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Prevalence of gastrointestinal stromal tumour (GIST) in the United Kingdom at different therapeutic lines: an epidemiologic model.

Abstract：

BACKGROUND:The prevalence of patients with gastrointestinal stromal tumourgst (GIST) who fail currently available treatments imatinib and sunitinib (third-line treatment-eligible GIST) is unknown, but is expected to be below an ultra-orphan disease threshold of 2/100,000 population used in England and Wales. Our study was designed to estimate the prevalence and absolute number of UK patients with unresectable/metastatic GIST at first-, second- and eventually third-line treatment. METHODS:Our open population model estimates the probability that the prevalence of UK third-line treatment-eligible GIST patients will remain under the ultra-orphan disease threshold. Model parameters for incidence, proportion of unresectable/metastatic disease and survival estimates for GIST patients were obtained from a targeted literature review and a UK cancer register. The robustness of the results was checked through differing scenarios taking extreme values of the input parameters. RESULTS:The base-case scenario estimated a prevalence of third-line treatment-eligible GIST of 1/100,000 and a prevalence count of 598 with a 99.9% likelihood of being below the ultra-orphan disease threshold. The extreme scenarios, one-way and probabilistic sensitivity analyses and threshold analysis confirmed the robustness of these results. CONCLUSIONS:The prevalence of third-line treatment-eligible GIST is very low and highly likely below the ultra-orphan disease threshold.

journal_name

BMC Cancer

journal_title

BMC cancer

authors

Starczewska Amelio JM,Cid Ruzafa J,Desai K,Tzivelekis S,Muston D,Khalid JM,Ashman P,Maguire A

doi

10.1186/1471-2407-14-364

subject

Has Abstract

pub_date

2014-05-24 00:00:00

pages

364

issn

1471-2407

pii

1471-2407-14-364

journal_volume

14

pub_type

杂志文章,评审

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