Literature DB >> 35467101

Does size matter? Two new deletions in the HBB gene cause β0-thalassemia.

Paloma Ropero1,2, Fernando Ataúlfo González Fernández3, Jorge M Nieto3,4, Valle Recasens5, Ángeles Montañés5, María José Murúzabal6, María Sarasa7, Cristina Fernández7, Ana Villegas3, Cuesta C Benavente3.   

Abstract

Most β-thalassemias are caused by mutations involving one or a limited number of nucleotides within the gene or its adjacent regions. They can be substitutions or deletions; in these cases, the loss ranges from a single nucleotide to even the entire HBB gene, so we wonder if the phenotype is due to the size of the deletion or the location of the mutation. To clarify this, we present two new deletions in the β-globin gene that cause β0-thalassemia. The hematological parameters were determined with an automated cell counter; the Hb A2 and Hb F levels were measured by performance liquid chromatography. Hemoglobins were analyzed by capillary zone electrophoresis (Sebia Capillarys Flex system) and ion-exchange HPLC (BioRad Variant II β-thalassemia Short Program). Molecular characterization was performed by automatic Sanger sequencing. The screening of common α-thalassemia point mutations and deletions in the world (21 in total) were carried out using multiplex PCR followed by reverse-hybridization with a commercial Alpha-Globin StripAssay kit. We have characterized two new mutations-(1) 1-bp deletion [CD61/62(-G)] [HBB:c.186_187delG], (2) 105-bp deletion [IVS-2-nt767-CD111] [HBB:c.316-84_333del]-and we have described, for first time in Spain, the 25-bp deletion [β nts 252 - 276 deleted] [HBB:c.93-22_95del] mutation. These mutations were classified as pathogenic by UniProt Variants confirmed according to the American College of Medical Genetics and Genomics guidelines. These mutations present a phenotype compatible with β0-thalassemia, supported by hematological parameters that correlate the degree of reduction in the synthesis of the β-globin chain. Identification of this type of mutation is important for genetic counselling of partners where both are carriers, so that they are aware of the genetic risk of having affected children, allowing them to take an informed decision about their reproductive choices.
© 2022. The Author(s), under exclusive licence to Springer-Verlag GmbH Germany, part of Springer Nature.

Entities:  

Keywords:  Anemia; Genetics; Hemoglobinopathy; Molecular diagnosis; Thalassemia

Mesh:

Substances:

Year:  2022        PMID: 35467101     DOI: 10.1007/s00277-022-04837-4

Source DB:  PubMed          Journal:  Ann Hematol        ISSN: 0939-5555            Impact factor:   3.673


  7 in total

Review 1.  β-Thalassemias.

Authors:  Ali T Taher; Khaled M Musallam; M Domenica Cappellini
Journal:  N Engl J Med       Date:  2021-02-25       Impact factor: 91.245

Review 2.  Molecular basis of β thalassemia and potential therapeutic targets.

Authors:  Swee Lay Thein
Journal:  Blood Cells Mol Dis       Date:  2017-06-20       Impact factor: 3.039

Review 3.  Genotype-phenotype correlations in beta-thalassemias.

Authors:  A Cao; R Galanello; M C Rosatelli
Journal:  Blood Rev       Date:  1994-03       Impact factor: 8.250

Review 4.  The molecular basis of β-thalassemia.

Authors:  Swee Lay Thein
Journal:  Cold Spring Harb Perspect Med       Date:  2013-05-01       Impact factor: 6.915

5.  Standards and guidelines for the interpretation of sequence variants: a joint consensus recommendation of the American College of Medical Genetics and Genomics and the Association for Molecular Pathology.

Authors:  Sue Richards; Nazneen Aziz; Sherri Bale; David Bick; Soma Das; Julie Gastier-Foster; Wayne W Grody; Madhuri Hegde; Elaine Lyon; Elaine Spector; Karl Voelkerding; Heidi L Rehm
Journal:  Genet Med       Date:  2015-03-05       Impact factor: 8.822

Review 6.  Beta-thalassemia.

Authors:  Antonio Cao; Renzo Galanello
Journal:  Genet Med       Date:  2010-02       Impact factor: 8.822

7.  Updates of the HbVar database of human hemoglobin variants and thalassemia mutations.

Authors:  Belinda Giardine; Joseph Borg; Emmanouil Viennas; Cristiana Pavlidis; Kamran Moradkhani; Philippe Joly; Marina Bartsakoulia; Cathy Riemer; Webb Miller; Giannis Tzimas; Henri Wajcman; Ross C Hardison; George P Patrinos
Journal:  Nucleic Acids Res       Date:  2013-10-16       Impact factor: 16.971

  7 in total

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