bims-conane Biomed News
on Congenital anemias
Issue of 2025–10–12
five papers selected by
João Conrado Khouri dos Santos, Universidade de São Paulo



  1. Hemoglobin. 2025 Oct 05. 1-4
      Dominant β-thalassemia is a rare form of thalassemia that is caused by a heterogenous group of molecular defects, including missense, nonsense, and frameshift mutations. Among the missense mutations involving the third exon of β-globin gene is the rare Hb Dieppe (HBB:c. 383A > G) which leads to a very unstable β-variant. In the current study we report this variant in an 8-year-old girl and her 35-year-old mother in an Iraqi Kurdish family, both presenting as β-thalassemia intermedia with moderate hypochromic anemia, increased hemoglobin F and borderline hemoglobin A2. This constitutes the first report of this variant from an Eastern Mediterranean country, and underscores the pivotal role of molecular studies to diagnose Hb Dieppe and other dominant β-thalassemias, since in most cases the resultant variants are undetectable by hemoglobin electrophoresis.
    Keywords:  Dominant β-thalassemia; HBB:c. 383A > G; Hb Dieppe; Iraq
    DOI:  https://doi.org/10.1080/03630269.2025.2567265
  2. Front Pediatr. 2025 ;13 1666585
       Background: Hereditary spherocytosis (HS) is a genetically inherited hemolytic anemia resulting from erythrocyte membrane defects, predominantly associated with genetic mutations in membrane protein genes such as ANK1 and SPTB. The disease exhibits considerable heterogeneity in both clinical manifestations and age of onset, presenting substantial diagnostic challenges for clinicians, particularly in pediatric cases.
    Case presentation: The patient was a 29-day-old boy who had experienced persistent anemia and a medical history of neonatal hyperbilirubinemia since birth. Upon admission, the infant lacked typical HS manifestations such as splenomegaly, jaundice, and spherocytosis on the peripheral blood smear. Whole-exome sequencing identified a novel frameshift mutation c.3556delG (EX30, NM_000037.4), resulting in an amino acid alteration p.Glu1186Lysfs*3. Subsequent Sanger sequencing-based family segregation analysis confirmed that this mutation originated from the paternal allele. Based on the characteristic clinical manifestations and genetic findings, a definitive diagnosis of HS was established.
    Conclusions: In neonates presenting with unexplained recurrent anemia, particularly those with a history of neonatal hyperbilirubinemia, HS should be suspected. Due to the atypical manifestations, genetic analysis serves as a pivotal tool in the early diagnosis of HS, and novel genetic mutations may be identified, which can subsequently be added to the genetic database.
    Keywords:  ANK1 gene; WES—whole-exome sequencing; frameshift mutation; hereditary spherocytosis; neonates
    DOI:  https://doi.org/10.3389/fped.2025.1666585
  3. Pak J Med Sci. 2025 Sep;41(9): 2600-2605
       Background & Objective: Thalassemia is a genetic blood disorder primarily influenced by β-globin gene mutations and various genetic modifiers. In Pakistan, especially among transfusion-dependent thalassemia (TDT) patients, data on these modifying factors remain limited, hindering accurate diagnosis and tailored management. This study aimed to reassess the diagnostic classification and determine the frequency of genetic determinants specifically alpha-thalassemia deletions and key polymorphisms (BCL11A and Xmn1-HBG2) associated with milder clinical phenotypes in multi-transfused thalassemia patients from the local population.
    Methodology: This descriptive cross-sectional study was conducted over a six-month period, from January to June 2019 and included 54 TDT patients aged up to 15 years from the Fatimid Foundation, Peshawar. Genetic analyses were performed for polymorphisms at Xmn-1-HBG2 and BCL11A, as well as for two alpha (α) and thirteen common beta (β) gene alterations. Utilizing the PureLinkTM Genomic DNA Kit, DNA was retrieved. The ARMS technique was used to identify β-thalassemia mutations, whereas Gap-PCR was used to identify α-thalassemia deletions. For Xmn-1 genotyping, we used RFLP-PCR, while ARMS-PCR was used to assess BCL11A polymorphisms.
    Results: All patients had either homozygous or compound heterozygous mutations in the β-globin gene. Of them, 38 patients had BCL11A polymorphisms, two had Xmn-1-HBG2 polymorphisms and 11 patients had a co-existing heterozygous α (3.7 kb) deletion. Homozygous Fr 8-9 was the variant with the most common mutation, occurring in 19 (35.2%) cases. Isolated β-globin gene mutations were found in just 13 cases. Together with the underlying β-globin mutation, 85.2% patients had an additional ameliorating genetic component (a BCL11A polymorphism, Xmn-1-HBG2 polymorphism, or α-globin gene mutation).
    Conclusion: Transfusion-dependent β-thalassemia patients in Peshawar Region often have co-existing genetic modifiers, resulting in a milder phenotype. Screening for these modifiers is recommended for specialized treatment in these children.
    Keywords:  Ameliorating factors; BCL11A; Genetic modifiers: polymorphism; Transfusion dependent thalassemia; Xmn-1; α mutations; β mutations
    DOI:  https://doi.org/10.12669/pjms.41.9.11571