TY - JOUR
T1 - A Photo Score for Aesthetic Outcome in Sagittal Synostosis
T2 - An ERN CRANIO Collaboration
AU - Gaillard, Linda
AU - Mathijssen, I. M.J.
AU - Versnel, S.
AU - Pleumeekers, M.
AU - Van Veelen, M. L.C.
AU - Delye, H.
AU - Haber, S.
AU - Evans, M.
AU - Parks, C.
AU - Kölby, L.
AU - Nowinski, D.
AU - Valentini, L.
AU - Tamburrini, G.
AU - Thomale, U. W.
AU - Schulz, M.
AU - Vuola, Pia
AU - Faria, C. C.
AU - Di Rocco, F.
AU - Vinchon, M.
AU - Mena-Bernal, J. Hinojosa
AU - Larysz, D.
AU - Larysz, P.
AU - Spacca, B.
AU - Due-Tønnessen, B. J.
AU - Munarriz, P. M.
AU - Rosas, K.
AU - Bermeo, D. F.L.
AU - Leikola, J.
N1 - Publisher Copyright:
© 2023 Lippincott Williams and Wilkins. All rights reserved.
PY - 2023/11/1
Y1 - 2023/11/1
N2 - European Reference Network (ERN) CRANIO is focused on optimizing care for patients with rare or complex craniofacial anomalies, including craniosynostosis and/or rare ear, nose, and throat disorders. The main goal of ERN CRANIO is to collect uniform data on treatment outcomes for multicenter comparison. We aimed to develop a reproducible and reliable suture-specific photo score that can be used for cross-center comparison of phenotypical severity of sagittal synostosis and aesthetic outcome of treatment. We conducted a retrospective study among nonsyndromic sagittal synostosis patients aged <19 years. We included preoperative and postoperative photo sets from 6 ERN CRANIO centers. Photo sets included bird's eye, lateral, and anterior-posterior views. The sagittal synostosis photo score was discussed in the working group, and consensus was obtained on its contents. Interrater agreement was assessed with weighted Fleiss' Kappa and intraclass correlation coefficients.The photo score consisted of frontal bossing, elongated skull, biparietal narrowness, temporal hollowing, vertex line depression, occipital bullet, and overall phenotype. Each item was scored as normal, mild, moderate, or severe. Results from 36 scaphocephaly patients scored by 20 raters showed kappa values ranging from 0.38 [95% bootstrap CI: 0.31, 0.45] for biparietal narrowness to 0.56 [95% bootstrap CI: 0.47, 0.64] for frontal bossing. Agreement was highest for the sum score of individual items [intraclass correlation coefficients agreement 0.69 [95% CI: 0.57, 0.82]. This is the first large-scale multicenter study in which experts investigated a photo score to assess the severity of sagittal synostosis phenotypical characteristics. Agreement on phenotypical characteristics was suboptimal (fair-moderate agreement) and highest for the summed score of individual photo score items (substantial agreement), indicating that although experts interpret phenotypical characteristics differently, there is consensus on overall phenotypical severity.
AB - European Reference Network (ERN) CRANIO is focused on optimizing care for patients with rare or complex craniofacial anomalies, including craniosynostosis and/or rare ear, nose, and throat disorders. The main goal of ERN CRANIO is to collect uniform data on treatment outcomes for multicenter comparison. We aimed to develop a reproducible and reliable suture-specific photo score that can be used for cross-center comparison of phenotypical severity of sagittal synostosis and aesthetic outcome of treatment. We conducted a retrospective study among nonsyndromic sagittal synostosis patients aged <19 years. We included preoperative and postoperative photo sets from 6 ERN CRANIO centers. Photo sets included bird's eye, lateral, and anterior-posterior views. The sagittal synostosis photo score was discussed in the working group, and consensus was obtained on its contents. Interrater agreement was assessed with weighted Fleiss' Kappa and intraclass correlation coefficients.The photo score consisted of frontal bossing, elongated skull, biparietal narrowness, temporal hollowing, vertex line depression, occipital bullet, and overall phenotype. Each item was scored as normal, mild, moderate, or severe. Results from 36 scaphocephaly patients scored by 20 raters showed kappa values ranging from 0.38 [95% bootstrap CI: 0.31, 0.45] for biparietal narrowness to 0.56 [95% bootstrap CI: 0.47, 0.64] for frontal bossing. Agreement was highest for the sum score of individual items [intraclass correlation coefficients agreement 0.69 [95% CI: 0.57, 0.82]. This is the first large-scale multicenter study in which experts investigated a photo score to assess the severity of sagittal synostosis phenotypical characteristics. Agreement on phenotypical characteristics was suboptimal (fair-moderate agreement) and highest for the summed score of individual photo score items (substantial agreement), indicating that although experts interpret phenotypical characteristics differently, there is consensus on overall phenotypical severity.
KW - aesthetic
KW - phenotype
KW - photo score
KW - Sagittal synostosis
KW - scaphocephaly
KW - 3126 Surgery, anesthesiology, intensive care, radiology
U2 - 10.1097/SCS.0000000000009732
DO - 10.1097/SCS.0000000000009732
M3 - Article
C2 - 37702534
AN - SCOPUS:85175728589
SN - 1049-2275
VL - 34
SP - 2279
EP - 2283
JO - Journal of Craniofacial Surgery
JF - Journal of Craniofacial Surgery
IS - 8
ER -