Preoperative Osseous Dysmorphology in Unilateral Complete Cleft Lip and Palate: A Quantitative Analysis of Computed Tomography Data (original) (raw)
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A Multivariate Analysis of Unilateral Cleft Lip and Palate Facial Skeletal Morphology
Unilateral cleft lip and palate (UCLP) occurs when the maxillary and nasal facial prominences fail to fuse correctly during development, resulting in a palatal cleft and clefted soft and hard tissues of the dentoalveolus. The UCLP deformity may compromise an individual's ability to eat, chew, and speak. In this retrospective cross-sectional study, cone beam computed tomography (CBCT) images of 7-17-year-old individuals born with UCLP (n ¼ 24) and age-and sex-matched controls (n ¼ 24) were assessed. Coordinate values of three-dimensional anatomical landmarks (n ¼ 32) were recorded from each CBCT image. Data were evaluated using principal coordinates analysis (PCOORD) and Euclidean distance matrix analysis (EDMA). Approximately 40% of morphometric variation is captured by PCOORD axes 1-3, and the negative and positive ends of each axis are associated with specific patterns of morphological differences. Approximately 36% of facial skeletal measures significantly differ by confidence interval testing (a ¼ 0.10) between samples. Although significant form differences occur across the facial skeleton, strong patterns of morphological differences were localized to the lateral and superioinferior aspects of the nasal aperture, particularly on the clefted side of the face. The UCLP deformity strongly influences facial skeletal morphology of the midface and oronasal facial regions, and to a lesser extent the upper and lower facial skeletons. The pattern of strong morphological differences in the oronasal region combined with differences across the facial complex suggests that craniofacial bones are integrated and covary, despite influences from the congenital cleft.
Three-dimensional craniofacial morphology in unilateral cleft lip and palate
Journal of Oral Science, 2014
Craniofacial morphology and asymmetry were compared before, during, and after puberty within and between patients with unilateral complete cleft lip and palate (UCCLP) and a non-cleft group. In the UCCLP group, the posterior cranial base and total cranial base were significantly shorter at all skeletal periods, the maxilla was significantly retruded and posteriorly rotated, and the mandible was significantly smaller and inferoposteriorly rotated. The angle between the nasal and mandibular plane and lower anterior facial height were significantly higher, and upper posterior facial height and total posterior height were significantly lower, in the UCCLP group. Except for an increase in the nasal cavity, no significant differences were detected in facial width. For all measurements, asymmetry on the horizontal plane was more significant than that on the vertical plane. Asymmetries in the UCCLP group were mostly detected during puberty. The UCCLP group had no distinctive mandibular asymmetry, as compared with the Class I group. In UCCLP patients, the cranial base, maxilla, and mandible were affected on the sagittal plane during all growth periods. However, horizontal asymmetries were mostly detected before and during puberty. Vertical asymmetries were less severe, and there was no distinctive mandibular asymmetry as compared with the Class I group.
Bilateral cleft lip and palate (BCLP) is caused by a lack of merging of maxillary and nasal facial prominences during development and morphogenesis. BCLP is associated with congenital defects of the oronasal facial region that can impair ingestion, mastication, speech, and dentofacial development. Using cone beam computed tomography (CBCT) images, 7-18 yr. old individuals born with BCLP (n = 15) and age- and sex-matched controls (n = 15) were retrospectively assessed. Coordinate values of three-dimensional facial skeletal anatomical landmarks (n = 32) were measured from each CBCT image. Data were evaluated using principal coordinates analysis (PCOORD) and Euclidean Distance Matrix Analysis (EDMA). PCOORD axes 1-3 explain approximately 45% of the morphological variation between samples, and specific patterns of morphological differences were associated with each axis. Approximately 30% of facial skeletal measures significantly differ by confidence interval testing (α = 0.10) between samples. While significant form differences occur across the facial skeleton, strong patterns of differences are localized to the lateral and superioinferior aspects of the nasal aperture. In conclusion, the BCLP deformity significantly alters facial skeletal morphology of the midface and oronasal regions of the face, but morphological differences were also found in the upper facial skeleton and to a lesser extent, the lower facial skeleton. This pattern of strong differences in the oronasal region of the facial skeleton combined with differences across the rest of the facial complex underscores the idea that bones of the craniofacial skeleton are integrated.
Cleft lip with or without cleft palate (CL/P) is a relatively common craniofacial malformation involving bony and soft-tissue disruptions of the nasolabial and dentoalveolar regions. The combination of CL/P and subsequent craniofacial surgeries to close the cleft and improve appearance of the cutaneous upper lip and nose can cause scarring and muscle pull, possibly resulting in soft-tissue depth asymmetries across the face. We tested the hypothesis that tissue depths in children with unilateral CL/P exhibit differences in symmetry across the sides of the face. Twenty-eight tissue depths were measured on cone-beam computed tomography images of children with unilateral CL/P (n = 55), aged 7 to 17 years, using Dolphin software (version 11.5). Significant differences in tissue depth symmetry were found around the cutaneous upper lip and nose in patients with unilateral CL/P.
American Journal of Orthodontics and Dentofacial Orthopedics, 2014
Introduction: In this study, we compared patterns of morphologic variations of the craniofacial skeleton between patients affected by clefts who were operated on and unaffected subjects, aiming to discuss possible morphofunctional consequences of treatment in craniofacial development. Methods: The lateral cephalograms of 76 subjects, comprising patients with operated unilateral cleft lip and palate (OpC) and a group matched for sex and age without cleft, were used. Thirteen landmarks were used as variables in geometric morphometric tests quantifying and describing overall shape variation, differences between group means, allometry, and upperlower face covariation. Results: The OpC group showed broader shape variations including noncleft group characteristics, but mainly a retrognathic maxilla, a vertically elongated face, a more open mandibular angle, and a more closed basicranial angle. Group means differed mainly in the maxillomandibular relationships. Allometry differed between groups, with the smallest OpC patients showing the most altered morphology. Upper and lower face covariation was stronger in the OpC group, showing mainly vertical changes in the anterior face. Conclusions: Operated patients affected by clefts achieve a broad range of morphologies; the most altered were found in those with skeletal Class III and small size. Furthermore, their strongest upper and lower face shape covariation suggests that a harmonic dental occlusion could be a key factor in achieving "normal" craniofacial morphology. (Am J Orthod Dentofacial Orthop 2014;146:346-54) C lefts of the lip and palate (CLP) are common congenital anomalies. The incidence is highly variable among populations. The highest incidence is found in Asians and Native Americans, with 1 in 500 live births, and the lowest in Africans, with 1 in 2500 births; white, Hispanic, and Latin populations (among these, Chileans) have intermediate incidence of 1 in 1000 live births. 1-4 Cleft etiology is multifactorial, comprising both genetic and environmental factors acting during intrauterine development. 5-7 Unilateral CLP (UCLP) is the most common cleft type. 5,8 Morphologic alterations of operated patients with UCLP have been widely reported in the literature, of which changes in maxillomandibular relationship are the most prevalent, as described below. Since unoperated subjects have the potential to develop a "normal" (ie, skeletal Class I) maxillomandibular relationship, 9,10 it has been proposed that altered maxillomandibular relationships are caused by the effect of surgery, particularly lip closure, on the developing bone and sutures. 11-13 This has led to the proposal of different protocols for the surgical management of CLP that concur in the importance of reconstructing the muscular anatomy of the lip and the soft palate after the third month of age. 12,14-16 Despite the differences in surgical approaches and treatment protocols, the morphologic features in operated patients with UCLP tend to be uniform. In general,
Longitudinal 3D Assessment of Facial Asymmetry in Unilateral Cleft Lip and Palate
The Cleft palate-craniofacial journal : official publication of the American Cleft Palate-Craniofacial Association, 2018
Longitudinal evaluation of asymmetry of the surgically managed unilateral cleft lip and palate (UCLP) to assess the impact of facial growth on facial appearance. Prospective study. Glasgow Dental Hospital and School, University of Glasgow, United Kingdom Patients: Fifteen UCLP infants. The 3-D facial images were captured before surgery, 4 months after surgery, and at 4-year follow-up using stereophotogrammetry. A generic mesh which is a mathematical facial mask that consists of thousands of points (vertices) was conformed on the generated 3-D images. Using Procustean analysis, an average facial mesh was obtained for each age-group. A mirror image of each average mesh was mathematically obtained for the analysis of facial dysmorphology. Facial asymmetry was assessed by measuring the distances between the corresponding vertices of the original and the mirror copy of the conformed meshes, and this was displayed in color-coded map. There was a clear improvement in the facial asymmetry f...
American Journal of Orthodontics and Dentofacial Orthopedics, 2008
Computerized tomography (CT) makes regional anatomy available as a series of crosssectional axial images that can be analyzed unobstructed by other anatomic features. In this study, we aimed to analyze the maxillary dentoalveolar and deeper structures in patients with surgically repaired complete unilateral cleft lip and palate (CLP) and to investigate whether significant asymmetry existed in the deeper midfacial structures, using axial CT. Methods: Axial CT scans of 15 children (7 boys, 8 girls; mean age, 11.59 Ϯ 1.34 years) with repaired complete unilateral CLP were acquired parallel to Reid's base line. Specific landmarks applicable for CT were defined and plotted to undertake a comprehensive Craniofacial CT Analysis of the midface. Twenty-six pairs of transverse and sagittal measurements made to a cranial base midsagittal construct and interspinosum fit line, respectively, were compared by using the paired t test. Results: Significant reduction of 19.05% in the nasal chamber width (2.54 Ϯ 1.49 mm; P Ͻ0.001), more distal position of the bony alar base (2.28 Ϯ 1.71 mm; P Ͻ0.001), and the sagittal position of the most prominent anterolateral point on the maxillary alveolar process (1.62 Ϯ 1.85 mm; P Ͻ0.01) were noted on the cleft side. The lower half of the bony nasal septum was consistently deviated toward the cleft side, and the nasal tip was deviated to the noncleft side in 60% of the subjects. The anterior nasal spine was deviated to the noncleft side in 73.3% of the subjects and a mean deviation of the anterior nasal spine of 2.84 Ϯ 3.90 mm toward the noncleft side was measured. Significant sagittal and transverse asymmetry in deeper midfacial regions was not observed. Unilateral dental-arch collapse was observed in 73.3% of the sample, bilateral collapse in 20%, and no collapse in 6.7%. The soft-tissue support of the alveolar margins of the cleft did not show significant differences between the cleft and noncleft sides. Conclusions: CT is an excellent method for quantifying surface and deep craniofacial structures. Most asymmetries and deformities in the 10-to 14-year-old patients with repaired complete unilateral CLP we studied were in the dentoalveolar area near the cleft and the nasal chamber and not in the deeper regions of the maxillary complex. (Am J Orthod Dentofacial Orthop 2008;134:418-29) b Professor and head,
Background. Stereophotogrammetry can be used to study facial morphology in both healthy individuals as well as subjects with orofacial clefts because it shows good reliability, ability to capture images rapidly, archival capabilities, and high resolution, and does not require ionizing radiation. This study aimed to compare the 3D facial morphology of infants born with unilateral cleft lip and palate (UCLP) with an age-matched normative 3D average face before and after primary closure of the lip and soft palate. Methods. Thirty infants with a non-syndromic complete unilateral cleft lip, alveolus, and palate participated in the study. 3D images were acquired at 3, 6, 9, and 12 months of age. All subjects were treated according to the primary surgical protocol consisting of surgical closure of the lip and the soft palate at 6 months of age. 3D images of UCLP patients at 3, 6 (pre-treatment), 9, and 12 months of age were superimposed on normative datasets of average facial morphology using the children's reference frame. Distance maps of the complete 3D facial surface and the nose, upper lip, chin, forehead, and cheek regions were developed. Results. Assessments of the facial morphology of UCLP and control subjects by using color-distance maps showed large differences in the upper lip region at the location of the cleft defect and an asymmetry at the nostrils at 3 and 6 months of age. At 9 months of age, the labial symmetry was completely restored although the tip of the nose towards the unaffected side showed some remnant asymmetry. At 12 months of age, the symmetry of the nose improved, with only some remnant asymmetry noted on both sides of the nasal tip. At all ages, the mandibular and chin regions of the UCLP patients were 2.5-5 mm posterior to those in the average controls. Conclusion. In patients with UCLP deviations from the normative average 3D facial morphology of agematched control subjects existed for the upper lip, nose, and even the forehead before lip and soft palate closure was performed. Compared to the controls symmetry in the upper lip was restored, and the shape of the upper lip showed less variation after primary lip and soft palate closure. At this early age, retrusion of the soft-tissue mandible and chin, however, seems to be developing already.
Background. Stereophotogrammetry can be used to study facial morphology in both healthy individuals as well as subjects with orofacial clefts because it shows good reliability, ability to capture images rapidly, archival capabilities, and high resolution, and does not require ionizing radiation. This study aimed to compare the 3D facial morphology of infants born with unilateral cleft lip and palate (UCLP) with an age-matched normative 3D average face before and after primary closure of the lip and soft palate. Methods. Thirty infants with a non-syndromic complete unilateral cleft lip, alveolus, and palate participated in the study. 3D images were acquired at 3, 6, 9, and 12 months of age. All subjects were treated according to the primary surgical protocol consisting of surgical closure of the lip and the soft palate at 6 months of age. 3D images of UCLP patients at 3, 6 (pre-treatment), 9, and 12 months of age were superimposed on normative datasets of average facial morphology using the children's reference frame. Distance maps of the complete 3D facial surface and the nose, upper lip, chin, forehead, and cheek regions were developed. Results. Assessments of the facial morphology of UCLP and control subjects by using color-distance maps showed large differences in the upper lip region at the location of the cleft defect and an asymmetry at the nostrils at 3 and 6 months of age. At 9 months of age, the labial symmetry was completely restored although the tip of the nose towards the unaffected side showed some remnant asymmetry. At 12 months of age, the symmetry of the nose improved, with only some remnant asymmetry noted on both sides of the nasal tip. At all ages, the mandibular and chin regions of the UCLP patients were 2.5-5 mm posterior to those in the average controls. Conclusion. In patients with UCLP deviations from the normative average 3D facial morphology of agematched control subjects existed for the upper lip, nose, and even the forehead before lip and soft palate closure was performed. Compared to the controls symmetry in the upper lip was restored, and the shape of the upper lip showed less variation after primary lip and soft palate closure. At this early age, retrusion of the soft-tissue mandible and chin, however, seems to be developing already.