What Is the Difference in Cranial Base Morphology in Isolated and Syndromic Bicoronal Synostosis?

Lu, Xiaona; Forte, Antonio J.; Wilson, Alexander T.; Park, Kitae Eric; Allam, Omar; Mozaffari, Mohammad Ali; Alperovich, Michael; Steinbacher, Derek M.; Alonso, Nivaldo; Persing, John A.

doi:10.1097/prs.0000000000007068

Cited by 10 publications

(10 citation statements)

References 46 publications

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“…All of the studies included were published in peer-reviewed journals. Six of the 13 studies were conducted in the United States 23 – 28 , three in Netherlands 29 – 31 , and one in France 32 , Brazil 33 , Japan 34 and Italy 35 . The most common method used in the studies was cephalometric measurement.…”

Section: Resultsmentioning

confidence: 99%

“…38 . A reduced anterior cranial base, despite AS's smaller maxilla, can make the SNA similar to that of controls because the N point would be located further posteriorly 23 , 35 .…”

Section: Discussionmentioning

confidence: 99%

“…In AS, synchondrosis of the upper and lower jaw occurs in the same time, between the ages of 2 and 6 23 , 42 . Syndromes may immobilize their midline cranial base due to the advanced closure of the Sphenooccipital and Ethmosphenoid synchondroses 23 . The development of the midface in a transverse direction is likely to be disproportionately compounded.…”

Section: Discussionmentioning

confidence: 99%

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Craniofacial morphology in Apert syndrome: a systematic review and meta-analysis

Alam

Alfawzan

Srivastava

et al. 2022

Sci Rep

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This meta-analysis aims to compare Apert syndrome (AS) patients with non-AS populations (not clinically or genetically diagnosed) on craniofacial cephalometric characteristics (CCC) to combine publicly available scientific information while also improving the validity of primary study findings. A comprehensive search was performed in the following databases: PubMed, Google Scholar, Scopus, Medline, and Web of Science, an article published between 1st January 2000 to October 17th, 2021. PRISMA (Preferred Reporting Items for Systematic Reviews and Meta-Analyses) guidelines were followed to carry out this systematic review. We used the PECO system to classify people with AS based on whether or not they had distinctive CCC compared to the non-AS population. Following are some examples of how PECO has been used: People with AS are labeled P; clinical or genetic diagnosis of AS is labeled E; individuals without AS are labeled C; CCC of AS are labeled O. Using the Newcastle–Ottawa Quality-Assessment-Scale, independent reviewers assessed the articles' methodological quality and extracted data. 13 studies were included in the systematic review. 8 out of 13 studies were score 7–8 in NOS scale, which indicated that most of the studies were medium to high qualities. Six case–control studies were analyzed for meta-analysis. Due to the wide range of variability in CCC, we were only able to include data from at least three previous studies. There was a statistically significant difference in N-S-PP (I2: 76.56%; P = 0.014; CI 1.27 to − 0.28) and Greater wing angle (I2: 79.07%; P = 0.008; CI 3.07–1.17) between AS and control subjects. Cleft palate, anterior open bite, crowding in the upper jaw, and hypodontia occurred more frequently among AS patients. Significant shortening of the mandibular width, height and length is the most reported feature in AS patients. CT scans can help patients with AS decide whether to pursue orthodontic treatment alone or to have their mouth surgically expanded. The role of well-informed orthodontic and maxillofacial practitioners is critical in preventing and rehabilitating oral health issues.

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Section: Resultsmentioning

confidence: 99%

“…38 . A reduced anterior cranial base, despite AS's smaller maxilla, can make the SNA similar to that of controls because the N point would be located further posteriorly 23 , 35 .…”

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

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Craniofacial morphology in Apert syndrome: a systematic review and meta-analysis

Alam

Alfawzan

Srivastava

et al. 2022

Sci Rep

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“…4,5 There are distinct and specific influences of isolated cranial vault suture synostosis and craniosynostosis syndromes on the development of the skull base (i.e., the interface between cranial vault and facial skeletons). 6,7 The anatomy of anterior and middle skull base has a great influence in the development of orbital and periorbital morphology. 8,9 As observed in a previous pilot study of a small group of Apert syndrome cases (n = 18), development of convexity of the anterior lateral orbital wall was not seen in Crouzon syndrome patients.…”

Section: Ementioning

confidence: 99%

Influence of Nonsyndromic Bicoronal Synostosis and Syndromic Influences on Orbit and Periorbital Malformation

Forte

Dinis

et al. 2022

Plastic &Amp; Reconstructive Surgery

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Background: Oculoorbital disproportion in patients with craniosynostosis has similarities and dissimilarities between syndromic and nonsyndromic cases. The authors hypothesized that these two conditions have specific individual influences as they relate to development of the orbital and periorbital skeletons. Methods: A total of 133 preoperative computed tomography scans (nonsyndromic bicoronal synostosis, n = 38; Apert syndrome bicoronal synostosis subtype, n = 33; Crouzon syndrome bicoronal synostosis subtype, n = 10; controls, n = 52) were included. Craniometric and volumetric analyses related to the orbit and periorbital anatomy were performed. Results: Orbital cavity volume was mildly restricted in nonsyndromic bicoronal synostosis (7 percent, p = 0.147), but more so in Apert and Crouzon syndromes [17 percent (p = 0.002) and 21 percent (p = 0.005), respectively]. The sphenoid side angle in Apert syndrome was wider than when compared to Crouzon syndrome (p = 0.043). The ethmoid side angle in Apert patients, however, was narrower (p = 0.066) than that in Crouzon patients. Maxilla anteroposterior length was more restricted in Apert syndrome than Crouzon syndrome (21 percent, p = 0.003) and nonsyndromic cases (26 percent, p < 0.001). The posterior nasal spine position was retruded in Crouzon syndrome (39 percent, p < 0.001), yet the anterior nasal spine position was similar in Apert and Crouzon syndromes. Conclusions: Orbit and periorbital malformation in syndromic craniosynostosis is likely the combined influence of syndromic influences and premature suture fusion. Apert syndrome expanded the anteriorly contoured lateral orbital wall associated with bicoronal synostosis, whereas Crouzon syndrome had more infraorbital rim retrusion, resulting in more severe exorbitism. Apert syndrome developed maxillary hypoplasia, in addition to the maxillary retrusion, observed in Crouzon syndrome and nonsyndromic bicoronal synostosis patients. CLINICAL QUESTION/LEVEL OF EVIDENCE: Risk, II.

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“…Studies by Burdi and Kreiborg bucked traditional thinking at the time in suggesting that fusion of skull base synchondroses rather than major sutures leads to midface growth restriction (Burdi et al, 1986; Kreiborg et al, 1993). Multiple studies implicate the spheno-occipital synchondrosis (SOS) (McGrath et al, 2012; Goldstein et al, 2014; Tahiri et al, 2014) and sphenoethmoidal synchondrosis (SES) (Burdi et al, 1986; Lu et al, 2020a) as minor sutures, which limit midface advancement when prematurely fused in Crouzon syndrome. Facial and circummaxillary sutures have also since been implicated in midface retrusion (Purushothaman et al, 2011; Meazzini et al, 2020).…”

Section: Introductionmentioning

confidence: 99%

Morphology of the Occipital Bones and Foramen Magnum Resulting From Premature Minor Suture Fusion in Crouzon Syndrome

Layton

Pontier

Bins

et al. 2022

The Cleft Palate Craniofacial Journal

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To identify skull-base growth patterns in Crouzon syndrome, we hypothesized premature minor suture fusion restricts occipital bone development, secondarily limiting foramen magnum expansion. Skull-base suture closure degree and cephalometric measurements were retrospectively studied using preoperative computed tomography (CT) scans and multiple linear regression analysis. Evaluation of multi-institutional CT images and 3D reconstructions from Wake Forest’s Craniofacial Imaging Database (WFCID). Sixty preoperative patients with Crouzon syndrome under 12 years-old were selected from WFCID. The control group included 60 age- and sex-matched patients without craniosynostosis or prior craniofacial surgery. None 2D and 3D cephalometric measurements. 3D volumetric evaluation of the basioccipital, exo-occipital, and supraoccipital bones revealed decreased growth in Crouzon syndrome, attributed solely to premature minor suture fusion. Spheno-occipital (β = −398.75; P < .05) and petrous-occipital (β = −727.5; P < .001) suture fusion reduced growth of the basioccipital bone; lambdoid suture (β = −14 723.1; P < .001) and occipitomastoid synchondrosis (β = −16 419.3; P < .001) fusion reduced growth of the supraoccipital bone; and petrous-occipital suture (β = −673.3; P < .001), anterior intraoccipital synchondrosis (β = −368.47; P < .05), and posterior intraoccipital synchondrosis (β = −6261.42; P < .01) fusion reduced growth of the exo-occipital bone. Foramen magnum morphology is restricted in Crouzon syndrome but not directly caused by early suture fusion. Premature minor suture fusion restricts the volume of developing occipital bones providing a plausible mechanism for observed foramen magnum anomalies.

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What Is the Difference in Cranial Base Morphology in Isolated and Syndromic Bicoronal Synostosis?

Cited by 10 publications

References 46 publications

Craniofacial morphology in Apert syndrome: a systematic review and meta-analysis

Craniofacial morphology in Apert syndrome: a systematic review and meta-analysis

Influence of Nonsyndromic Bicoronal Synostosis and Syndromic Influences on Orbit and Periorbital Malformation

Morphology of the Occipital Bones and Foramen Magnum Resulting From Premature Minor Suture Fusion in Crouzon Syndrome

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