Craniofacial morphological variability in orthodontic patients with non-syndromic orofacial clefts: an approach using geometric morphometrics.
Cephalometry
Cleft lip and palate
Cleft palate
Craniofacial morphology
Geometric morphometrics
Shape
Journal
Clinical oral investigations
ISSN: 1436-3771
Titre abrégé: Clin Oral Investig
Pays: Germany
ID NLM: 9707115
Informations de publication
Date de publication:
02 Jul 2024
02 Jul 2024
Historique:
received:
27
11
2023
accepted:
20
06
2024
medline:
2
7
2024
pubmed:
2
7
2024
entrez:
2
7
2024
Statut:
epublish
Résumé
Orofacial clefts are complex congenital anomalies that call for comprehensive treatment based on a thorough assessment of the anatomy. This study aims to examine the effect of cleft type on craniofacial morphology using geometric morphometrics. We evaluated lateral cephalograms of 75 patients with bilateral cleft lip and palate, 63 patients with unilateral cleft lip and palate, and 76 patients with isolated cleft palate. Generalized Procrustes analysis was performed on 16 hard tissue landmark coordinates. Shape variability was studied with principal component analysis. In a risk model approach, the first nine principal components (PC) were used to examine the effect of cleft type. We found statistically significant differences in the mean shape between cleft types. The difference is greatest between bilateral cleft lip and palate and isolated cleft palate (distance of means 0.026, P = 0.0011). Differences between cleft types are most pronounced for PC4 and PC5 (P = 0.0001), which together account for 10% of the total shape variation. PC4 and PC5 show shape differences in the ratio of the upper to the lower face, the posterior mandibular height, and the mandibular angle. Cleft type has a statistically significant but weak effect on craniofacial morphological variability in patients with non-syndromic orofacial clefts, mainly in the vertical dimension. Understanding the effects of clefts on craniofacial morphology is essential to providing patients with treatment tailored to their specific needs. This study contributes to the literature particularly due to our risk model approach in lieu of a prediction model.
Identifiants
pubmed: 38954126
doi: 10.1007/s00784-024-05796-y
pii: 10.1007/s00784-024-05796-y
doi:
Types de publication
Journal Article
Langues
eng
Sous-ensembles de citation
IM
Pagination
409Informations de copyright
© 2024. The Author(s).
Références
Salari N, Darvishi N, Heydari M, Bokaee S, Darvishi F, Mohammadi M (2022) Global prevalence of cleft palate, cleft lip and cleft palate and lip: a comprehensive systematic review and meta-analysis. J Stomatol Oral Maxillofac Surg 123(2):110–120. https://doi.org/10.1016/j.jormas.2021.05.008
doi: 10.1016/j.jormas.2021.05.008
Weinberg SM, Neiswanger K, Martin RA, Mooney MP, Kane AA, Wenger SL, Losee JE et al (2006) The Pittsburgh oral-facial cleft study: expanding the Cleft phenotype. Background and justification. Cleft Palate-Cran J 43(1):7–20. https://doi.org/10.1597/04-122r1.1
doi: 10.1597/04-122r1.1
Goyenc YB, Gurel HG, Memili B (2008) Craniofacial morphology in children with operated complete unilateral cleft lip and palate. J Craniofac Surg 19(5):1396–1401. https://doi.org/10.1097/SCS.0b013e318184333d
doi: 10.1097/SCS.0b013e318184333d
van den Dungen GM, Ongkosuwito EM, Aartman IHA, Prahl-Andersen B (2008) Craniofacial morphology of Dutch patients with bilateral cleft lip and palate and noncleft controls at the age of 15 years. Cleft Palate-Cran J 45(6):661–666. https://doi.org/10.1597/07-166.1
doi: 10.1597/07-166.1
Bishara SE (2002) Facial and dental relationships in individuals with cleft lip and/or palate. Oral Maxil Surg Clin 14(4):411–424. https://doi.org/10.1016/S1042-3699(02)00043-2
doi: 10.1016/S1042-3699(02)00043-2
Jensen ED, Poirier BF, Oliver KJ, Roberts R, Anderson PJ, Jamieson LM (2023) Childhood experiences and perspectives of individuals with Orofacial clefts: a qualitative systematic review. Cleft Palate-Cran J 60(7):888–899. https://doi.org/10.1177/10556656221084542
doi: 10.1177/10556656221084542
de Cuyper E, Dochy F, de Leenheer E, van Hoecke H (2019) The impact of cleft lip and/or palate on parental quality of life: a pilot study. Int J Pediatr Otorhi 126:109598. https://doi.org/10.1016/j.ijporl.2019.109598
doi: 10.1016/j.ijporl.2019.109598
Mossey PA, Modell B (2012) Epidemiology of oral clefts 2012: an international perspective. Front Oral Biol 16:1–18. https://doi.org/10.1159/000337464
doi: 10.1159/000337464
Dixon MJ, Marazita ML, Beaty TH, Murray JC (2011) Cleft lip and palate: understanding genetic and environmental influences. Nat Rev Genet 12(3):167–178. https://doi.org/10.1038/nrg2933
doi: 10.1038/nrg2933
Krogman WM, Jain RB, Oka SW (1982) Craniofacial growth in different cleft types from one month to ten years. Cleft Palate J 19(3):206–211
López-Giménez A, Silvestre-Rangil J, Silvestre F-J, Paredes-Gallardo V (2018) Craniofacial cephalometric morphologies in different cleft types: a retrospective cross-sectional study of 212 patients. Oral Radiol 34(2):127–135. https://doi.org/10.1007/s11282-017-0290-z
doi: 10.1007/s11282-017-0290-z
Smahel Z, Hradiský D, Müllerová Z (1999) Multivariate comparison of craniofacial morphology in different types of facial clefts. Acta Chir Plast 41(2):59–65
Bishara SE, de Arrendondo RS, Vales HP, Jakobsen JR (1985) Dentofacial relationships in persons with unoperated clefts: comparisons between three cleft types. Amer J Orthod 87(6):481–507. https://doi.org/10.1016/0002-9416(85)90086-7
doi: 10.1016/0002-9416(85)90086-7
Krey K-F, Dannhauer K-H, Hemprich A, Reich S (2009) Studies on the craniofacial morphology of adult cleft patients using euclidean distance matrix analysis (EDMA): a cephalometric study. J Orofac Orthop 70(5):396–406. https://doi.org/10.1007/s00056-009-9920-3
doi: 10.1007/s00056-009-9920-3
Da Silva Filho OG, Carvalho Lauris RC, Capelozza Filho L, Semb G (1998) Craniofacial morphology in adult patients with unoperated complete bilateral cleft lip and palate. Cleft Palate-Cran J 35(2):111–119. https://doi.org/10.1597/1545-1569_1998_035_0111_cmiapw_2.3.co_2
doi: 10.1597/1545-1569_1998_035_0111_cmiapw_2.3.co_2
Liao Y-F, Yang I-Y, Wang R, Yun C, Huang C-S (2010) Two-stage palate repair with delayed hard palate closure is related to favorable maxillary growth in unilateral cleft lip and palate. Plast Reconstr Surg 125(5):1503–1510. https://doi.org/10.1097/PRS.0b013e3181d5132a
doi: 10.1097/PRS.0b013e3181d5132a
Liu Y, Huang H, Shi B, Wang Y (2020) The influence of lip repair on the growth of Craniofacial structures in bilateral cleft lip and palate patients with unoperated palate from childhood to Adulthood. J Craniofac Surg 31(5):1218–1222. https://doi.org/10.1097/SCS.0000000000006368
doi: 10.1097/SCS.0000000000006368
Li Y, Shi B, Song Q-G, Zuo H, Zheng Q (2006) Effects of lip repair on maxillary growth and facial soft tissue development in patients with a complete unilateral cleft of lip, alveolus and palate. J Cranio Maxill Surg 34(6):355–361. https://doi.org/10.1016/j.jcms.2006.03.005
doi: 10.1016/j.jcms.2006.03.005
Delaire J, Precious D (1986) Influence of the nasal septum on maxillonasal growth in patients with congenital labiomaxillary cleft. Cleft Palate J 23(4):270–277
Friede H (2007) Maxillary growth controversies after two-stage palatal repair with delayed hard palate closure in unilateral cleft lip and palate patients: perspectives from literature and personal experience. Cleft Palate-Cran J 44(2):129–136. https://doi.org/10.1597/06-037.1
doi: 10.1597/06-037.1
James Rohlf F, Marcus LF (1993) A revolution in morphometrics. Trends Ecol Evol 8(4):129–132. https://doi.org/10.1016/0169-5347(93)90024-J
doi: 10.1016/0169-5347(93)90024-J
Klingenberg CP (2013) Visualizations in geometric morphometrics: how to read and how to make graphs showing shape changes. Hystrix 24(1):15–24. https://doi.org/10.4404/hystrix-24.1-7691
doi: 10.4404/hystrix-24.1-7691
Bugaighis I, O’Higgins P, Tiddeman B, Mattick C, Ben Ali O, Hobson R (2010) Three-dimensional geometric morphometrics applied to the study of children with cleft lip and/or palate from the North East of England. Eur J Orthodont 32(5):514–521. https://doi.org/10.1093/ejo/cjp140
doi: 10.1093/ejo/cjp140
Doberschütz PH, Schwahn C, Krey K-F (2022) Cephalometric analyses for cleft patients: a statistical approach to compare the variables of Delaire’s craniofacial analysis to Bergen analysis. Clin Oral Invest 26(1):353–364. https://doi.org/10.1007/s00784-021-04006-3
doi: 10.1007/s00784-021-04006-3
Baccetti T, Franchi L, McNamara JA (2005) The cervical vertebral maturation (CVM) method for the Assessment of Optimal treatment timing in Dentofacial Orthopedics. Semin Orthod 11(3):119–129. https://doi.org/10.1053/j.sodo.2005.04.005
doi: 10.1053/j.sodo.2005.04.005
Hasund A (1974) Klinische Kephalometrie für die Bergen-Technik, 1st edn. University of Bergen, Bergen
Koch J (1969) Diagnose, Unterteilung und Dokumentation Der Lippen-, Kiefer-, Gaumen- Und Segelspalten. Deutsche Zahn-, Mund- Und Kieferheilkunde Mit Zentralblatt für die gesamte Zahn-, Mund- Und Kieferheilkunde; Organ Der Deutschen Gesellschaft für Zahn-. Mund- und Kieferheilkunde 52(5/6):187–196
Rohlf FJ, Slice D (1990) Extensions of the Procrustes Method for the optimal superimposition of landmarks. Syst Zool 39(1):40. https://doi.org/10.2307/2992207
doi: 10.2307/2992207
Zelditch ML, Swiderski DL, Sheets HD (2004) Geometric morphometrics for biologists: a primer, 2 edn. Elsevier, Amsterdam. https://doi.org/10.1016/B978-0-12-778460-1.X5000-5
doi: 10.1016/B978-0-12-778460-1.X5000-5
Hernán MA, Robins JM (2020) Causal inference: what if. Chapman & Hall/CRC, Boca Raton, Fla
Pearl J (2009) Causality: models, reasoning, and inference, 2 edn. Cambridge University Press, Cambridge
doi: 10.1017/CBO9780511803161
VanderWeele TJ, Rothman KJ, Lash TL (2021) Confounding and confounders. In: Lash TL, VanderWeele TJ, Haneuse S, Rothman KJ (eds) Modern epidemiology, 4th edn. Wolters Kluwer, Philadelphia, pp 263–286
Rothman KJ (2012) Epidemiology: An Introduction, Second edition. Oxford University Press, Oxford. https://doi.org/10.1007/s10654-012-9732-4
Tennant PWG, Murray EJ, Arnold KF, Berrie L, Fox MP, Gadd SC et al (2021) Use of directed acyclic graphs (DAGs) to identify confounders in applied health research: review and recommendations. Int J Epidemiol 50(2):620–632. https://doi.org/10.1093/ije/dyaa213
doi: 10.1093/ije/dyaa213
Textor J, Hardt J, Knüppel S (2011) DAGitty: a graphical tool for analyzing causal diagrams. Epidemiology 22(5):745. https://doi.org/10.1097/ede.0b013e318225c2be
doi: 10.1097/ede.0b013e318225c2be
Adams DC, Otárola-Castillo E (2013) Geomorph: an r package for the collection and analysis of geometric morphometric shape data. Methods Ecol Evol 4(4):393–399. https://doi.org/10.1111/2041-210X.12035
doi: 10.1111/2041-210X.12035
Hotelling H (1933) Analysis of a complex of statistical variables into principal components. J Educ Psychol 24(6):417–441. https://doi.org/10.1037/h0071325
doi: 10.1037/h0071325
Cleall JF, BeGole EA, Chebib FS (1979) Craniofacial morphology: a principal component analysis. Amer J Orthod 75(6):650–666. https://doi.org/10.1016/0002-9416(79)90097-6
doi: 10.1016/0002-9416(79)90097-6
R Core Team (2021) R: a language and environment for statistical computing. R Foundation for Statistical Computing, Vienna, Austria
RStudio Team (2022) RStudio: Integrated development for R. RStudio, PBC. Boston, MA. http://www.rstudio.com/
Wasserstein RL, Lazar NA (2016) The ASA Statement on p -Values: context, process, and purpose. Am Stat 70(2):129–133. https://doi.org/10.1080/00031305.2016.1154108
doi: 10.1080/00031305.2016.1154108
Harrell FE (2015) Regression modeling strategies: with applications to Linear models, Logistics and Ordinal Regression, and Survival Analysis, 2nd edn. Springer, Cham. https://doi.org/10.1007/978-3-319-19425-7
doi: 10.1007/978-3-319-19425-7
James G, Witten D, Hastie T, Tibshirani R (2021) An introduction to statistical learning, 2nd edn. Springer, New York, NY. https://doi.org/10.1007/978-1-0716-1418-1
doi: 10.1007/978-1-0716-1418-1
Toro-Ibacache V, Cortés Araya J, Díaz Muñoz A, Manríquez Soto G (2014) Morphologic variability of nonsyndromic operated patients affected by cleft lip and palate: a geometric morphometric study. Am J Orthod Dentofac 146(3):346–354. https://doi.org/10.1016/j.ajodo.2014.06.002
doi: 10.1016/j.ajodo.2014.06.002
Latif A, Kuijpers MAR, Rachwalski M, Latief BS, Kuijpers-Jagtman AM, Fudalej PS (2020) Morphological variability in unrepaired bilateral clefts with and without cleft palate evaluated with geometric morphometrics. J Anat 236(3):425–433. https://doi.org/10.1111/joa.13118
doi: 10.1111/joa.13118
Ye B, Wu Y, Zhou Y, Jing H, Hu J, Zhang G (2015) A comparative cephalometric study for adult operated cleft palate and unoperated cleft palate patients. J Cranio Maxill Surg 43(7):1218–1223. https://doi.org/10.1016/j.jcms.2015.04.015
doi: 10.1016/j.jcms.2015.04.015
Viñas MJ, Galiotto-Barba F, Cortez-Lede MG, Rodríguez-González MÁ, Moral I, Delso E et al (2022) Craniofacial and three-dimensional palatal analysis in cleft lip and palate patients treated in Spain. Sci Rep 12(1):18837. https://doi.org/10.1038/s41598-022-23584-0
doi: 10.1038/s41598-022-23584-0
Da Silva Filho OG, Valladares Neto J, Capelloza Filho L, de Souza Freitas JA (2003) Influence of lip repair on craniofacial morphology of patients with complete bilateral cleft lip and palate. Cleft Palate-Cran J 40(2):144–153. https://doi.org/10.1597/1545-1569_2003_040_0144_iolroc_2.0.co_2
doi: 10.1597/1545-1569_2003_040_0144_iolroc_2.0.co_2
Friede H, Pruzansky S (1972) Longitudinal study of growth in bilateral cleft lip and palate, from infancy to adolescence. Plast Reconstr Surg 49(4):392–403. https://doi.org/10.1097/00006534-197204000-00005
doi: 10.1097/00006534-197204000-00005
Trotman C-A, Ross RB, Trotman CA, Ross RB (1993) Craniofacial growth in bilateral cleft lip and palate: ages six years to adulthood. Cleft Palate-Cran J 30(3):261–273. https://doi.org/10.1597/1545-1569_1993_030_0261_cgibcl_2.3.co_2
doi: 10.1597/1545-1569_1993_030_0261_cgibcl_2.3.co_2
Capelozza Filho L, Normando AD, Da Silva Filho OG (1996) Isolated influences of lip and palate surgery on facial growth: comparison of operated and unoperated male adults with UCLP. Cleft Palate-Cran J 33(1):51–56. https://doi.org/10.1597/1545-1569_1996_033_0051_iiolap_2.3.co_2
doi: 10.1597/1545-1569_1996_033_0051_iiolap_2.3.co_2
Good PM, Mulliken JB, Padwa BL (2007) Frequency of Le Fort I osteotomy after repaired cleft lip and palate or cleft palate. Cleft Palate-Cran J 44(4):396–401. https://doi.org/10.1597/06-075.1
doi: 10.1597/06-075.1
Cardini A, Seetah K, Barker G (2015) How many specimens do I need? Sampling error in geometric morphometrics: testing the sensitivity of means and variances in simple randomized selection experiments. Zoomorphology 134(2):149–163. https://doi.org/10.1007/s00435-015-0253-z
doi: 10.1007/s00435-015-0253-z
Allori AC, Mulliken JB, Meara JG, Shusterman S, Marcus JR (2017) Classification of cleft Lip/Palate: then and now. Cleft Palate-Cran J 54(2):175–188. https://doi.org/10.1597/14-080
doi: 10.1597/14-080
Formby WA, Nanda RS, Currier GF (1994) Longitudinal changes in the adult facial profile. Am J Orthod Dentofac 105(5):464–476. https://doi.org/10.1016/S0889-5406(94)70007-9
doi: 10.1016/S0889-5406(94)70007-9
Forsberg CM (1979) Facial morphology and ageing: a longitudinal cephalometric investigation of young adults. Eur J Orthodont 1(1):15–23. https://doi.org/10.1093/ejo/1.1.15
doi: 10.1093/ejo/1.1.15
Houston WJ, Maher RE, McElroy D, Sherriff M (1986) Sources of error in measurements from cephalometric radiographs. Eur J Orthodont 8(3):149–151. https://doi.org/10.1093/ejo/8.3.149
doi: 10.1093/ejo/8.3.149
Da Silveira HLD, Silveira HED (2006) Reproducibility of cephalometric measurements made by three radiology clinics. Angle Orthod 76(3):394–399
Lagravère MO, Low C, Flores-Mir C, Chung R, Carey JP, Heo G et al (2010) Intraexaminer and interexaminer reliabilities of landmark identification on digitized lateral cephalograms and formatted 3-dimensional cone-beam computerized tomography images. Am J Orthod Dentofac 137(5):598–604. https://doi.org/10.1016/j.ajodo.2008.07.018
doi: 10.1016/j.ajodo.2008.07.018
Heidbüchel KL, Kuijpers-Jagtman AM, Freihofer HP (1994) Facial growth in patients with bilateral cleft lip and palate: a cephalometric study. Cleft Palate-Cran J 31(3):210–216. https://doi.org/10.1597/1545-1569_1994_031_0210_fgipwb_2.3.co_2
doi: 10.1597/1545-1569_1994_031_0210_fgipwb_2.3.co_2
Cramon-Taubadel v, Frazier N, Lahr BC MM (2007) The problem of assessing landmark error in geometric morphometrics: theory, methods, and modifications. Am J Phys Anthropol 134(1):24–35. https://doi.org/10.1002/ajpa.20616
doi: 10.1002/ajpa.20616
Klingenberg CP (2021) How exactly did the nose get that long? A critical rethinking of the Pinocchio Effect and how shape changes relate to landmarks. Evol Biol 48(1):115–127. https://doi.org/10.1007/s11692-020-09520-y
doi: 10.1007/s11692-020-09520-y