Mosaicism for r(X) and der(X)del(X)(p11.23)dup(X)(p11.21p11.22) provides insight into the possible mechanism of rearrangement
2008

Mosaicism for r(X) and der(X) provides insight into chromosome rearrangement

Sample size: 1 publication Evidence: moderate

Author Information

Author(s): Oleg A. Shchelochkov, M. Lance Cooper, Zhishuo Ou, Sandra Peacock, Svetlana A. Yatsenko, Chester W. Brown, Ping Fang, Pawel Stankiewicz, Sau Wai Cheung

Primary Institution: Baylor College of Medicine

Hypothesis

We hypothesize that a prezygotic asymmetric crossing over within a loop formed during meiosis in an X chromosome with a paracentric inversion resulted in an intermediate dicentric chromosome.

Conclusion

The study reveals a unique cytogenetic abnormality involving mosaicism for r(X) and a deleted Xp derivative chromosome, providing insights into the mechanisms of chromosome rearrangement.

Supporting Evidence

  • The patient presented with failure to thrive, muscular hypotonia, and minor facial anomalies.
  • Brain MRI revealed mild thinning of the corpus callosum and an asymmetric myelination pattern.
  • Array comparative genome hybridization analysis revealed mosaicism for the X chromosome and a deletion of the short arm of an X chromosome.

Takeaway

This study looks at a girl with a special type of chromosome problem that caused her to have health issues, helping us understand how these problems can happen.

Methodology

The study used array comparative genome hybridization, G-banded chromosome analysis, and X chromosome inactivation studies to analyze the patient's chromosomes.

Limitations

Parental chromosomes were not available for further analysis to confirm the proposed mechanisms.

Participant Demographics

The participant was a 10-month-old girl with clinical features of Turner syndrome.

Digital Object Identifier (DOI)

10.1186/1755-8166-1-16

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