EHA Library - The official digital education library of European Hematology Association (EHA)

NEW LIKELY PATHOGENIC VARIANTS IN THREE SPANISH PATIENTS WITH MILD HEMOPHILIA A AND B: GENETIC COUNSELING.
Author(s): ,
Maria Civeira Marín
Affiliations:
hematology,Hospital Universitario Miguel Servet,zaragoza,Spain
,
Raquel Lahoz Alonso
Affiliations:
genetics,Hospital Universitario Miguel Servet,zaragoza,Spain
,
Amaia López Peña
Affiliations:
hematology,Hospital Universitario Miguel Servet,zaragoza,Spain
,
Carmen Rodríguez Lefler
Affiliations:
hematology,Hospital Universitario Miguel Servet,zaragoza,Spain
,
Carlos francisco Hernández Mata
Affiliations:
hematology,Hospital Universitario Miguel Servet,zaragoza,Spain
,
Marta Moreno Carbonell
Affiliations:
hematology,Hospital Universitario Miguel Servet,zaragoza,Spain
,
Eduardo Gonzalez
Affiliations:
hematology,Hospital Universitario Miguel Servet,zaragoza,Spain
,
Ana Gomez Martínez
Affiliations:
hematology,Hospital Universitario Miguel Servet,zaragoza,Spain
,
Sofia Martin-consuegra Ramos
Affiliations:
hematology,Hospital Universitario Miguel Servet,zaragoza,Spain
,
Ricardo González Tarancón
Affiliations:
genetics,Hospital Universitario Miguel Servet,zaragoza,Spain
,
Silvia Izquierdo Álvarez
Affiliations:
genetics,Hospital Universitario Miguel Servet,zaragoza,Spain
,
Nuria Fernández Mosteirín
Affiliations:
hematology,Hospital Universitario Miguel Servet,zaragoza,Spain
Jose Manuel Calvo Villas
Affiliations:
hematology,Hospital Universitario Miguel Servet,zaragoza,Spain
EHA Library. Civeira M. 06/09/21; 325338; EP578
Maria Civeira
Maria Civeira
Contributions
Abstract
Presentation during EHA2021: All e-poster presentations will be made available as of Friday, June 11, 2021 (09:00 CEST) and will be accessible for on-demand viewing until August 15, 2021 on the Virtual Congress platform.

Abstract: EP578

Type: E-Poster Presentation

Session title: Bleeding disorders (congenital and acquired)

Background

Mild hemophilia A (HA) and B (HB) are X-linked recessive hereditary diseases, characterized by factor VIII and IX deficiency, respectively. Patients with mild or moderate hemophilia mostly present specific mutations in F8 gene (Xq28) or F9 (Xq22), detectable by direct sequencing. More than 4000 mutations have been detected so far and genotype analysis should be offered in selected patients. Therefore, genetic assessment is important to provide reproductive options.

Aims

We report three new likely pathogenic variants in three male Spanish patients. Patient 1 presented asymptomatic prolonged pre-operatory aPTT and the others were studied due to hemorrhagic complications of surgery. All presented isolated reduced factor VIII or IX activity levels.

Methods

Molecular genetic studies: DNA was extracted from peripheral blood. Coding exons and adjacent intronic regions of F8 and F9 gene were amplified by polymerase chain reaction (PCR) and sequenced on Applied Biosystems® 3500DX Genetic Analyzer. The results were checked in HGMD, ClinVar, dbSNP, gnomAD, and 1000G international databases and different in silico prediction algorithms, such as: PolyPhen-2, SIFT, Mutation Taster, Align GVGD, REVEL and others were consulted.

Results

Three new missense variants were found (table 1). These variants have not been previously reported in the literature or in any of the databases consulted. They do affect the physical-chemical characteristics of the protein (in silico prediction). Therefore, they are classified as likely pathogenic. Because of this, we extended the study to first-degree relatives; finding carriers that were not diagnose at that time. A 20-year-old daughter (patient 1) and a sister (patient 2) received genetic counseling.

Conclusion

These new genetic variants confirm the clinical suspicion of mild hemophilia. The molecular genetic analysis in the family members helps finding asymptomatic carriers so we can provide them genetic counseling. Future reproductive options will be considered in the youngest patient and any family members if needed, firstly and foremostly, prenatal fetal sex chromosome testing by maternal plasma DNA sequencing on female carriers.

Keyword(s): Diagnosis, Gene expression, Hemophilia A, Hemophilia B

Presentation during EHA2021: All e-poster presentations will be made available as of Friday, June 11, 2021 (09:00 CEST) and will be accessible for on-demand viewing until August 15, 2021 on the Virtual Congress platform.

Abstract: EP578

Type: E-Poster Presentation

Session title: Bleeding disorders (congenital and acquired)

Background

Mild hemophilia A (HA) and B (HB) are X-linked recessive hereditary diseases, characterized by factor VIII and IX deficiency, respectively. Patients with mild or moderate hemophilia mostly present specific mutations in F8 gene (Xq28) or F9 (Xq22), detectable by direct sequencing. More than 4000 mutations have been detected so far and genotype analysis should be offered in selected patients. Therefore, genetic assessment is important to provide reproductive options.

Aims

We report three new likely pathogenic variants in three male Spanish patients. Patient 1 presented asymptomatic prolonged pre-operatory aPTT and the others were studied due to hemorrhagic complications of surgery. All presented isolated reduced factor VIII or IX activity levels.

Methods

Molecular genetic studies: DNA was extracted from peripheral blood. Coding exons and adjacent intronic regions of F8 and F9 gene were amplified by polymerase chain reaction (PCR) and sequenced on Applied Biosystems® 3500DX Genetic Analyzer. The results were checked in HGMD, ClinVar, dbSNP, gnomAD, and 1000G international databases and different in silico prediction algorithms, such as: PolyPhen-2, SIFT, Mutation Taster, Align GVGD, REVEL and others were consulted.

Results

Three new missense variants were found (table 1). These variants have not been previously reported in the literature or in any of the databases consulted. They do affect the physical-chemical characteristics of the protein (in silico prediction). Therefore, they are classified as likely pathogenic. Because of this, we extended the study to first-degree relatives; finding carriers that were not diagnose at that time. A 20-year-old daughter (patient 1) and a sister (patient 2) received genetic counseling.

Conclusion

These new genetic variants confirm the clinical suspicion of mild hemophilia. The molecular genetic analysis in the family members helps finding asymptomatic carriers so we can provide them genetic counseling. Future reproductive options will be considered in the youngest patient and any family members if needed, firstly and foremostly, prenatal fetal sex chromosome testing by maternal plasma DNA sequencing on female carriers.

Keyword(s): Diagnosis, Gene expression, Hemophilia A, Hemophilia B

By clicking “Accept Terms & all Cookies” or by continuing to browse, you agree to the storing of third-party cookies on your device to enhance your user experience and agree to the user terms and conditions of this learning management system (LMS).

Cookie Settings
Accept Terms & all Cookies