| Literature DB >> 35467101 |
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.Entities:
Keywords: Anemia; Genetics; Hemoglobinopathy; Molecular diagnosis; Thalassemia
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Year: 2022 PMID: 35467101 DOI: 10.1007/s00277-022-04837-4
Source DB: PubMed Journal: Ann Hematol ISSN: 0939-5555 Impact factor: 3.673