| Literature DB >> 34982304 |
Tábata Takahashi França1, Lucila Akune Barreiros2, Ranieri Coelho Salgado2, Sarah Maria da Silva Napoleão2, Lillian Nunes Gomes2, Janáira Fernandes Severo Ferreira3, Carolina Prando4,5,6, Cristina Worm Weber7, Regina Sumiko Watanabe Di Gesu8, Cecilia Montenegro9, Carolina Sanchez Aranda10,11, Gisele Kuntze4, Aidé Tamara Staines-Boone12, Edna Venegas-Montoya12, Juan Carlos Aldave Becerra13, Liliana Bezrodnik14, Daniela Di Giovanni14, Ileana Moreira14, Gisela Analia Seminario14, Andrea Cecilia Gómez Raccio14, Mayra de Barros Dorna15, Nelson Augusto Rosário-Filho16, Herberto Jose Chong-Neto16, Elisa de Carvalho17, Milena Baptistella Grotta18, Julio Cesar Orellana19, Miguel Garcia Dominguez20, Oscar Porras21, Laura Sasia22, Karina Salvucci22, Emilio Garip22, Luiz Fernando Bacarini Leite23, Wilma Carvalho Neves Forte24, Fernanda Pinto-Mariz25, Ekaterini Goudouris25, María Enriqueta Nuñez Nuñez26, Magdalena Schelotto9, Laura Berrón Ruiz27, Diana Inés Liberatore28, Hans D Ochs29, Otavio Cabral-Marques2,30,31, Antonio Condino-Neto32,33.
Abstract
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.Entities:
Keywords: CD40 ligand; CD40L deficiency; Inborn error of immunity; Latin American Society for Immunodeficiencies registry; Primary immunodeficiency; X-HIGM
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Year: 2022 PMID: 34982304 DOI: 10.1007/s10875-021-01182-5
Source DB: PubMed Journal: J Clin Immunol ISSN: 0271-9142 Impact factor: 8.317