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Published in: Clinical Oral Investigations 7/2019

Open Access 01-07-2019 | Original Article

Deviating dental arch morphology in mild coronal craniosynostosis syndromes

Authors: T. M. Choi, L. Kragt, J. A. C. Goos, I. M. J. Mathijssen, E. B. Wolvius, E. M. Ongkosuwito

Published in: Clinical Oral Investigations | Issue 7/2019

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Abstract

Objectives

To determine whether the intramaxillary relationship of patients with Muenke syndrome and Saethre-Chotzen syndrome or TCF12-related craniosynostosis are systematically different than those of a control group.

Material and methods

Forty-eight patients (34 patients with Muenke syndrome, 8 patients with Saethre-Chotzen syndrome, and 6 patients with TCF12-related craniosynostosis) born between 1982 and 2010 (age range 4.84 to 16.83 years) that were treated at the Department of Oral Maxillofacial Surgery, Special Dental Care and Orthodontics, Children’s Hospital Erasmus University Medical Center, Sophia, Rotterdam, the Netherlands, were included. Forty-seven syndromic patients had undergone one craniofacial surgery according to the craniofacial team protocol. The dental arch measurements intercanine width (ICW), intermolar width (IMW), arch depth (AD), and arch length (AL) were calculated. The control group existed of 329 nonsyndromic children.

Results

All dental arch dimensions in Muenke (ICW, IMW, AL, p < 0.001, ADmax, p = 0.008; ADman, p = 0.002), Saethre-Chotzen syndrome, or TCF12-related craniosynostosis patients (ICWmax, p = 0.005; ICWman, IMWmax, AL, p < 0.001) were statistically significantly smaller than those of the control group.

Conclusions

In this study, we showed that the dental arches of the maxilla and the mandible of patients with Muenke syndrome and Saethre-Chotzen syndrome or TCF12-related craniosynostosis are smaller compared to those of a control group.

Clinical relevance

To gain better understanding of the sutural involvement in the midface and support treatment capabilities of medical and dental specialists in these patients, we suggest the concentration of patients with Muenke and Saethre-Chotzen syndromes or TCF12-related craniosynostosis in specialized teams for a multi-disciplinary approach and treatment.
Literature
1.
go back to reference Reitsma JH, Ongkosuwito EM, Buschang PH, Prahl-Andersen B (2012) Facial growth in patients with Apert and Crouzon syndromes compared to normal children. Cleft Palate Craniofac J 49:185–193CrossRef Reitsma JH, Ongkosuwito EM, Buschang PH, Prahl-Andersen B (2012) Facial growth in patients with Apert and Crouzon syndromes compared to normal children. Cleft Palate Craniofac J 49:185–193CrossRef
2.
go back to reference Stavropoulos D, Bartzela T, Bronkhorst E, Mohlin B, Hagberg C (2011) Dental agenesis patterns of permanent teeth in Apert syndrome. Eur J Oral Sci 119:198–203CrossRef Stavropoulos D, Bartzela T, Bronkhorst E, Mohlin B, Hagberg C (2011) Dental agenesis patterns of permanent teeth in Apert syndrome. Eur J Oral Sci 119:198–203CrossRef
3.
go back to reference Stavropoulos D, Bartzela T, Tarnow P, Mohlin B, Kahnberg KE, Hagberg C (2011) Dental agenesis patterns in Crouzon syndrome. Swed Dent J 35:195–201PubMed Stavropoulos D, Bartzela T, Tarnow P, Mohlin B, Kahnberg KE, Hagberg C (2011) Dental agenesis patterns in Crouzon syndrome. Swed Dent J 35:195–201PubMed
4.
go back to reference Reitsma JH, Elmi P, Ongkosuwito EM, Buschang PH, Prahl-Andersen B (2013) A longitudinal study of dental arch morphology in children with the syndrome of Crouzon or Apert. Eur J Oral Sci 121:319–327CrossRef Reitsma JH, Elmi P, Ongkosuwito EM, Buschang PH, Prahl-Andersen B (2013) A longitudinal study of dental arch morphology in children with the syndrome of Crouzon or Apert. Eur J Oral Sci 121:319–327CrossRef
5.
go back to reference Kress W, Schropp C, Lieb G, Petersen B, Büsse-Ratzka M, Kunz J, Reinhart E, Schäfer WD, Sold J, Hoppe F, Pahnke J, Trusen A, Sörensen N, Krauss J, Collmann H (2006) Saethre-Chotzen syndrome caused by TWIST 1 gene mutations: functional differentiation from Muenke coronal synostosis syndrome. Eur J Hum Genet 14:39–48CrossRef Kress W, Schropp C, Lieb G, Petersen B, Büsse-Ratzka M, Kunz J, Reinhart E, Schäfer WD, Sold J, Hoppe F, Pahnke J, Trusen A, Sörensen N, Krauss J, Collmann H (2006) Saethre-Chotzen syndrome caused by TWIST 1 gene mutations: functional differentiation from Muenke coronal synostosis syndrome. Eur J Hum Genet 14:39–48CrossRef
6.
go back to reference Honnebier MB, Cabiling DS, Hetlinger M, McDonald-McGinn DM, Zackai EH, Bartlett SP (2008) The natural history of patients treated for Fgfr3-associated (Muenke-type) craniosynostosis. Plast Reconstr Surg 121:919–931CrossRef Honnebier MB, Cabiling DS, Hetlinger M, McDonald-McGinn DM, Zackai EH, Bartlett SP (2008) The natural history of patients treated for Fgfr3-associated (Muenke-type) craniosynostosis. Plast Reconstr Surg 121:919–931CrossRef
7.
go back to reference Agochukwu NB, Solomon BD, Muenke M (2012) Impact of genetics on the diagnosis and clinical management of syndromic craniosynostoses. Childs Nerv Syst 28:1447–1463CrossRef Agochukwu NB, Solomon BD, Muenke M (2012) Impact of genetics on the diagnosis and clinical management of syndromic craniosynostoses. Childs Nerv Syst 28:1447–1463CrossRef
8.
go back to reference Buchanan EP, Xue AS, Hollier LH Jr (2014) Craniofacial syndromes. Plast Reconstr Surg 134:128e–153eCrossRef Buchanan EP, Xue AS, Hollier LH Jr (2014) Craniofacial syndromes. Plast Reconstr Surg 134:128e–153eCrossRef
9.
go back to reference Moloney DM, Wall SA, Ashworth GJ, Oldridge M, Glass IA, Francomano CA, Muenke M, Wilkie AOM (1997) Prevalence of Pro250Arg mutation of fibroblast growth factor receptor 3 in coronal craniosynostosis. Lancet 349:1059–1062CrossRef Moloney DM, Wall SA, Ashworth GJ, Oldridge M, Glass IA, Francomano CA, Muenke M, Wilkie AOM (1997) Prevalence of Pro250Arg mutation of fibroblast growth factor receptor 3 in coronal craniosynostosis. Lancet 349:1059–1062CrossRef
10.
go back to reference de Jong T, Mathijssen IM, Hoogeboom AJ (2011) Additional phenotypic features of Muenke syndrome in 2 Dutch families. J Craniofac Surg 22:571–575CrossRef de Jong T, Mathijssen IM, Hoogeboom AJ (2011) Additional phenotypic features of Muenke syndrome in 2 Dutch families. J Craniofac Surg 22:571–575CrossRef
11.
go back to reference Sharma VP, Fenwick AL, Brockop MS, McGowan SJ, Goos JA, Hoogeboom AJ, Brady AF, Jeelani NO, Lynch SA, Mulliken JB, Murray DJ, Phipps JM, Sweeney E, Tomkins SE, Wilson LC, Bennett S, Cornall RJ, Broxholme J, Kanapin A; 500 Whole-Genome Sequences (WGS500) Consortium, Johnson D, Wall SA, van der Spek PJ, Mathijssen IM, Maxson RE, Twigg SR, Wilkie AO (2013) Mutations in TCF12, encoding a basic helix-loop-helix partner of TWIST1, are a frequent cause of coronal craniosynostosis. Nat Genet 45:304–307 Sharma VP, Fenwick AL, Brockop MS, McGowan SJ, Goos JA, Hoogeboom AJ, Brady AF, Jeelani NO, Lynch SA, Mulliken JB, Murray DJ, Phipps JM, Sweeney E, Tomkins SE, Wilson LC, Bennett S, Cornall RJ, Broxholme J, Kanapin A; 500 Whole-Genome Sequences (WGS500) Consortium, Johnson D, Wall SA, van der Spek PJ, Mathijssen IM, Maxson RE, Twigg SR, Wilkie AO (2013) Mutations in TCF12, encoding a basic helix-loop-helix partner of TWIST1, are a frequent cause of coronal craniosynostosis. Nat Genet 45:304–307
12.
go back to reference Paumard-Hernandez B, Berges-Soria J, Barroso E et al (2015) Expanding the mutation spectrum in 182 Spanish probands with craniosynostosis: identification and characterization of novel Tcf12 variants. Eur J Hum Genet 23:907–914CrossRef Paumard-Hernandez B, Berges-Soria J, Barroso E et al (2015) Expanding the mutation spectrum in 182 Spanish probands with craniosynostosis: identification and characterization of novel Tcf12 variants. Eur J Hum Genet 23:907–914CrossRef
13.
go back to reference Howard TD, Paznekas WA, Green ED, Chiang LC, Ma N, Ortiz De Luna RI, Garcia Delgado C, Gonzalez-Ramos M, Kline AD, Jabs EW (1997) Mutations in TWIST, a basic helix-loop-helix transcription factor, in Saethre-Chotzen syndrome. Nat Genet 15:36–41CrossRef Howard TD, Paznekas WA, Green ED, Chiang LC, Ma N, Ortiz De Luna RI, Garcia Delgado C, Gonzalez-Ramos M, Kline AD, Jabs EW (1997) Mutations in TWIST, a basic helix-loop-helix transcription factor, in Saethre-Chotzen syndrome. Nat Genet 15:36–41CrossRef
14.
go back to reference Goos JA, Fenwick AL, Swagemakers SM et al (2016) Identification of intragenic exon deletions and duplication of TCF12 by whole genome or targeted sequencing as a cause of TCF12-related craniosynostosis. Hum Mutat 37:732–736CrossRef Goos JA, Fenwick AL, Swagemakers SM et al (2016) Identification of intragenic exon deletions and duplication of TCF12 by whole genome or targeted sequencing as a cause of TCF12-related craniosynostosis. Hum Mutat 37:732–736CrossRef
15.
go back to reference di Rocco F, Baujat G, Arnaud E, Renier D, Laplanche JL, Daire VC, Collet C (2014) Clinical spectrum and outcomes in families with coronal synostosis and Tcf12 mutations. Eur J Hum Genet 22:1413–1416CrossRef di Rocco F, Baujat G, Arnaud E, Renier D, Laplanche JL, Daire VC, Collet C (2014) Clinical spectrum and outcomes in families with coronal synostosis and Tcf12 mutations. Eur J Hum Genet 22:1413–1416CrossRef
16.
go back to reference Prahl-Andersen B, Kowalski CJ (1973) A mixed longitudinal, interdisciplinary study of the growth and development of Dutch children. Growth 37:281–295PubMed Prahl-Andersen B, Kowalski CJ (1973) A mixed longitudinal, interdisciplinary study of the growth and development of Dutch children. Growth 37:281–295PubMed
17.
go back to reference Lippold C, Kirschneck C, Schreiber K, Abukiress S, Tahvildari A, Moiseenko T, Danesh G (2015) Methodological accuracy of digital and manual model analysis in orthodontics—a retrospective clinical study. Comput Biol Med 62:103–109CrossRef Lippold C, Kirschneck C, Schreiber K, Abukiress S, Tahvildari A, Moiseenko T, Danesh G (2015) Methodological accuracy of digital and manual model analysis in orthodontics—a retrospective clinical study. Comput Biol Med 62:103–109CrossRef
18.
go back to reference Shrout PE, Fleiss JL (1979) Intraclass correlations: uses in assessing rater reliability. Psychol Bull 86:420–428CrossRef Shrout PE, Fleiss JL (1979) Intraclass correlations: uses in assessing rater reliability. Psychol Bull 86:420–428CrossRef
19.
go back to reference Ousterhout DK, Melsen B (1982) Cranial base deformity in Apert’s syndrome. Plast Reconstr Surg 69:254–263CrossRef Ousterhout DK, Melsen B (1982) Cranial base deformity in Apert’s syndrome. Plast Reconstr Surg 69:254–263CrossRef
20.
go back to reference Friede H, Lilja J, Andersson H, Johanson B (1983) Growth of the anterior cranial base after craniotomy in infants with premature synostosis of the coronal suture. Scand J Plast Reconstr Surg 17:99–108CrossRef Friede H, Lilja J, Andersson H, Johanson B (1983) Growth of the anterior cranial base after craniotomy in infants with premature synostosis of the coronal suture. Scand J Plast Reconstr Surg 17:99–108CrossRef
21.
go back to reference Krarup S, Darvann TA, Larsen P, Marsh JL, Kreiborg S (2005) Three-dimensional analysis of mandibular growth and tooth eruption. J Anat 207:669–682CrossRef Krarup S, Darvann TA, Larsen P, Marsh JL, Kreiborg S (2005) Three-dimensional analysis of mandibular growth and tooth eruption. J Anat 207:669–682CrossRef
22.
go back to reference Kreiborg S, Cohen MM Jr (1992) The oral manifestations of Apert syndrome. J Craniofac Genet Dev Biol 12:41–48PubMed Kreiborg S, Cohen MM Jr (1992) The oral manifestations of Apert syndrome. J Craniofac Genet Dev Biol 12:41–48PubMed
23.
go back to reference Allen D, Rebellato J, Sheats R, Ceron AM (2003) Skeletal and dental contributions to posterior crossbites. Angle Orthod 73:515–524PubMed Allen D, Rebellato J, Sheats R, Ceron AM (2003) Skeletal and dental contributions to posterior crossbites. Angle Orthod 73:515–524PubMed
24.
go back to reference Vucic S, de Vries E, Eilers PH, Willemsen SP, Kuijpers MA, Prahl-Andersen B, Jaddoe VW, Hofman A, Wolvius EB, Ongkosuwito EM (2014) Secular trend of dental development in Dutch children. Am J Phys Anthropol 155:91–98CrossRef Vucic S, de Vries E, Eilers PH, Willemsen SP, Kuijpers MA, Prahl-Andersen B, Jaddoe VW, Hofman A, Wolvius EB, Ongkosuwito EM (2014) Secular trend of dental development in Dutch children. Am J Phys Anthropol 155:91–98CrossRef
25.
go back to reference Fredriks AM, van Buuren S, Burgmeijer RJ, Meulmeester JF, Beuker RJ, Brugman E, Roede MJ, Verloove-Vanhorick SP, Wit JM (2000) Continuing positive secular growth change in The Netherlands 1955-1997. Pediatr Res 47(3):316–323CrossRef Fredriks AM, van Buuren S, Burgmeijer RJ, Meulmeester JF, Beuker RJ, Brugman E, Roede MJ, Verloove-Vanhorick SP, Wit JM (2000) Continuing positive secular growth change in The Netherlands 1955-1997. Pediatr Res 47(3):316–323CrossRef
26.
go back to reference Fredriks AM, van Buuren S, Wit JM, Verloove-Vanhorick SP (2000) Body index measurements in 1996-7 compared with 1980. Arch Dis Child 82:107–112CrossRef Fredriks AM, van Buuren S, Wit JM, Verloove-Vanhorick SP (2000) Body index measurements in 1996-7 compared with 1980. Arch Dis Child 82:107–112CrossRef
27.
go back to reference Mul D, Fredriks AM, van Buuren S, Oostdijk W, Verloove-Vanhorick SP, Wit JM (2001) Pubertal development in The Netherlands 1965-1997. Pediatr Res 50:479–486CrossRef Mul D, Fredriks AM, van Buuren S, Oostdijk W, Verloove-Vanhorick SP, Wit JM (2001) Pubertal development in The Netherlands 1965-1997. Pediatr Res 50:479–486CrossRef
28.
go back to reference Woller JL, Kim KB, Behrents RG, Buschang PH (2014) An assessment of the maxilla after rapid maxillary expansion using cone beam computed tomography in growing children. Dental Press J Orthod 19:26–35CrossRef Woller JL, Kim KB, Behrents RG, Buschang PH (2014) An assessment of the maxilla after rapid maxillary expansion using cone beam computed tomography in growing children. Dental Press J Orthod 19:26–35CrossRef
Metadata
Title
Deviating dental arch morphology in mild coronal craniosynostosis syndromes
Authors
T. M. Choi
L. Kragt
J. A. C. Goos
I. M. J. Mathijssen
E. B. Wolvius
E. M. Ongkosuwito
Publication date
01-07-2019
Publisher
Springer Berlin Heidelberg
Published in
Clinical Oral Investigations / Issue 7/2019
Print ISSN: 1432-6981
Electronic ISSN: 1436-3771
DOI
https://doi.org/10.1007/s00784-018-2710-9

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