Unusual Causes of β Thalassemia Trait: Discovery of another Three Novel SUPT5H Variants

  • Nik Fatma Fairuz Nik Mohd Hasan
  • , Ahlem Achour
  • , Tamara Koopmann
  • , Adriaan van Gammeren
  • , Joep van der Leeuw
  • , Huib Ceelie
  • , Daniel Stieber
  • , Frank Baas
  • , Cornelis L. Harteveld*
  • *Corresponding author for this work

Research output: Contribution to journalArticleResearchpeer-review

2 Citations (Scopus)

Abstract

Beta (β) thalassemia is an inherited disorder that occurs following mutations or deletions in the β globin gene. Rarely, it is caused by variants in genes coding for erythroid transcriptional factors or trans-acting factors. Here, we report three novel variants of SUPT5H revealed by next generation sequencing. This, gene has been progressively acknowledged as a mimicker of β thalassemia trait in two independent individuals and one family. These individuals have the same features, including hypochromic microcytic indices, increased Hb A2 levels, without mutations in the β globin gene. The three novel SUPT5H variants identified in this study (c.1168_1169del, c.2688del and c.307+1G>A) are frameshift variants leading to a premature stop codon or an intronic variant predicted to alter the splice site consensus sequence by in silico software. All three variants are characterized as Loss-of-Function variants either by generating a truncated protein or haplo-insufficiency due to nonsense-mediated decay. These findings confirm the general observation that most variants in SUPT5H associated with a β thalassemia trait phenotype are Loss-of-Function variants. This gene should be considered as a potential target gene in the genetic diagnosis of any unsolved cases of increased HbA2 and unexplained inconsistency of phenotype and genotype of β thalassemia intermedia.

Original languageEnglish
Pages (from-to)145-148
Number of pages4
JournalHemoglobin
Volume49
Issue number2
DOIs
Publication statusPublished - 30 Mar 2025
Externally publishedYes

Keywords

  • Increased Hb A
  • SUPT5H
  • mutation
  • β thalassemia

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