: Congenital factor XI (FXI) deficiency is a mild trauma-related bleeding disorder with estimated worldwide prevalence of one per 1 million. The disorder is less frequent in Iran and a few studies have been performed on Iranian patients. In the current study, we assessed molecular, laboratory and clinical features of two Iranian patients with congenital FXI deficiency and their families. Clinical features and demographic data of the patients were assessed by the physician and a staff member trained specifically to deal with patients with bleeding disorders. FXI activity and antigen assays were performed for seven members of the two families and genotyping was performed by direct sequencing of all F11 gene exons and intron-exon boundaries as well as the untranslated regions. Five members of the two families were affected by FXI deficiency. Both patients experienced prolonged epistaxis, whereas other family members were asymptomatic. Two gene defects were observed in the patients and their families. Two disease-causing mutations were c.943G>A (p.Glu315Lys) missense and the four-nucleotide deletion (g.27849-27852del) in exon 15. The gene deletion was observed in homozygote state in the patient with severe FXI deficiency (FXI activity <1%) and heterozygote state in the parent, whereas the c.943G>A mutation was detected in heterozygote state and was accompanied by epistaxis in the patient. FXI deficiency is a mild bleeding disorder that is caused by heterogeneous molecular defects.
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http://dx.doi.org/10.1097/MBC.0000000000000862 | DOI Listing |
J Pediatr Hematol Oncol
December 2024
Pediatrics Clinic, Bakirkoy Dr. Sadi Konuk Training and Research Hospital, University of Health Sciences, Istanbul, Turkey.
Background: Rare factor deficiency (RFD) is characterized by a deficiency of factor (F)I, FII, FV, FVII, FX, FXI, FXII, FXIII, or a combined deficiency of FV+FVIII or vitamin K-dependent factors. The prevalence of RFD ranges from 1/1,000,000 to 3,000,000. Combined deficiencies of vitamin K-related factors have been described in 30 families worldwide, and these patients can present with a wide range of clinical symptoms, from mucocutaneous bleeding to life-threatening symptoms such as central nervous system and gastrointestinal bleeding.
View Article and Find Full Text PDFRev Esp Anestesiol Reanim (Engl Ed)
December 2024
Servicio de Anestesiología y Reanimación, Complejo Hospitalario Universitario Insular Materno Infantil de Gran Canaria, Spain.
Factor XI (FXI) deficiency is a rare bleeding disorder characterized by a quantitative or qualitative deficiency of FXI. The symptoms are highly variable, and the severity and site of bleeding is unpredictable and does not necessarily correlate with FXI levels. FXI deficiency is classified by phenotype: bleeding or non-bleeding, depending on the clinical manifestations.
View Article and Find Full Text PDFRare bleeding disorders (RBDs) represent 3 to 5% of congenital bleeding disorders and are primarily inherited in an autosomal recessive manner, with increased prevalence in consanguineous populations. Clinically, RBDs can be accompanied by mild to severe bleeding episodes, often assessed using bleeding assessment tools (BATs) such as the International Society on Thrombosis and Hemostasis (ISTH)-BAT. However, the correlation between bleeding severity and coagulation factor activity levels remains inconsistent.
View Article and Find Full Text PDFAnimals (Basel)
November 2024
Department of Physiological Sciences, Institute of Veterinary Medicine, Warsaw University of Life Sciences, 02-776 Warsaw, Poland.
Genetic disorders are caused by a hereditary change in the structure of DNA that may hurt the health and life of animals. Several recessive haplotypes and a few causative mutations are known in Holstein Friesian cattle: CDH (Holstein cholesterol deficiency), haplotypes with a homozygous deficiency in Holstein (HH1, HH3, HH4, HH5, HH6, HH7), BLAD (bovine leukocyte adhesion deficiency), DUMPS (deficiency of uridine monophosphate synthase), FXI (factor XI deficiency), HHM (mule foot, syndactyly), and BC (citrullinaemia). From a breeding point of view, these genetic diseases have highly negative effects and are a significant problem for breeders, exposing them to economic losses and hurting animal welfare.
View Article and Find Full Text PDFBr J Cardiol
May 2024
Professor of Cardiology Cardiovascular Research Unit, Clinical Research Facility, Northern General Hospital, Sheffield, S5 7AU.
Factor (F) XI or XIa inhibition has Fattracted interest due to the protection from thrombotic events and minimal bleeding tendency observed in FXI-deficient individuals. The prospect of uncoupling the management of thrombosis from the bleeding risk inadvertently associated with current therapy inspired the development of agents directed towards this step in the coagulation process. This review describes the physiological rationale behind FXI/FXIa inhibition and the pharmacological properties of existing FXI/FXIa inhibitors.
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