Literature DB >> 23616389

Interrater reliability of a phenotypic assessment tool for the ear morphology in microtia.

Daniela V Luquetti1, Babette S Saltzman, Kathleen C Sie, Craig B Birgfeld, Brian G Leroux, Kelly N Evans, James M Smartt, David D Tieu, Daniel J Dudley, Carrie L Heike.   

Abstract

The Elements of Morphology Standard Terminology working group published standardized definitions for external ear morphology. The primary objective of our study was to use these descriptions to evaluate the interrater reliability for specific features associated with microtia. We invited six raters from three different subspecialities to rate 100 ear photographs on 32 features. We calculated overall and within specialty and professional experience intraclass correlation coefficients (ICC) and 95% confidence intervals. A total of 600 possible observations were recorded for each feature. The overall interrater reliability ranged from 0.04 (95% CI: 0.00-0.14) for the width of the antihelix inferior crus to 0.93 (95% CI: 0.91-0.95) for the presence of the inferior crux of the antihelix. The reliability for quantitative characteristics such as length or width of an ear structure was generally lower than the reliability for qualitative characteristics (e.g., presence or absence of an ear structure). Categories with very poor interrater reliability included anti-helix inferior crux width (0.04, 95% CI: 0.00-0.14), crux helix extension (0.17, 95% CI 0.00-0.37), and shape of the incisura (0.14, 95% CI: 0.01-0.27). There were no significant differences in reliability estimates by specialty or professional experience for most variables. Our study showed that it is feasible to systematically characterize many of structures of the ear that are affected in microtia. We incorporated these descriptions into a standardized phenotypic assessment tool (PAT-Microtia) that might be used in multicenter research studies to identify sub-phenotypes for future studies of microtia.
Copyright © 2013 Wiley Periodicals, Inc.

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Year:  2013        PMID: 23616389      PMCID: PMC3664118          DOI: 10.1002/ajmg.a.35963

Source DB:  PubMed          Journal:  Am J Med Genet A        ISSN: 1552-4825            Impact factor:   2.802


  9 in total

1.  Classification of congenital deformities of the auricle.

Authors:  H Weerda
Journal:  Facial Plast Surg       Date:  1988-10       Impact factor: 1.446

Review 2.  Microtia.

Authors:  R C Tanzer
Journal:  Clin Plast Surg       Date:  1978-07       Impact factor: 2.017

3.  Fetal genetic risk of isolated cleft lip only versus isolated cleft lip and palate: a subphenotype analysis using two population-based studies of orofacial clefts in Scandinavia.

Authors:  Astanand Jugessur; Min Shi; Håkon Kristian Gjessing; Rolv Terje Lie; Allen James Wilcox; Clarice Ring Weinberg; Kaare Christensen; Abee Lowman Boyles; Sandra Daack-Hirsch; Truc Trung Nguyen; Lene Christiansen; Andrew Carl Lidral; Jeffrey Clark Murray
Journal:  Birth Defects Res A Clin Mol Teratol       Date:  2010-12-07

4.  Detailed assessment of the ear in Cornelia de Lange syndrome: comparison with a control sample using the new dysmorphology guidelines.

Authors:  Alasdair G W Hunter; Julianne S Collins; Matthew A Deardorff; Ian D Krantz
Journal:  Am J Med Genet A       Date:  2009-10       Impact factor: 2.802

5.  Elements of morphology: introduction.

Authors:  Judith E Allanson; Leslie G Biesecker; John C Carey; Raoul C M Hennekam
Journal:  Am J Med Genet A       Date:  2009-01       Impact factor: 2.802

6.  Elements of morphology: standard terminology for the ear.

Authors:  Alasdair Hunter; Jaime L Frias; Gabriele Gillessen-Kaesbach; Helen Hughes; Kenneth Lyons Jones; Louise Wilson
Journal:  Am J Med Genet A       Date:  2009-01       Impact factor: 2.802

7.  Photographic protocol for image acquisition in craniofacial microsomia.

Authors:  Carrie L Heike; Laura P Stueckle; Erik T Stuhaug; Luiz A Pimenta; Amelia F Drake; Daniela Vivaldi; Kathleen C Y Sie; Craig B Birgfeld
Journal:  Head Face Med       Date:  2011-12-30       Impact factor: 2.151

8.  Targeted capture and massively parallel sequencing of 12 human exomes.

Authors:  Sarah B Ng; Emily H Turner; Peggy D Robertson; Steven D Flygare; Abigail W Bigham; Choli Lee; Tristan Shaffer; Michelle Wong; Arindam Bhattacharjee; Evan E Eichler; Michael Bamshad; Deborah A Nickerson; Jay Shendure
Journal:  Nature       Date:  2009-08-16       Impact factor: 49.962

Review 9.  Statistics review 9: one-way analysis of variance.

Authors:  Viv Bewick; Liz Cheek; Jonathan Ball
Journal:  Crit Care       Date:  2004-03-01       Impact factor: 9.097

  9 in total
  2 in total

1.  Phenotypic sub-grouping in microtia using a statistical and a clinical approach.

Authors:  Daniela V Luquetti; Babette S Saltzman; Carrie L Heike; Kathleen C Sie; Craig B Birgfeld; Kelly N Evans; Brian G Leroux
Journal:  Am J Med Genet A       Date:  2015-02-05       Impact factor: 2.802

Review 2.  The genetics of auricular development and malformation: new findings in model systems driving future directions for microtia research.

Authors:  Timothy C Cox; Esra D Camci; Siddharth Vora; Daniela V Luquetti; Eric E Turner
Journal:  Eur J Med Genet       Date:  2014-05-29       Impact factor: 2.708

  2 in total

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