Phenotypic and Genetic Characterization of a Cohort of Pediatric Wilson Disease Patients
2011

Study of Pediatric Wilson Disease in Egypt

Sample size: 77 publication 10 minutes Evidence: moderate

Author Information

Author(s): Abdel Ghaffar Tawhid, Elsayed Solaf M, Elnaghy Suzan, Shadeed Ahmed, Elsobky Ezzat S, Schmidt Hartmut

Primary Institution: Yassin Abdel Ghaffar Charity Center for Liver Disease and Research, Cairo, Egypt

Hypothesis

The study aims to characterize the clinical, laboratory, and genetic features of Wilson disease in a pediatric population in Egypt.

Conclusion

Egyptian children with Wilson disease present with early Kayser-Fleischer rings and early onset of liver and neurological disease.

Supporting Evidence

  • Patients had a younger age of onset (median: 10 years) compared to other populations.
  • 97.6% of symptomatic patients had Kayser-Fleischer rings.
  • Low ceruloplasmin levels were found in 93.5% of patients.
  • Parental consanguinity was high at 78.9%, indicating a genetic component.
  • 71.42% of patients on long-term D-penicillamine improved or were stable during follow-up.

Takeaway

This study looked at children in Egypt with Wilson disease, which affects how their bodies handle copper, and found they often show symptoms earlier than kids in other countries.

Methodology

The study included 77 patients from 50 families, with data collected retrospectively through record analysis and patient interviews, and diagnosis confirmed by ATP7B gene sequencing.

Potential Biases

Potential bias due to the high rate of consanguinity in the population, which may affect the generalizability of the findings.

Limitations

The study is limited by the retrospective design and the challenges in obtaining accurate 24-hour urine collections for copper excretion tests.

Participant Demographics

The cohort included 75 children and 2 adult cousins, with 43 males and 34 females, and a high rate of parental consanguinity (78.9%).

Statistical Information

P-Value

0.017

Statistical Significance

p<0.05

Digital Object Identifier (DOI)

10.1186/1471-2431-11-56

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