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A family longevity selection score: ranking sibships by their longevity, size, and availability for study.

ABSTRACT: Family studies of exceptional longevity can potentially identify genetic and other factors contributing to long life and healthy aging. Although such studies seek families that are exceptionally long lived, they also need living members who can provide DNA and phenotype information. On the basis of these considerations, the authors developed a metric to rank families for selection into a family study of longevity. Their measure, the family longevity selection score (FLoSS), is the sum of 2 components: 1) an estimated family longevity score built from birth-, gender-, and nation-specific cohort survival probabilities and 2) a bonus for older living siblings. The authors examined properties of FLoSS-based family rankings by using data from 3 ongoing studies: the New England Centenarian Study, the Framingham Heart Study, and screenees for the Long Life Family Study. FLoSS-based selection yields families with exceptional longevity, satisfactory sibship sizes and numbers of living siblings, and high ages. Parameters in the FLoSS formula can be tailored for studies of specific populations or age ranges or with different conditions. The first component of the FLoSS also provides a conceptually sound survival measure to characterize exceptional longevity in individuals or families in various types of studies and correlates well with later-observed longevity.

SUBMITTER: Sebastiani P

PROVIDER: S-EPMC2800272 | biostudies-other | 2009 Dec

REPOSITORIES: biostudies-other

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A family longevity selection score: ranking sibships by their longevity, size, and availability for study.

Sebastiani Paola P Hadley Evan C EC Province Michael M Christensen Kaare K Rossi Winifred W Perls Thomas T TT Ash Arlene S AS

American journal of epidemiology 20091112 12

Family studies of exceptional longevity can potentially identify genetic and other factors contributing to long life and healthy aging. Although such studies seek families that are exceptionally long lived, they also need living members who can provide DNA and phenotype information. On the basis of these considerations, the authors developed a metric to rank families for selection into a family study of longevity. Their measure, the family longevity selection score (FLoSS), is the sum of 2 compo ...[more]

PMID: 19910380

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Project description:BackgroundAlthough human longevity tends to cluster within families, genetic studies on longevity have had limited success in identifying longevity loci. One of the main causes of this limited success is the selection of participants. Studies generally include sporadically long-lived individuals, i.e. individuals with the longevity phenotype but without a genetic predisposition for longevity. The inclusion of these individuals causes phenotype heterogeneity which results in power reduction and bias. A way to avoid sporadically long-lived individuals and reduce sample heterogeneity is to include family history of longevity as selection criterion using a longevity family score. A main challenge when developing family scores are the large differences in family size, because of real differences in sibship sizes or because of missing data.MethodsWe discussed the statistical properties of two existing longevity family scores: the Family Longevity Selection Score (FLoSS) and the Longevity Relatives Count (LRC) score and we evaluated their performance dealing with differential family size. We proposed a new longevity family score, the mLRC score, an extension of the LRC based on random effects modeling, which is robust for family size and missing values. The performance of the new mLRC as selection tool was evaluated in an intensive simulation study and illustrated in a large real dataset, the Historical Sample of the Netherlands (HSN).ResultsEmpirical scores such as the FLOSS and LRC cannot properly deal with differential family size and missing data. Our simulation study showed that mLRC is not affected by family size and provides more accurate selections of long-lived families. The analysis of 1105 sibships of the Historical Sample of the Netherlands showed that the selection of long-lived individuals based on the mLRC score predicts excess survival in the validation set better than the selection based on the LRC score .ConclusionsModel-based score systems such as the mLRC score help to reduce heterogeneity in the selection of long-lived families. The power of future studies into the genetics of longevity can likely be improved and their bias reduced, by selecting long-lived cases using the mLRC.

Project description:Introduction/Context: The term core value (CV) can be defined as fundamental beliefs or principles, guiding one's behavior in a social context. Though core competencies of family medicine (FM) have been clearly defined by WONCA, there has been an ongoing debate on what the CVs are for family doctors (FDs). Ukraine is a developing country in the middle of Europe with a population of 43 million inhabitants, gained independence from the Soviet Union in 1991. Ukraine is a low-income country, developing a modern European healthcare system, especially regarding FM. To implement WONCA standards, it is mandatory to assess the ongoing understanding of CVs in clinical daily practice among active FDs, working in different countries of Europe including Ukraine. Research questions: How do Ukrainian FDs (Delphi group experts) define the CVs of FM in Ukraine and how important are these CVs to a wider population of Ukrainian FDs in their everyday practice? Methods: A mixed method study was conducted in two steps during August and September 2020 in Ukraine. The first part was a qualitative Delphi round (three rounds) design among 20 Ukrainian FDs who were familiar with teaching and terms like CV. A consensus list of six CVs has emerged from the Delphi round study. The second part was a quantitative survey among Ukrainian FDs, who were not specially used to discussing CVs. The consensus list of those six CVs was then submitted to 2000 FDs (randomly selected) who were not involved in the Delphi team, to rank those values from one to nine, according to the importance from their personal point of view. Demographic characteristics have been assessed for all the participants of the Delphi round and quantitative survey. Results: Twenty FDs were involved as experts in the first Delphi round, whereas only five experts continued their participation in the second and the third rounds of the survey. The following six CVs emerged from the Delphi round: comprehensive approach, care coordination, first recourse, continuity of care, integrated approach, and patient and family centered care. The final sample consisted of 375 FDs (19% response rate). There were 323 (88.7%) female and 34 (9.3%) male FDs in the sample. The mean age of the participants was 44.6±13.5 years. Discussion/Conclusion: Defining CVs for FM by Ukrainian FDs in a given socio-economical and historical-cultural setting is crucial to optimize primary medical care and to guarantee an appropriate and successful implementation of WONCA standards as well as CVs in different countries including those where reformation of the health system is ongoing.

Dataset Information

A family longevity selection score: ranking sibships by their longevity, size, and availability for study.

Publications

A family longevity selection score: ranking sibships by their longevity, size, and availability for study.

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