Compliance with periodic surveillance for Von-Hippel-Lindau disease.

Abstract

METHODS

In this nationwide study, Von Hippel-Lindau disease mutation carriers and those at 50% risk were invited to complete a questionnaire assessing (compliance with) advice given for periodic surveillance. Medical record data on compliance with recommended radiologic surveillance examinations were also collected.

CONCLUSIONS

The majority of those with or at risk of Von Hippel-Lindau disease reported having received and being fully compliant with screening advice. However, in many cases, the advice given was only partially consistent with published guidelines, and screening delays were observed. Efforts should be undertaken to stimulate guideline-based surveillance advice and to minimize screening delay.

RESULTS

Of the 84 (77%) participants, 78 indicated having received advice to undergo periodic surveillance. Of these, 71 reported being fully compliant with that advice. In 64% of the cases, this advice was only partially consistent with published guidelines. Based on medical record data, between one quarter and one third of individuals did not undergo surveillance as recommended in the guidelines for central nervous system lesions and one half for visceral lesions. Screening delay for central nervous system lesions was significantly higher in one hospital and in those cases where "the advice given" deviated from the guidelines.

PURPOSE

To assess compliance with a periodic surveillance regimen for Von Hippel-Lindau disease.

More about this publication

Genetics in medicine : official journal of the American College of Medical Genetics
  • Volume 13
  • Issue nr. 6
  • Pages 519-27
  • Publication date 01-06-2011

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