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Original article
Digital assessment of the fetal alcohol syndrome facial phenotype: reliability and agreement study
  1. Tracey W Tsang1,2,
  2. Zoe Laing-Aiken1,
  3. Jane Latimer2,
  4. James Fitzpatrick1,2,
  5. June Oscar3,
  6. Maureen Carter4,
  7. Elizabeth J Elliott1,2,5
  1. 1 The University of Sydney, Discipline of Child and Adolescent Health, Sydney Medical School, The Children’s Hospital at Westmead, Westmead, New South Wales, Australia
  2. 2 The George Institute for Global Health, Sydney Medical School, The University of Sydney, Sydney, New South Wales, Australia
  3. 3 Marninwarntikura Women’s Resource Centre, Fitzroy Crossing, Western Australia, Australia
  4. 4 Nindilingarri Cultural Health Services, Fitzroy Crossing, Western Australia, Australia
  5. 5 Clinical School at The Children's Hospital at Westmead, The Sydney Children’s Hospital Networks (Westmead), Westmead, New South Wales, Australia
  1. Correspondence to Dr Tracey W Tsang; tracey.tsang{at}sydney.edu.au

Abstract

Purpose To examine the three facial features of fetal alcohol syndrome (FAS) in a cohort of Australian Aboriginal children from two-dimensional digital facial photographs to: (1) assess intrarater and inter-rater reliability; (2) identify the racial norms with the best fit for this population; and (3) assess agreement with clinician direct measures.

Methods Photographs and clinical data for 106 Aboriginal children (aged 7.4–9.6 years) were sourced from the Lililwan Project. Fifty-eight per cent had a confirmed prenatal alcohol exposure and 13 (12%) met the Canadian 2005 criteria for FAS/partial FAS. Photographs were analysed using the FAS Facial Photographic Analysis Software to generate the mean PFL three-point ABC-Score, five-point lip and philtrum ranks and four-point face rank in accordance with the 4-Digit Diagnostic Code. Intrarater and inter-rater reliability of digital ratings was examined in two assessors. Caucasian or African American racial norms for PFL and lip thickness were assessed for best fit; and agreement between digital and direct measurement methods was assessed.

Results Reliability of digital measures was substantial within (kappa: 0.70–1.00) and between assessors (kappa: 0.64–0.89). Clinician and digital ratings showed moderate agreement (kappa: 0.47–0.58). Caucasian PFL norms and the African American Lip-Philtrum Guide 2 provided the best fit for this cohort.

Conclusion In an Aboriginal cohort with a high rate of FAS, assessment of facial dysmorphology using digital methods showed substantial inter- and intrarater reliability. Digital measurement of features has high reliability and until data are available from a larger population of Aboriginal children, the African American Lip-Philtrum Guide 2 and Caucasian (Strömland) PFL norms provide the best fit for Australian Aboriginal children.

  • child
  • fetal alcohol spectrum disorders
  • face
  • dysmorphology
  • measurement

This is an Open Access article distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/

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Footnotes

  • Contributors TWT conceptualised and designed the study and analyses reported, was an assessor of the photographs (A2), managed and analysed the data, drafted and critically revised the manuscript and approved the final manuscript as submitted. ZL-A was an assessor of the photographs (A1), assisted with the first draft of the manuscript, data entry and initial analyses and approved the final manuscript. JL was involved in the conceptualisation and design of the Lililwan Project as a Chief Investigator from which the data were collected, provided critical revisions to the manuscript and approved the final manuscript as submitted. JF was involved in the conceptualisation and design of the Lililwan Project as a Chief Investigator from which the data were collected, took some of the facial photographs used in this study and approved the final manuscript as submitted. JO was involved in the conceptualisation and design of the Lililwan Project as a Chief Investigator from which the data were collected, gave input on cultural adaptations and approved the final manuscript as submitted. MC was involved in the conceptualisation and design of the Lililwan Project as a Chief Investigator from which the data were collected, consulted on cultural adaptations and approved the final manuscript as submitted. EJE was involved in the conceptualisation and design of the Lililwan Project as a Chief Investigator from which the data were collected, and was involved in the conceptualisation of the study reported and taking of some of the photographs used in this study. She provided critical revisions and approved the final manuscript as submitted.

  • Funding TWT was funded by a National Health and Medical Research Council (NHMRC) Project Grant (#: 1024474), while EJE was funded by NHMRC Practitioner Fellowships (#: 457084 and 1021480). ZL was supported by a Summer Research Scholarship funded by The University of Sydney (Sydney Medical School), the NHMRC-funded Lililwan Project, and the Australian Paediatric Surveillance Unit. JL was supported byan Australian Research Council Future Fellowship (#: 0130007). JF was supported by a McCusker Clinical Research Fellowship. The Lililwan Project (from which the photos analysed were sourced) was funded by the NHMRC, the Department of Families, Housing, Community Services & Indigenous Affairs, the Department of Health and Ageing and The University of Sydney (Sydney Medical School). The authors have no financial relationships relevant to this article to disclose.

  • Competing interests None declared.

  • Ethics approval The University of Sydney, Western Australian Aboriginal Health Information Ethics Committee, Western Australian Country Health Services Board, Kimberley Aboriginal Health Planning Forum.

  • Provenance and peer review Not commissioned; externally peer reviewed.

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