Evidence map›Paper›PMID 41169533›Full record

ArticleResearch and practice in thrombosis and haemostasis2025

Deleterious variants cluster in the A3 domain of factor VIII in people with severe hemophilia A and inhibitors.

Luciana Werneck Zuccherato, Renan Pedra Souza, Ricardo Mesquita Camelo, Márcio Antônio Portugal Santana, Maíse Moreira Dias, Letícia Lemos Jardim, Andrea Gonçalves de Oliveira, Claudia Santos Lorenzato, Monica Hermida Cerqueira, Vivian Karla Brognoli Franco and 12 more

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Article in Research and practice in thrombosis and haemostasis, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. An erratum has been issued. Not yet cited in PubMed.

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1 · What the graph read from it

What it found

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2 · The registry

The trial behind it

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3 · Its place in the literature

Who cites it

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4 · The record

Corrections and comments

5 · Who and what money

Authors and funding

22 authors.

Luciana Werneck ZuccheratoUniversidade Federal de Minas Gerais, Belo Horizonte, Brazil.
Renan Pedra SouzaUniversidade Federal de Minas Gerais, Belo Horizonte, Brazil.
Ricardo Mesquita CameloUniversidade Federal de Minas Gerais, Belo Horizonte, Brazil.
Márcio Antônio Portugal SantanaFundação HEMOMINAS, Belo Horizonte, Brazil.
Maíse Moreira DiasUniversidade Federal de Minas Gerais, Belo Horizonte, Brazil.
Letícia Lemos JardimUniversidade Federal de Minas Gerais, Belo Horizonte, Brazil.
Andrea Gonçalves de OliveiraFundação HEMOMINAS, Belo Horizonte, Brazil.
Claudia Santos LorenzatoHemocentro do Paraná (HEMEPAR), Curitiba, Brazil.
Monica Hermida CerqueiraInstituto Estadual de Hematologia do Rio de Janeiro (HEMORIO), Rio de Janeiro, Brazil.
Vivian Karla Brognoli FrancoCentro de Hematologia e Hemoterapia de Santa Catarina (HEMOSC), Florianópolis, Brazil.
Rosangela de Albuquerque RibeiroCentro de Hematologia e Hemoterapia do Ceará (HEMOCE), Fortaleza, Brazil.
Leina Yukari EttoHemocentro da Paraíba (HEMOÍBA), João Pessoa, Brazil.
Maria do Rosario Ferraz RobertiHemocentro de Goiás (HEMOGO), Goiânia, Brazil.
Fábia Michelle Rodrigues de Araújo CalladoFundação de Hematologia e Hemoterapia de Pernambuco (HEMOPE), Recife, Brazil.
Maria Aline Ferreira de CerqueiraCentro de Hematologia e Hemoterapia do Piauí (HEMOPI), Teresina, Brazil.
Ieda Solange de Souza PintoCentro de Hematologia e Hemoterapia do Pará (HEMOPA), Belém, Brazil.
Andrea Aparecida GarciaHemocentro de São José do Rio Preto, São José do Rio Preto, Brazil.
Tania Hissa AnegawaHemocentro Regional de Londrina (HEMEPAR Londrina), Londrina, Brazil.
Daniele Campos Fontes NevesFundação Hemocentro de Rondônia (FHEMERON), Porto Velho, Brazil.
Daniel Gonçalves ChavesFundação HEMOMINAS, Belo Horizonte, Brazil.
Suely Meireles RezendeUniversidade Federal de Minas Gerais, Belo Horizonte, Brazil.
HEMFIL and the Brazilian Immune Tolerance (BrazIT) Study

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Background: Hemophilia A (HA) is an X-linked disorder due to deleterious variants in the factor VIII (FVIII) gene ( Objectives: We aimed to evaluate the spectrum of Methods: We performed genotyping of intron 1 and intron 22 inversions (Inv22) and Results: We included 265 people with severe hemophilia A. We identified deleterious variants in 98.1% of them, totaling 97 unique mutations, of which 32 (33.0%) are novel. Inv22, nonsense, small insertion/deletion, large deletion, and missense variants accounted for 48.3%, 14.7%, 11.3%, 9.8%, and 7.5% of the variants identified. Variants clustered in the FVIII A3 domain were more often associated with people with HA with inhibitors (INH+) than those without inhibitors (INH-) (21.0% vs 0.0%; Conclusion: These findings highlight the role of

Indexed as

alloantibodiesfactor VIII mutationfactor VIII protein domainhemophilia Ainhibitor

Identifiers

PMID41169533
PMCPMC12570200

What Socratic holds

Textmetadata
LicenceCC BY-NC-ND
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Registered trials

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the Socratic graph.