Literature DB >> 29948449

Low-level parental mosaicism in an apparent de novo case of Peutz-Jeghers syndrome.

G I Butel-Simoes1, A D Spigelman2,3, R J Scott4, R E Vilain5.   

Abstract

We report the case of a female found to have mosaicism for mutation in the STK11 gene, with the mutant allele expressed in her gametes, evident by her affected offspring, and in her gastrointestinal tract demonstrated on an excised polyp analysed for diagnosis. Mosaicism for Peutz-Jeghers syndrome (PJS) has been reported in a small number of cases previously but a clinical presentation such as this has not previously been described. This finding of mosaicism was several years after initial investigations failed to identify the same STK11 mutation in this woman whose son was diagnosed with PJS at a young age. This case highlights the importance of considering mosaicism as an explanation for apparent de novo cases of PJS syndrome. It also has implications for genetic counselling, predictive testing and cancer screening.

Entities:  

Keywords:  De novo; Gonadal; Mosaic; Peutz–Jeghers syndrome (PJS); STK11; Somatic

Mesh:

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Year:  2019        PMID: 29948449     DOI: 10.1007/s10689-018-0093-3

Source DB:  PubMed          Journal:  Fam Cancer        ISSN: 1389-9600            Impact factor:   2.375


  2 in total

1.  Two cases of somatic STK11 mosaicism in Danish patients with Peutz-Jeghers syndrome.

Authors:  Anne Marie Jelsig; Birgitte Bertelsen; Isabel Forss; John Gásdal Karstensen
Journal:  Fam Cancer       Date:  2020-06-06       Impact factor: 2.375

Review 2.  Rare Hereditary Gynecological Cancer Syndromes.

Authors:  Takafumi Watanabe; Shu Soeda; Yuta Endo; Chikako Okabe; Tetsu Sato; Norihito Kamo; Makiko Ueda; Manabu Kojima; Shigenori Furukawa; Hidekazu Nishigori; Toshifumi Takahashi; Keiya Fujimori
Journal:  Int J Mol Sci       Date:  2022-01-29       Impact factor: 5.923

  2 in total

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