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CD40 Ligand Deficiency in Latin America: Clinical, Immunological, and Genetic Characteristics.
França, Tábata Takahashi; Barreiros, Lucila Akune; Salgado, Ranieri Coelho; Napoleão, Sarah Maria da Silva; Gomes, Lillian Nunes; Ferreira, Janáira Fernandes Severo; Prando, Carolina; Weber, Cristina Worm; Di Gesu, Regina Sumiko Watanabe; Montenegro, Cecilia; Aranda, Carolina Sanchez; Kuntze, Gisele; Staines-Boone, Aidé Tamara; Venegas-Montoya, Edna; Becerra, Juan Carlos Aldave; Bezrodnik, Liliana; Di Giovanni, Daniela; Moreira, Ileana; Seminario, Gisela Analia; Raccio, Andrea Cecilia Gómez; Dorna, Mayra de Barros; Rosário-Filho, Nelson Augusto; Chong-Neto, Herberto Jose; de Carvalho, Elisa; Grotta, Milena Baptistella; Orellana, Julio Cesar; Dominguez, Miguel Garcia; Porras, Oscar; Sasia, Laura; Salvucci, Karina; Garip, Emilio; Leite, Luiz Fernando Bacarini; Forte, Wilma Carvalho Neves; Pinto-Mariz, Fernanda; Goudouris, Ekaterini; Nuñez, María Enriqueta Nuñez; Schelotto, Magdalena; Ruiz, Laura Berrón; Liberatore, Diana Inés; Ochs, Hans D; Cabral-Marques, Otavio; Condino-Neto, Antonio.
Afiliação
  • França TT; Department of Immunology, Institute of Biomedical Sciences, University of São Paulo, São Paulo, Brazil. tabata.t.franca@gmail.com.
  • Barreiros LA; Department of Immunology, Institute of Biomedical Sciences, University of São Paulo, São Paulo, Brazil.
  • Salgado RC; Department of Immunology, Institute of Biomedical Sciences, University of São Paulo, São Paulo, Brazil.
  • Napoleão SMDS; Department of Immunology, Institute of Biomedical Sciences, University of São Paulo, São Paulo, Brazil.
  • Gomes LN; Department of Immunology, Institute of Biomedical Sciences, University of São Paulo, São Paulo, Brazil.
  • Ferreira JFS; Hospital Infantil Albert Sabin, Fortaleza, Brazil.
  • Prando C; Hospital Pequeno Príncipe, Curitiba, Brazil.
  • Weber CW; Faculdades Pequeno Príncipe, Curitiba, Brazil.
  • Di Gesu RSW; Instituto de Pesquisa Pelé Pequeno Príncipe, Curitiba, Brazil.
  • Montenegro C; Centro de Saúde Rocha Weber, Caxias do Sul, Brazil.
  • Aranda CS; Hospital da Criança Conceição, Grupo Hospitalar Conceição, Porto Alegre, Brazil.
  • Kuntze G; Centro Hospitalario Pereira Rossell, Montevideo, Uruguay.
  • Staines-Boone AT; Serviço de Alergia e Imunologia, Departamento de Pediatria, Universidade Federal de São Paulo, São Paulo, Brazil.
  • Venegas-Montoya E; Jeffrey Modell Center São Paulo, São Paulo, Brazil.
  • Becerra JCA; Hospital Pequeno Príncipe, Curitiba, Brazil.
  • Bezrodnik L; Immunology Service, Hospital de Especialidades Unidad Médica de Alta Especialidad (UMAE, Instituto Mexicano del Seguro Social (IMSS), Monterrey, México.
  • Di Giovanni D; Immunology Service, Hospital de Especialidades Unidad Médica de Alta Especialidad (UMAE, Instituto Mexicano del Seguro Social (IMSS), Monterrey, México.
  • Moreira I; Hospital Nacional Edgardo Rebagliati Martins, Lima, Perú.
  • Seminario GA; Grupo de Imunologia, Hospital de Niños Ricardo Gutierrez, Buenos Aires, Argentina.
  • Raccio ACG; Grupo de Imunologia, Hospital de Niños Ricardo Gutierrez, Buenos Aires, Argentina.
  • Dorna MB; Grupo de Imunologia, Hospital de Niños Ricardo Gutierrez, Buenos Aires, Argentina.
  • Rosário-Filho NA; Grupo de Imunologia, Hospital de Niños Ricardo Gutierrez, Buenos Aires, Argentina.
  • Chong-Neto HJ; Grupo de Imunologia, Hospital de Niños Ricardo Gutierrez, Buenos Aires, Argentina.
  • de Carvalho E; Divisão de Alergia e Imunologia, Departamento de Pediatria, Instituto da Criança, Hospital das Clínicas da Faculdade de Medicina da Universidade de São Paulo, São Paulo, Brazil.
  • Grotta MB; Serviço de Alergia e Imunologia, Universidade Federal do Paraná, Curitiba, Brazil.
  • Orellana JC; Serviço de Alergia e Imunologia, Universidade Federal do Paraná, Curitiba, Brazil.
  • Dominguez MG; Gastroenterology and Hepatology Clínic, Brasilia Childrens Hospital, Brasília, Brazil.
  • Porras O; Department of Pediatrics, State University of Campinas Medical School, Campinas, Brazil.
  • Sasia L; Division Alergia e Imunologia Clinica, Hospital de Niños de La Santísima Trinidad, Córdoba, Argentina.
  • Salvucci K; Hospital Pediatrico de Sinaloa Rigoberto Aguilar Pico, Culiacan, México.
  • Garip E; Hospital Nacional de Niños Dr. Carlos Sáenz Herrera, San José, Costa Rica.
  • Leite LFB; Hospital Infantil Municipal de Córdoba, Córdoba, Argentina.
  • Forte WCN; Hospital Infantil Municipal de Córdoba, Córdoba, Argentina.
  • Pinto-Mariz F; Hospital Infantil Municipal de Córdoba, Córdoba, Argentina.
  • Goudouris E; Department of Pediatrics, Immunodeficiency Sector, Irmandade da Santa Casa de Misericórdia de São Paulo, São Paulo, Brazil.
  • Nuñez MEN; Immunology Discipline, Santa Casa de São Paulo School of Medical Sciences, São Paulo, Brazil.
  • Schelotto M; Department of Pediatrics, School of Medicine, Federal University of Rio de Janeiro, Rio de Janeiro, Brazil.
  • Ruiz LB; Department of Pediatrics, School of Medicine, Federal University of Rio de Janeiro, Rio de Janeiro, Brazil.
  • Liberatore DI; Department of Pediatrics, Division of Pediatric Allergy and Immunology, Hospital Civil de Guadalajara Dr. Juan I. Menchaca, Guadalajara, México.
  • Ochs HD; Centro Hospitalario Pereira Rossell, Montevideo, Uruguay.
  • Cabral-Marques O; Unidad de Investigación en Inmunodeficiencias, Instituto Nacional de Pediatría, Ciudad del México, México.
  • Condino-Neto A; Hospital Italiano Buenos Aires, Buenos Aires, Argentina.
J Clin Immunol ; 42(3): 514-526, 2022 04.
Article em En | MEDLINE | ID: mdl-34982304
CD40 ligand (CD40L) deficiency is a rare inborn error of immunity presenting with heterogeneous clinical manifestations. While a detailed characterization of patients affected by CD40L deficiency is essential to an accurate diagnosis and management, information about this disorder in Latin American patients is limited. We retrospectively analyzed data from 50 patients collected by the Latin American Society for Immunodeficiencies registry or provided by affiliated physicians to characterize the clinical, laboratory, and molecular features of Latin American patients with CD40L deficiency. The median age at disease onset and diagnosis was 7 months and 17 months, respectively, with a median diagnosis delay of 1 year. Forty-seven patients were genetically characterized revealing 6 novel mutations in the CD40LG gene. Pneumonia was the most common first symptom reported (66%). Initial immunoglobulin levels were variable among patients. Pneumonia (86%), upper respiratory tract infections (70%), neutropenia (70%), and gastrointestinal manifestations (60%) were the most prevalent clinical symptoms throughout life. Thirty-five infectious agents were reported, five of which were not previously described in CD40L deficient patients, representing the largest number of pathogens reported to date in a cohort of CD40L deficient patients. The characterization of the largest cohort of Latin American patients with CD40L deficiency adds novel insights to the recognition of this disorder, helping to fulfill unmet needs and gaps in the diagnosis and management of patients with CD40L deficiency.
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Texto completo: 1 Base de dados: MEDLINE Assunto principal: Ligante de CD40 / Síndromes de Imunodeficiência Tipo de estudo: Diagnostic_studies / Etiology_studies / Incidence_studies / Observational_studies / Risk_factors_studies Limite: Humans Idioma: En Revista: J Clin Immunol Ano de publicação: 2022 Tipo de documento: Article País de afiliação: Brasil

Texto completo: 1 Base de dados: MEDLINE Assunto principal: Ligante de CD40 / Síndromes de Imunodeficiência Tipo de estudo: Diagnostic_studies / Etiology_studies / Incidence_studies / Observational_studies / Risk_factors_studies Limite: Humans Idioma: En Revista: J Clin Immunol Ano de publicação: 2022 Tipo de documento: Article País de afiliação: Brasil