Literature DB >> 34982304

CD40 Ligand Deficiency in Latin America: Clinical, Immunological, and Genetic Characteristics.

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.
© 2022. The Author(s), under exclusive licence to Springer Science+Business Media, LLC, part of Springer Nature.

Entities:  

Keywords:  CD40 ligand; CD40L deficiency; Inborn error of immunity; Latin American Society for Immunodeficiencies registry; Primary immunodeficiency; X-HIGM

Mesh:

Substances:

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


  51 in total

1.  First report of the Hyper-IgM syndrome Registry of the Latin American Society for Immunodeficiencies: novel mutations, unique infections, and outcomes.

Authors:  Otavio Cabral-Marques; Stefanie Klaver; Lena F Schimke; Évelyn H Ascendino; Taj Ali Khan; Paulo Vítor Soeiro Pereira; Angela Falcai; Alexander Vargas-Hernández; Leopoldo Santos-Argumedo; Liliana Bezrodnik; Ileana Moreira; Gisela Seminario; Daniela Di Giovanni; Andrea Gómez Raccio; Oscar Porras; Cristina Worm Weber; Janaíra Fernandes Ferreira; Fabiola Scancetti Tavares; Elisa de Carvalho; Claudia França Cavalcante Valente; Gisele Kuntze; Miguel Galicchio; Alejandra King; Nelson Augusto Rosário-Filho; Milena Baptistella Grota; Maria Marluce dos Santos Vilela; Regina Sumiko Watanabe Di Gesu; Simone Lima; Leiva de Souza Moura; Eduardo Talesnik; Eli Mansour; Pérsio Roxo-Junior; Juan Carlos Aldave; Ekaterine Goudouris; Fernanda Pinto-Mariz; Laura Berrón-Ruiz; Tamara Staines-Boone; Wilmer O Córdova Calderón; María del Carmen Zarate-Hernández; Anete S Grumach; Ricardo Sorensen; Anne Durandy; Troy R Torgerson; Beatriz Tavares Costa Carvalho; Francisco Espinosa-Rosales; Hans D Ochs; Antonio Condino-Neto
Journal:  J Clin Immunol       Date:  2014-01-09       Impact factor: 8.317

Review 2.  Update on the hyper immunoglobulin M syndromes.

Authors:  E Graham Davies; Adrian J Thrasher
Journal:  Br J Haematol       Date:  2010-02-23       Impact factor: 6.998

Review 3.  The hyper IgM syndromes: Epidemiology, pathogenesis, clinical manifestations, diagnosis and management.

Authors:  Reza Yazdani; Saba Fekrvand; Sepideh Shahkarami; Gholamreza Azizi; Bobak Moazzami; Hassan Abolhassani; Asghar Aghamohammadi
Journal:  Clin Immunol       Date:  2018-11-13       Impact factor: 3.969

4.  X-linked hyper IgM syndrome: clinical, immunological and molecular features in patients from India.

Authors:  Manisha Madkaikar; Maya Gupta; Sushant Chavan; Khushnooma Italia; Mukesh Desai; Rashid Merchant; Nita Radhakrishnan; Kanjaksha Ghosh
Journal:  Blood Cells Mol Dis       Date:  2014-06-11       Impact factor: 3.039

Review 5.  The hyper IgM syndromes.

Authors:  Nashmia Qamar; Ramsay L Fuleihan
Journal:  Clin Rev Allergy Immunol       Date:  2014-04       Impact factor: 8.667

6.  Expanding the clinical and genetic spectrum of human CD40L deficiency: the occurrence of paracoccidioidomycosis and other unusual infections in Brazilian patients.

Authors:  Otavio Cabral-Marques; Lena-Friederike Schimke; Paulo Vítor Soeiro Pereira; Angela Falcai; João Bosco de Oliveira; Mary J Hackett; Paolo Ruggero Errante; Cristina Worm Weber; Janaíra Fernandes Ferreira; Gisele Kuntze; Nelson Augusto Rosário-Filho; Hans D Ochs; Troy R Torgerson; Beatriz Tavares Costa Carvalho; Antonio Condino-Neto
Journal:  J Clin Immunol       Date:  2011-12-23       Impact factor: 8.317

Review 7.  Clinical Phenotypes of Hyper-IgM Syndromes.

Authors:  M Teresa de la Morena
Journal:  J Allergy Clin Immunol Pract       Date:  2016 Nov - Dec

8.  Long-term outcomes of 176 patients with X-linked hyper-IgM syndrome treated with or without hematopoietic cell transplantation.

Authors:  M Teresa de la Morena; David Leonard; Troy R Torgerson; Otavio Cabral-Marques; Mary Slatter; Asghar Aghamohammadi; Sharat Chandra; Luis Murguia-Favela; Francisco A Bonilla; Maria Kanariou; Rongras Damrongwatanasuk; Caroline Y Kuo; Christopher C Dvorak; Isabelle Meyts; Karin Chen; Lisa Kobrynski; Neena Kapoor; Darko Richter; Daniela DiGiovanni; Fatima Dhalla; Evangelia Farmaki; Carsten Speckmann; Teresa Español; Anna Shcherbina; Imelda Celine Hanson; Jiri Litzman; John M Routes; Melanie Wong; Ramsay Fuleihan; Suranjith L Seneviratne; Trudy N Small; Ales Janda; Liliana Bezrodnik; Reinhard Seger; Andrea Gomez Raccio; J David M Edgar; Janet Chou; Jordan K Abbott; Joris van Montfrans; Luis Ignacio González-Granado; Nancy Bunin; Necil Kutukculer; Paul Gray; Gisela Seminario; Srdjan Pasic; Victor Aquino; Christian Wysocki; Hassan Abolhassani; Morna Dorsey; Charlotte Cunningham-Rundles; Alan P Knutsen; John Sleasman; Beatriz Tavares Costa Carvalho; Antonio Condino-Neto; Eyal Grunebaum; Helen Chapel; Hans D Ochs; Alexandra Filipovich; Mort Cowan; Andrew Gennery; Andrew Cant; Luigi D Notarangelo; Chaim M Roifman
Journal:  J Allergy Clin Immunol       Date:  2016-09-30       Impact factor: 10.793

9.  Morbidity and mortality of Iranian patients with hyper IgM syndrome: a clinical analysis.

Authors:  Hassan Abolhassani; Fatemeh Akbari; Babak Mirminachi; Saeed Bazregari; Ehsan Hedayat; Nima Rezaei; Asghar Aghamohammadi
Journal:  Iran J Immunol       Date:  2014-06       Impact factor: 1.603

10.  Hyper IgM Syndrome: a Report from the USIDNET Registry.

Authors:  Emily A Leven; Patrick Maffucci; Hans D Ochs; Paul R Scholl; Rebecca H Buckley; Ramsay L Fuleihan; Raif S Geha; Coleen K Cunningham; Francisco A Bonilla; Mary Ellen Conley; Ronald M Ferdman; Vivian Hernandez-Trujillo; Jennifer M Puck; Kathleen Sullivan; Elizabeth A Secord; Manish Ramesh; Charlotte Cunningham-Rundles
Journal:  J Clin Immunol       Date:  2016-05-17       Impact factor: 8.542

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