Environmental risk factors for inhibitor development in children with haemophilia A

A case-control study

Elena Santagostino, Maria Elisa Mancuso, Angiola Rocino, Giacomo Mancuso, Maria Gabriella Mazzucconi, Annarita Tagliaferri, Maria Messina, Pier Mannuccio Mannucci

Research output: Contribution to journalArticle

163 Citations (Scopus)

Abstract

This case-control study investigated the interactions between genetic and environmental factors and inhibitor development in 108 children with haemophilia A exclusively treated with recombinant factor VIII (FVIII). Sixty patients with inhibitors were compared with 48 inhibitor-free controls. Family history of inhibitors and null mutations in the FVIII gene were more prevalent in cases than in controls (20% vs. 2%, P = 0·001 and 83% vs. 64%, P = 0·04, respectively). On the other hand, there was no difference between cases and controls for such putative risk factors of inhibitor development as amniocentesis/villocentesis, premature/caesarean birth, breast-feeding, treatment during infections/vaccinations, surgical procedures and central nervous system bleeding. A trend was found for an increased risk of inhibitor development in children first treated at a young age (

Original languageEnglish
Pages (from-to)422-427
Number of pages6
JournalBritish Journal of Haematology
Volume130
Issue number3
DOIs
Publication statusPublished - Aug 2005

Fingerprint

Factor VIII
Hemophilia A
Child Development
Case-Control Studies
Amniocentesis
Premature Birth
Breast Feeding
Vaccination
Central Nervous System
Hemorrhage
Mutation
Infection
Genes
Therapeutics

Keywords

  • FVIII gene mutations
  • Haemophilia
  • Inhibitors
  • Prophylaxis
  • Risk factors

ASJC Scopus subject areas

  • Hematology

Cite this

Environmental risk factors for inhibitor development in children with haemophilia A : A case-control study. / Santagostino, Elena; Mancuso, Maria Elisa; Rocino, Angiola; Mancuso, Giacomo; Mazzucconi, Maria Gabriella; Tagliaferri, Annarita; Messina, Maria; Mannucci, Pier Mannuccio.

In: British Journal of Haematology, Vol. 130, No. 3, 08.2005, p. 422-427.

Research output: Contribution to journalArticle

Santagostino, Elena ; Mancuso, Maria Elisa ; Rocino, Angiola ; Mancuso, Giacomo ; Mazzucconi, Maria Gabriella ; Tagliaferri, Annarita ; Messina, Maria ; Mannucci, Pier Mannuccio. / Environmental risk factors for inhibitor development in children with haemophilia A : A case-control study. In: British Journal of Haematology. 2005 ; Vol. 130, No. 3. pp. 422-427.
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