{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,2,27]],"date-time":"2026-02-27T04:27:59Z","timestamp":1772166479645,"version":"3.50.1"},"reference-count":12,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2022,1,15]],"date-time":"2022-01-15T00:00:00Z","timestamp":1642204800000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2022,1,15]],"date-time":"2022-01-15T00:00:00Z","timestamp":1642204800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["BMC Res Notes"],"published-print":{"date-parts":[[2022,12]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Objective<\/jats:title>\n                    <jats:p>Life expectancy can be estimated accurately from a cohort of individuals born in the same year and followed from birth to death. However, due to the resource-consuming nature of following a cohort prospectively, life expectancy is often assessed based upon retrospective death record reviews. This conventional approach may lead to potentially biased estimates, in particular when estimating life expectancy of rare diseases such as Morquio syndrome A. We investigated the accuracy of life expectancy estimation using death records by simulating the survival of individuals with Morquio syndrome A under four different scenarios.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>When life expectancy was constant during the entire period, using death data did not result in a biased estimate. However, when life expectancy increased over time, as is often expected to be the case in rare diseases, using only death data led to a substantial underestimation of life expectancy. We emphasize that it is therefore crucial to understand how estimates of life expectancy are obtained, to interpret them in an appropriate context, and to assess estimation methods within a sensitivity analysis framework, similar to the simulations performed herein.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1186\/s13104-021-05894-0","type":"journal-article","created":{"date-parts":[[2022,1,15]],"date-time":"2022-01-15T02:03:09Z","timestamp":1642212189000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Understanding bias when estimating life expectancy from age at death: a simulation approach applied to Morquio syndrome A"],"prefix":"10.1186","volume":"15","author":[{"given":"Xue","family":"Yin","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jaeil","family":"Ahn","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1400-3398","authenticated-orcid":false,"given":"Simina M.","family":"Boca","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"297","published-online":{"date-parts":[[2022,1,15]]},"reference":[{"key":"5894_CR1","doi-asserted-by":"publisher","first-page":"257","DOI":"10.1080\/00324720600895876","volume":"60","author":"JR Goldstein","year":"2006","unstructured":"Goldstein JR, Wachter KW. Relationships between period and cohort life expectancy: gaps and lags. Popul Stud. 2006;60:257\u201369.","journal-title":"Popul Stud"},{"key":"5894_CR2","volume-title":"The methods and materials of demography. 2nd ed","author":"H Shryock","year":"1973","unstructured":"Shryock H, Siegel J, Larmon E. The methods and materials of demography. 2nd ed. Suitland: U.S. Bureau of the Census; 1973."},{"key":"5894_CR3","doi-asserted-by":"publisher","first-page":"45","DOI":"10.5617\/njhe.1290","volume":"5","author":"FR Lichtenberg","year":"2017","unstructured":"Lichtenberg FR. The impact of biomedical innovation on longevity and health. Nord J Health Econ. 2017;5:45\u201357.","journal-title":"Nord J Health Econ"},{"key":"5894_CR4","doi-asserted-by":"publisher","first-page":"533","DOI":"10.1007\/978-90-481-9996-9_25","volume-title":"International handbook of adult mortality","author":"M Guillot","year":"2011","unstructured":"Guillot M. Period versus cohort life expectancy. In: Rogers R, Crimmins E, editors. International handbook of adult mortality. Dordrecht: Springer; 2011. p. 533\u201349."},{"key":"5894_CR5","doi-asserted-by":"publisher","DOI":"10.1186\/s13023-014-0173-x","author":"RM Leadley","year":"2014","unstructured":"Leadley RM, Lang S, Misso K, et al. A systematic review of the prevalence of Morquio A syndrome: challenges for study reporting in rare diseases. Orphanet J Rare Dis. 2014. https:\/\/doi.org\/10.1186\/s13023-014-0173-x.","journal-title":"Orphanet J Rare Dis"},{"key":"5894_CR6","doi-asserted-by":"publisher","DOI":"10.1186\/s13023-014-0192-7","author":"K Bhattacharya","year":"2014","unstructured":"Bhattacharya K, Balasubramaniam S, Choy Y, et al. Overcoming the barriers to diagnosis of Morquio A syndrome. 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