AI Article Synopsis

  • Rothmund-Thomson syndrome (RTS) is a rare genetic disorder with symptoms like a progressive rash, sparse hair, short stature, and an increased cancer risk, particularly for osteosarcoma and skin cancer.
  • It is caused by mutations in the ANAPC1 or RECQL4 genes, with two types identified based on the gene affected.
  • A case study is presented of an African girl with Type 2 RTS who had unique symptoms of precocious puberty and anal stenosis, emphasizing the importance of identifying deep intronic variants when typical mutations are absent.

Article Abstract

Rothmund-Thomson syndrome (RTS) is a rare autosomal recessive disorder characterized by a rash that progresses to poikiloderma. Other common features include sparse hair, eyelashes and eyebrows, short stature, variable skeletal abnormalities, dental defects, cataracts, hypogonadism, and an increased risk for cancer, especially osteosarcoma and skin cancer. RTS is caused by biallelic pathogenic variants in ANAPC1 (Type 1 RTS) or RECQL4 (Type 2 RTS). We present an African girl with Type 2 RTS caused by a nonsense variant and an intronic variant in RECQL4. The patient presented precocious puberty, which has not been previously reported in RTS and that was treated with a GnRH analog, and anal stenosis, which has only been reported once. This case highlights the need to consider deep intronic variants in patients with RTS when pathogenic variants in the coding regions and exon/intron boundaries are not identified and expands the phenotypic spectrum of this disorder.

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http://dx.doi.org/10.1002/ajmg.a.62980DOI Listing

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