Deviating dental arch morphology in mild coronal craniosynostosis syndromes

Choi, T. M.; Kragt, Lea; Goos, Jacqueline A.C.; Mathijssen, Irene M J; Wolvius, Eppo B.; Ongkosuwito, Edwin M.

doi:10.1007/s00784-018-2710-9

Cited by 6 publications

(6 citation statements)

References 28 publications

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“…The vertical growth pattern, jaw complex, and proclination of the upper incisors (PC2), which is characterized by NSL/NL, NL/ML, and Ils/NL, were decreased in the Muenke syndrome and TCF12 -related craniosynostosis compared to patients of the Dutch control group. The proclination of the upper incisors can be the result of a relatively smaller maxilla in sagittal dimension with a normal mandible, causing the maxillary upper incisors to procline [ 16 ]. PC2 analysis for the Saethre-Chotzen syndrome was not different compared to patients of the Dutch control group.…”

Section: Discussionmentioning

confidence: 99%

“…Patients with Muenke syndrome, Saethre-Chotzen syndrome, and TCF12 -related craniosynostosis have a more vertical craniofacial buildup compared to the control group. Also, these syndromic patients have smaller dental arch dimensions [ 16 ]. Although these patients have distinctive craniofacial and dental features, the standard typical conventional therapy does not exist.…”

Section: Discussionmentioning

confidence: 99%

“…Subsequently, in these syndromes, this results in deviant dental arch dimensions [ 15 ]. Smaller maxillary dental arch dimensions also have been found in Muenke syndrome, Saethre-Chotzen syndrome, and TCF12 -related craniosynostosis [ 16 ]. Therefore, we expect that midface hypoplasia would also be present in these syndromic craniosynostosis patients.…”

Section: Introductionmentioning

confidence: 99%

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Craniofacial morphology and growth in Muenke syndrome, Saethre-Chotzen syndrome, and TCF12-related craniosynostosis

et al. 2021

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Objectives To determine whether the midface of patients with Muenke syndrome, Saethre-Chotzen syndrome, or TCF12-related craniosynostosis is hypoplastic compared to skeletal facial proportions of a Dutch control group. Material and methods We included seventy-four patients (43 patients with Muenke syndrome, 22 patients with Saethre-Chotzen syndrome, and 9 patients with TCF12-related craniosynostosis) who were referred between 1990 and 2020 (age range 4.84 to 16.83 years) and 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. The control group consisted of 208 healthy children. Results Cephalometric values comprising the midface were decreased in Muenke syndrome (ANB: β = –1.87, p = 0.001; and PC1: p < 0,001), Saethre-Chotzen syndrome (ANB: β = –1.76, p = 0.001; and PC1: p < 0.001), and TCF12-related craniosynostosis (ANB: β = –1.70, p = 0.015; and PC1: p < 0.033). Conclusions In this study, we showed that the midface is hypoplastic in Muenke syndrome, Saethre-Chotzen syndrome, and TCF12-related craniosynostosis compared to the Dutch control group. Furthermore, the rotation of the maxilla and the typical craniofacial buildup is significantly different in these three craniosynostosis syndromes compared to the controls. Clinical relevance The maxillary growth in patients with Muenke syndrome, Saethre-Chotzen syndrome, or TCF12-related craniosynostosis is impaired, leading to a deviant dental development. Therefore, timely orthodontic follow-up is recommended. In order to increase expertise and support treatment planning by medical and dental specialists for these patients, and also because of the specific differences between the syndromes, we recommend the management of patients with Muenke syndrome, Saethre-Chotzen syndrome, or TCF12-related craniosynostosis in specialized multidisciplinary teams.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Craniofacial morphology and growth in Muenke syndrome, Saethre-Chotzen syndrome, and TCF12-related craniosynostosis

et al. 2021

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“…Because this mutation has been found recently, an extensive description of the phenotype is not yet available ( 11 ). All these three craniosynostosis syndromes also have smaller dental arch dimensions ( 17 ).…”

Section: Introductionmentioning

confidence: 99%

Evaluation of dental maturity in Muenke syndrome, Saethre-Chotzen syndrome, and TCF12-related craniosynostosis

Choi

Kramer

Goos

et al. 2021

European Journal of Orthodontics

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Objectives To determine whether dental maturity (dental development) was delayed in patients with Muenke syndrome, Saethre-Chotzen syndrome, and TCF12-related craniosynostosis, compared with a Dutch control group without syndromes. Materials and methods This study included 60 patients (38 patients with Muenke syndrome, 17 patients with Saethre-Chotzen syndrome, and 5 with TCF12-related craniosynostosis), aged 5.8–16.8 years that were treated at the Department of Oral Maxillofacial Surgery, Special Dental Care, and Orthodontics, in Sophia Children’s Hospital, Erasmus University Medical Center, Rotterdam, the Netherlands. Dental age was calculated according to Demirjian’s index of dental maturity. The control group included 451 children without a syndrome. Results Compared with the control group, dental development was delayed by an average of one year in 5- to 8-year-old patients with Muenke syndrome (P = 0.007) and in 8- to 10-year-old patients with Saethre-Chotzen syndrome (P = 0.044), but not in patients with TCF12-related craniosynostosis. Conclusions Our results indicated that dental development was delayed by one year, on average, in patients with Muenke syndrome and Saethre-Chotzen syndrome, compared with a Dutch control group without syndromes. Implications Our findings have improved the understanding of dental development in patients with Muenke and Saethre-Chotzen syndrome. These results can provide guidance on whether the orthodontist needs to consider growth disturbances related to dental development.

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“…Though heterogeneous in terms of pattern of suture fusion, brachycephaly is the most frequently seen 2,3 . In terms of facial features, the most commonly described clinical features of SCS include facial asymmetry, a broad and depressed nasal bridge, low hairline, eyelid ptosis, telorbitism, low-set ears with a prominent superior and/or inferior crus, maxillary hypoplasia, mandibular prognathism, and a high-arched palate 4,5 . Less common symptoms include deviated nasal septum, tear duct stenosis, downward slanting palpebral fissures, blepharophimosis, visual impairment, strabismus, and hearing loss 2,6–8 .…”

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confidence: 99%