":"337","article-title":"Viral infection and fibromyalgia","volume":"7","author":"Branco","year":"1994","journal-title":"Acta Medica Port"},{"key":"ref7","doi-asserted-by":"publisher","first-page":"100155","DOI":"10.1016\/j.jcvp.2023.100155","article-title":"Unique evidence of atypical lymphocytes and flower cells in indigenous Xikrin do Bacaj\u00e1 people infected with HTLV-2","volume":"3","author":"Vallinoto","year":"2023","journal-title":"J Clin Virol Plus"},{"key":"ref8","doi-asserted-by":"publisher","first-page":"3891","DOI":"10.3390\/ijms22083891","article-title":"Fibromyalgia: pathogenesis, mechanisms, diagnosis and treatment options update","volume":"22","author":"Siracusa","year":"2021","journal-title":"Int J Mol Sci"},{"key":"ref9","first-page":"33","article-title":"The fibrosits syndrome","volume":"3","author":"Graham","year":"1953","journal-title":"Bull Rheum Dis"},{"key":"ref10","first-page":"928","article-title":"Two contributions to understanding of the \"fibrositis\" syndrome","volume":"28","author":"Smythe","year":"1977","journal-title":"Bull Rheum Dis"},{"key":"ref11","first-page":"S118","article-title":"Chronic widespread pain and fibromyalgia: could there be some relationships with infections and vaccinations?","volume":"29","author":"Cassisi","year":"2011","journal-title":"Clin Exp Rheumatol"},{"key":"ref12","doi-asserted-by":"publisher","first-page":"332","DOI":"10.1186\/1743-422X-10-332","article-title":"Lack of evidence for retroviral infections formerly related to chronic fatigue in Spanish fibromyalgia patients","volume":"10","author":"Oltra","year":"2013","journal-title":"Virol J"},{"key":"ref13","first-page":"2300","article-title":"Higher prevalence of fibromyalgia in patients infected with human T cell lymphotropic virus type I","volume":"33","author":"Cruz","year":"2006","journal-title":"J Rheumatol"},{"key":"ref14","doi-asserted-by":"publisher","first-page":"864861","DOI":"10.3389\/fpubh.2022.864861","article-title":"The relevance of a diagnostic and counseling service for people living with HTLV-1\/2 in a metropolis of the Brazilian Amazon","volume":"10","author":"Lopes","year":"2022","journal-title":"Front Public Health"},{"key":"ref15","doi-asserted-by":"publisher","first-page":"e00005419","DOI":"10.1590\/0102-311X00005419","article-title":"HTLV: a stigmatizing infection?","volume":"35","author":"Garcia","year":"2019","journal-title":"Cad Saude Publica"},{"key":"ref16","doi-asserted-by":"publisher","first-page":"622","DOI":"10.1093\/trstmh\/trab173","article-title":"Neurodegenerative disease in association with sexual transmission of human T-cell lymphotropic virus type 2 subtype b in Argentina","volume":"116","author":"Blanco","year":"2022","journal-title":"Trans R Soc Trop Med Hyg"},{"key":"ref17","doi-asserted-by":"publisher","first-page":"228","DOI":"10.12688\/f1000research.17479.1","article-title":"40 years of the human T-cell leukemia virus: past, present, and future","volume":"8","author":"Tagaya","year":"2019","journal-title":"F1000 Faculty Rev"},{"key":"ref18","doi-asserted-by":"publisher","first-page":"61","DOI":"10.1515\/nipt-2023-0025","article-title":"Virology, pathogenesis, epidemiology and clinical management of HTLV-1 infection","volume":"3","author":"de Mendoza","year":"2024","journal-title":"Proc 30th HTLV Eur Res Net (HERN 2023) NeuroImmune Pharmacol Ther"},{"key":"ref19","doi-asserted-by":"publisher","first-page":"11685","DOI":"10.1073\/pnas.1920346117","article-title":"Mortality and risk of progression to adult T cell leukemia\/lymphoma in HTLV-1-associated myelopathy\/tropical spastic paraparesis","volume":"117","author":"Nagasaka","year":"2020","journal-title":"Proc Natl Acad Sci USA"},{"key":"ref20","doi-asserted-by":"publisher","first-page":"1","DOI":"10.3390\/v10010001","article-title":"HTLV-1, the other pathogenic yet neglected human retrovirus: from transmission to therapeutic treatment","volume":"10","author":"Futsch","year":"2017","journal-title":"Viruses"},{"key":"ref21","doi-asserted-by":"publisher","first-page":"1893","DOI":"10.1016\/S0140-6736(18)30974-7","article-title":"Time to eradicate HTLV-1: an open letter to WHO","volume":"391","author":"Martin","year":"2018","journal-title":"Lancet"},{"key":"ref22","doi-asserted-by":"publisher","first-page":"1546","DOI":"10.3390\/ph16111546","article-title":"New perspectives about drug candidates targeting HTLV-1 and related diseases","volume":"16","author":"Silva","year":"2023","journal-title":"Pharmaceuticals"},{"key":"ref23","doi-asserted-by":"publisher","first-page":"1073779","DOI":"10.3389\/fimmu.2023.1073779","article-title":"Advances in preventive vaccine development against HTLV-1 infection: a systematic review of the last 35 years","volume":"14","author":"Santana","year":"2023","journal-title":"Front Immunol"},{"key":"ref24","doi-asserted-by":"publisher","first-page":"4","DOI":"10.1186\/s12977-020-0512-z","article-title":"The challenge of describing the epidemiology of HTLV in the Amazon region of Brazil","volume":"17","author":"Ishak","year":"2020","journal-title":"Retrovirology"},{"key":"ref25","doi-asserted-by":"publisher","first-page":"1217134","DOI":"10.3389\/fmicb.2023.1217134","article-title":"Long-term prevalence follow-up (1967-2022) of HTLV-2 among vulnerable indigenous populations in the Amazon region of Brazil","volume":"14","author":"Ishak","year":"2023","journal-title":"Front Microbiol"},{"key":"ref26","doi-asserted-by":"publisher","first-page":"e2020605","DOI":"10.1590\/0037-8682-605-2020","article-title":"Brazilian protocol for sexually transmitted infections 2020: human T-cell lymphotropic virus (HTLV) infection","volume":"54","author":"Rosadas","year":"2021","journal-title":"Rev Soc Bras Med Trop"},{"key":"ref27","doi-asserted-by":"publisher","first-page":"e1705","DOI":"10.1371\/journal.pntd.0001705","article-title":"Living invisible: HTLV-1-infected persons and the lack of care in public health","volume":"6","author":"Zihlmann","year":"2012","journal-title":"PLoS Negl Trop Dis"},{"key":"ref28","doi-asserted-by":"publisher","first-page":"497","DOI":"10.1016\/j.bjid.2020.08.009","article-title":"Limitations in daily activities, risk awareness, social participation, and pain in patients with HTLV-1 using the SALSA and participation scales","volume":"24","author":"Aben-Athar","year":"2020","journal-title":"Braz J Infect Dis"},{"key":"ref29","doi-asserted-by":"publisher","first-page":"859889","DOI":"10.3389\/fmed.2022.859889","article-title":"Occupational therapy for people living with human T-Lymphotropic virus 1: importance and effectiveness of an intervention plan at the level of prevention and rehabilitation","volume":"9","author":"Sampaio","year":"2022","journal-title":"Front Med"},{"key":"ref30","doi-asserted-by":"publisher","first-page":"54","DOI":"10.1016\/j.genhosppsych.2021.08.012","article-title":"Prevalence and factors associated with depression and anxiety in people living with HTLV-1: a systematic review with meta-analysis and meta-regression","volume":"73","author":"Souza","year":"2021","journal-title":"Gen Hosp Psychiatry"},{"key":"ref31","doi-asserted-by":"publisher","first-page":"60","DOI":"10.1016\/S0140-6736(22)00821-2","article-title":"Complex post-traumatic stress disorder","volume":"400","author":"Maercker","year":"2022","journal-title":"Lancet"},{"key":"ref32","doi-asserted-by":"publisher","first-page":"891","DOI":"10.1080\/14737175.2020.1794824","article-title":"Fibromyalgia patients should always be screened for post-traumatic stress disorder","volume":"20","author":"Nardi","year":"2020","journal-title":"Expert Rev Neurother"},{"key":"ref33","doi-asserted-by":"publisher","first-page":"311","DOI":"10.1080\/08964289.2020.1758611","article-title":"Blunted pain modulation response to induced stress in women with fibromyalgia with and without posttraumatic stress disorder comorbidity: new evidence of hypo-reactivity to stress in fibromyalgia?","volume":"47","author":"L\u00f3pez-L\u00f3pez","year":"2021","journal-title":"Behav Med"},{"key":"ref34","doi-asserted-by":"publisher","first-page":"143","DOI":"10.1111\/pcn.12820","article-title":"Inflammation and post-traumatic stress disorder","volume":"73","author":"Hori","year":"2019","journal-title":"Psychiatry Clin Neurosci"},{"key":"ref35","first-page":"1941","article-title":"Established fibromyalgia syndrome and parvovirus B19 infection","volume":"20","author":"Berg","year":"1993","journal-title":"J Rheumatol"},{"key":"ref36","doi-asserted-by":"publisher","first-page":"194","DOI":"10.1007\/s13365-018-0703-8","article-title":"The role of HHV-6 and HHV-7 infections in the development of fibromyalgia","volume":"25","author":"Krumina","year":"2019","journal-title":"J Neurovirol"},{"key":"ref37","doi-asserted-by":"publisher","first-page":"121","DOI":"10.1016\/j.ijid.2019.05.010","article-title":"Cognitive impairment in HTLV-1-associated myelopathy, proviral load and inflammatory markers","volume":"84","author":"Champs","year":"2019","journal-title":"Int J Infect Dis"},{"key":"ref38","doi-asserted-by":"publisher","first-page":"e44","DOI":"10.1017\/S1463423621000773","article-title":"Amigos de fibro (fibro friends): development of an educational program for the health promotion of fibromyalgia patients","volume":"23","author":"Antunes","year":"2022","journal-title":"Prim Health Care Res Dev"},{"key":"ref39","doi-asserted-by":"publisher","first-page":"22","DOI":"10.3390\/v15010022","article-title":"HTLV-1\/2 in indigenous peoples of the Brazilian Amazon: seroprevalence, molecular characterization and socio behavioral factors related to risk of infection","volume":"15","author":"Abreu","year":"2023","journal-title":"Viruses"}],"container-title":["Frontiers in Medicine"],"original-title":[],"link":[{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fmed.2024.1419801\/full","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2024,8,23]],"date-time":"2024-08-23T04:33:27Z","timestamp":1724387607000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fmed.2024.1419801\/full"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,8,23]]},"references-count":39,"alternative-id":["10.3389\/fmed.2024.1419801"],"URL":"https:\/\/doi.org\/10.3389\/fmed.2024.1419801","relation":{},"ISSN":["2296-858X"],"issn-type":[{"value":"2296-858X","type":"electronic"}],"subject":[],"published":{"date-parts":[[2024,8,23]]},"article-number":"1419801"}}