Dental Subphenotypes in Infants With Orofacial Clefts-A Longitudinal Population-Based Retrospective Radiographic Study of the Primary and Secondary Dentitions.

OBJECTIVES: To determine the developmental patterns of primary and secondary dentitions in infants with orofacial clefts. DESIGN: Retrospective, longitudinal, population-based cohort study. MATERIALS: Longitudinal records and radiographs of 192 nonsyndromic Northern European infants with isolated unilateral cleft lip (UCL, n = 111) and isolated cleft palate (CP, n = 81). METHODS: Radiographic assessments of primary and secondary dentition anomalies and dental maturation, by gender and cleft severity for comparisons between the groups and with historical controls. RESULTS: In infants with UCL, the frequencies of dental anomalies were high in both primary (38.7%) and secondary (18.0%) dentitions. Primary and secondary dentition anomalies were not observed in infants with CP and different in the UCL group (P = .003). Risk differences involved primary supernumerary teeth (P = .0001) and talon cusp formation (P = .0001), and secondary tooth agenesis (P = .001) of the maxillary lateral incisor on the side of the cleft lip. Delayed primary and secondary dental maturation occurred in the UCL and CP groups, greater in infants with UCL (P < .0001). Primary and secondary dental maturation featured sexual dimorphism with greater delay in males (UCL, P < .0001; CP, .0001 > P = .001). The effect of cleft severity on dental maturation was significant in infants with UCL (P = .0361) and CP (P = .0175) in the primary but not in the secondary dentition. CONCLUSIONS: There were different dental anomalies in the primary and secondary dentitions in operated infants with UCL and no dental anomalies in unoperated infants with CP. Dental maturation was delayed in infants with UCL and CP with greater delay in males compared to females.

Facial Asymmetry in Nonsyndromic and Muenke Syndrome-Associated Unicoronal Synostosis: A 3-Dimensional Study Based on Facial Surfaces Extracted From CT Scans.

OBJECTIVE: To quantify soft tissue facial asymmetry (FA) in children with nonsyndromic and Muenke syndrome-associated unicoronal synostosis (NS-UCS and MS-UCS), hypothesizing that MS-UCS presents with significantly larger FA than NS-UCS. DESIGN: Retrospective cohort study. PATIENTS AND METHODS: Twenty-one children (mean age: 0.6 years; range: 0.1-1.4 years) were included in the study (NS-UCS = 14; MS-UCS = 7). From presurgical computed tomography scans, facial surfaces were constructed for analysis. A landmark guided atlas was deformed to match each patient's surface, obtaining spatially detailed left-right point correspondence. Facial asymmetry was calculated in each surface point across the face, as the length (mm) of an asymmetry vector, with its Cartesian components providing 3 directions. Mean FA was calculated for the full face, and the forehead, eye, nose, cheek, mouth, and chin regions. RESULTS: For the full face, a significant difference of 2.4 mm (P = .001) was calculated between the 2 groups, predominately in the transverse direction (1.5 mm; P < .001). The forehead and chin regions presented with the largest significant difference, 3.5 mm (P = .002) and 3.2 mm (P < .001), respectively; followed by the eye (2.4 mm; P = .004), cheek (2.2 mm; P = .004), nose (1.7 mm; P = .001), and mouth (1.4 mm; P = .009) regions. The transverse direction presented with the largest significant difference in the forehead, chin, mouth, and nose regions, the sagittal direction in the cheek region, and the vertical direction in the eye region. CONCLUSIONS: Muenke syndrome-associated unicoronal synostosis presented with significantly larger FA in all regions compared to NS-UCS. The largest significant differences were found in the forehead and chin regions, predominantly in the transverse direction.

Delayed maturation and reduced crown width of the permanent first mandibular molar in all subgroups of cleft lip and palate.

OBJECTIVES: Surgical closure of the cleft damages nerves and blood supply in the area and may possibly negatively influence dental development in children with cleft lip and palate (CLP). Previous studies of the permanent first mandibular molar (M1inf ) in children with unilateral complete CLP found delayed maturation and decreased follicle/crown width before any surgical interventions. This study aimed to investigate if these findings are representative for the total population of individuals born with CLP. SETTING AND SAMPLE POPULATION: A population-based consecutive longitudinal sample of 224 children with clefts: 183 with CLP (66 with complete [47 unilateral, 19 bilateral], 117 with incomplete [70 unilateral, 47 bilateral], and 41 with unilateral incomplete cleft lip (controls), was examined pre- and post-lip closure (at 2 and 22 months of age). MATERIAL AND METHODS: In lateral cephalometric X-rays (obtained as part of a standard treatment protocol), M1inf follicle maturation and follicle/crown width were assessed. Mantel-Haenszel test and Pearson's correlation coefficient R were used to describe relationships. Differences between group means were tested using Wilcoxon rank sum test. Significance level was set to 5%. RESULTS: Follicle maturation was significantly delayed in all groups with CLP compared to that in controls (P < .01). Follicle and crown width were significantly reduced in all CLP groups compared with those in controls (P < .01). Sex did not influence these variables. CONCLUSION: Delayed follicle maturation and decreased follicle/crown/tooth size were found to be part of the congenital traits characterizing individuals with all types of CLP and not a result of surgical iatrogenesis.

Three-dimensional assessment of facial morphology in children and adolescents with juvenile idiopathic arthritis and moderate to severe TMJ involvement using 3D surface scans.

OBJECTIVE: This study aims to (1) assess the facial morphology in juvenile idiopathic arthritis (JIA) subjects with moderate to severe temporomandibular joint (TMJ) involvement using 3D surface scans and (2) compare the facial morphology in these subjects to that in JIA subjects without TMJ involvement. METHODS: Sixty JIA subjects were included and grouped as follows: group 1 (no involvement group), JIA without TMJ involvement; Group 2 (unilateral group), JIA with moderate to severe unilateral TMJ involvement; and group 3 (bilateral group), JIA with bilateral TMJ involvement. Standard orientation of all surfaces was accomplished. The means and variabilities of facial morphology in groups 2 and 3 were assessed and compared with those of group 1 in three dimensions, respectively. RESULTS: Group 2 (unilateral group) exhibited a more retruded and wider chin, shorter mandibular height, and more prominent cheek (2, 2, 5, and 2 mm, on average, respectively) on the affected side and a more retruded and narrower chin and more prominent malar region (4, 3, and 2 mm, on average, respectively) on the unaffected side compared with group 1 (no involvement group) (p < 0.05). Group 3 (bilateral group) exhibited a more retruded chin, shorter mandibular height, more prominent upper cheeks, and narrower perioral region (5, 5, 3, and 2 mm, respectively) compared with group 1 (no involvement group) (p < 0.05). CONCLUSIONS: In JIA subjects with moderate to severe unilateral or bilateral TMJ involvement, the affected side(s) revealed similar facial dysmorphology with reduced mandibular height, chin retrusion, and prominent upper cheek. CLINICAL RELEVANCE: Three-dimensional surface scans can be a non-ionizing indicator of signs of TMJ involvement in JIA subjects.

Spatially Detailed 3D Quantification of Improved Facial Symmetry After Surgery in Children With Unicoronal Synostosis.

OBJECTIVE: To assess improvement of soft-tissue facial symmetry in children surgically treated for unicoronal synostosis (UCS) in infancy, to correlate pre- and postsurgical facial asymmetry and to evaluate whether the improvement was visually recognizable. DESIGN: Case-controlled follow-up. PATIENTS/SETTINGS: Eleven Danish children diagnosed with UCS were included, 3 of whom had tested positive for Muenke mutation. Preoperative computed tomography scans and postoperative 3dMD surfaces were available for measurements. A control group of healthy children matched for age and sex was employed. MAIN OUTCOME MEASURES: Pre- and postsurgical facial asymmetry was analyzed using a computerized method capable of objective and spatially detailed quantification in 3-dimension (transverse, vertical, and sagittal directions). Asymmetry was evaluated in the facial region and 6 subregions (forehead, mouth, eyes, nose, cheek, and chin). RESULTS: The largest significant improvement was seen in the sagittal direction of the facial (1.9 mm), forehead (2.0 mm), and cheek (3.4 mm) regions. Small but significant improvements were also seen in the mouth, chin, and eye regions. No significant improvement was seen in the nose region. Significant correlations were found between the pre- and postsurgically calculated facial asymmetry and between calculated asymmetry and clinical validation scores. CONCLUSIONS: All patients presented with improved facial symmetry after surgery and the improvements were visually recognizable. However, only 1 (9.1%) of the 11 patients reached a level of facial asymmetry as low as that seen in the control group. The best outcome was, in general, seen in cases with mild facial asymmetry presurgically.

Parental age in relation to the severity of cleft lip and/or palate.

OBJECTIVE: To investigate parental age in relation to the severity of cleft diagnosis in a population-based consecutive sample of individuals with clefts. SETTING/SAMPLE: Retrospective, consecutive. MATERIAL/METHODS: The sample comprised 545 consecutive cases with nonsyndromic clefts (437 individuals with cleft lip with/without cleft palate [CL ± P] and 106 individuals with isolated cleft palate [CP]) and parental ages recorded. The groups were subdivided according to the extent/severity of cleft. Unilateral clefts were divided according to left/right sidedness. Logistic regression was used to analyse the association between parental age and risk of severe cleft separately for CL ± P and CP, as well as between parental age and risk of right-sided cleft. RESULTS: In CL ± P, the risk of a complete cleft in the offspring increases with maternal age when the paternal age exceeds approximately 29 years. Moreover, the risk is higher when both parents are old than when both parents are young. In CP, no statistically significant results were identified. However, there were clear trends that indicated a similar pattern as that for CL ± P. No association was identified between increased parental age and the sidedness of clefts. CONCLUSIONS: Parental age seems to contribute to cleft severity, as older parents showed a higher risk of having offspring with a complete cleft than younger parents.

Facial Asymmetry in Children with Unicoronal Synostosis Who Have Undergone Craniofacial Reconstruction in Infancy.

OBJECTIVE: Quantitatively assess 3D spatially detailed soft-tissue facial asymmetry in children who had undergone craniofacial reconstruction for Unicoronal Synostosis (UCS), and compare the facial asymmetry to control patients. It was hypothesized that there would be no significant differences in the facial asymmetry between the groups. DESIGN: Clinical, retrospective follow-up study. Methodological study. SETTING: Primary care center. PATIENTS/PARTICIPANTS: Twenty-two children with UCS were selected after review of records. INCLUSION CRITERIA: isolated UCS; surgically treated for UCS within the first 19 months of life, without secondary reconstruction; and DNA analysis for the Muenke mutation. An age- and sex-matched control group was employed. INTERVENTIONS: The UCS group had undergone bilateral craniotomy of the frontal bone with unilateral supraorbital rim advancement. MAIN OUTCOME MEASURE(S): Using 3D surface scanning, a detailed map of 3D asymmetry presenting the amount of asymmetry in the sagittal, vertical, and transverse directions was calculated for six facial subregions. RESULTS: The facial asymmetry in the UCS group was significantly larger than in the control group for all regions, to the largest extent in the sagittal direction (level of significance: 5%). The regions with the most pronounced asymmetry were cheeks (mean: 5.45 mm; SD: 1.83 mm), forehead (mean: 5.00 mm; SD: 1.57 mm), and eyes (mean: 4.26 mm; SD: 1.44 mm). CONCLUSIONS: Ninety percent of the UCS patients in the study had significant facial asymmetry throughout the facial area. The study demonstrates a methodology of facial asymmetry quantification well suited for soft-tissue surgical outcome evaluations and long-term follow-up studies in patients with craniofacial anomalies.

Facial morphology in children and adolescents with juvenile idiopathic arthritis and moderate to severe temporomandibular joint involvement.

INTRODUCTION: The aims of this study were to (1) assess lateral facial morphology in children and adolescents with juvenile idiopathic arthritis and moderate to severe temporomandibular joint (TMJ) involvement, (2) compare the lateral facial morphology of these subjects with and without TMJ involvement using cephalograms and 3-dimensional (3D) facial photographs, and (3) compare and correlate the results of the 3D photographic and cephalometric analyses. METHODS: Sixty patients with juvenile idiopathic arthritis were included and grouped as follows: group 1, juvenile idiopathic arthritis patients without TMJ involvement; group 2, juvenile idiopathic arthritis patients with moderate to severe unilateral TMJ involvement; and group 3, juvenile idiopathic arthritis patients with moderate to severe bilateral TMJ involvement. Lateral cephalograms were used to assess and compare lateral facial morphologies between the groups. Lateral projections of oriented 3D photographs were superimposed on the lateral cephalograms. The results of the lateral 3D photographic analysis were correlated with those of lateral cephalometric analysis. RESULTS: Group 3 showed the most severe growth disturbances, including more retrognathic mandible and retruded chin, steep occlusal and mandibular planes, and more hyperdivergent type (P <0.01). Group 2 showed similar growth disturbances, but to a lesser extent than did group 3. Photographic variables were significantly correlated with the soft tissue and skeletal variables of cephalograms (0.5 < r < 0.9; P <0.001). CONCLUSIONS: Subjects with juvenile idiopathic arthritis and unilateral or bilateral moderate to severe TMJ involvement had significant growth disturbances. Early intervention is recommended for these patients to prevent unfavorable facial development. Furthermore, with proper orientation, 3D photographs can be used as an alternative to conventional lateral cephalograms and 2-dimensional photographs.

Clinical and digital assessment of tooth wear.

The aim of this study was to compare the assessment of tooth wear performed on digital models with the one conducted at the clinical examination. Seventy-eight volunteers (29 males and 49 females, age range 20-30 years) with at least 24 teeth, normal oral function, and a neutral transverse relationship were examined. During the clinical examination, dental wear was registered according to the Basic Erosive Wear Examination (BEWE) index. Subsequently, the BEWE index was blindly applied by two examiners on digital models obtained from the volunteers. Data were analyzed using weighted Cohen's kappa coefficient and correlation tests with a confidence interval of 95%. All volunteers showed signs of tooth wear. Anterior teeth showed increased severity of tooth wear than first molars. Early loss of tooth substance could be identified on the digital models, including in areas with challenging direct intraoral visual access. Approximately 50% of the scores based on clinical examination agreed with those based on examination of digital models (k = 0.543, p < 0.01). A moderate, positive correlation was observed between scores registered clinically and on digital models (Spearman's rho = 0.560, p < 0.001). Considering the rather low agreement between the clinical and digital scores, alternatives to using BEWE on digital models are needed.

Early postnatal development of the mandibular permanent first molar in infants with isolated cleft palate.

BACKGROUND: Based on measurements on dental casts, smaller permanent teeth in children with cleft palate have previously been reported in the literature; however, the early maturation of teeth and the size of the follicles and crowns have not been investigated. HYPOTHESIS: The maturation of the mandibular permanent first molar (M1(inf)) is delayed, and the mesiodistal diameters of the follicle and crown of M1(inf), respectively, are reduced in children with isolated cleft palate (ICP). DESIGN: Retrospective, longitudinal. Cephalometric X-rays were available for 2 and 22 months old children with clefts (64 children with ICP, and a control group of 38 children with unilateral incomplete cleft lip). The width of the follicle and the crown of M1(inf), and the maturation of M1(inf) were assessed. Intra-observer error was acceptable. RESULTS: M1(inf) maturation was delayed in children with ICP at both 2 and 22 months of age. The mesiodistal diameter of the crown of M1(inf) in the ICP group was reduced. Thus, the two hypotheses could not be refuted. CONCLUSIONS: Children with ICP showed smaller dimensions of the M1(inf) , and in addition, the maturation of M1(inf) was delayed.

3D analysis of facial asymmetry in subjects with juvenile idiopathic arthritis.

OBJECTIVE: To investigate and compare facial asymmetry in subjects with JIA with unilateral, bilateral or no TM joint (TMJ) involvement. METHODS: Eighty-one subjects with JIA: 22 with unilateral TMJ involvement (Group 1), 15 with bilateral TMJ involvement (Group 2) and 44 with no TMJ involvement (Group 3). Panoramic X-rays and three-dimensional (3D) photographs (surface scans) were obtained for all subjects. Panoramic X-rays were rated for severity of TMJ involvement. For each individual, a spatially detailed facial asymmetry map was created from the 3D photograph. Mean and variability of asymmetry were calculated for each of the three groups and compared. RESULTS: Distinct patterns of asymmetry were found in Groups 1 and 2. With mean asymmetry values up to 3.5 mm, Group 1 exhibited a significantly greater amount of asymmetry in a broad band along the lower jaw extending from the region of the condyle to the chin than Group 2. The mean facial asymmetry (1 S.D.) for each JIA group was 2.3 (0.9), 2.0 (0.7), 1.7 (0.5) mm for Groups 1, 2 and 3, respectively. CONCLUSION: JIA subjects with TMJ involvement displayed patterns of facial asymmetry consistent with the destruction of the condylar growth centre, leading to mandibular asymmetry with displacement of the bony chin. Facial asymmetry quantification was found to be an effective method for assessing both the amount and the localization and spatial extent of asymmetry in all 3Ds.

Mandibular dimensions and growth in 11- to 26-week-old Danish fetuses studied by 3D ultrasound.

OBJECTIVE: To present normative data on prenatal mandibular morphology and growth. MATERIAL AND METHODS: Fifty-four normal fetuses (Danish Caucasian) were included in the study (gestational age: 11-26 weeks). Fetuses were scanned using a GE Voluson 730 Expert 3D scanner. Scans were visualized and analyzed using GE 4DVIEW software. Mandibular dimensions [base length (B), ramus height (H), and total length (L)] and the mandibular angle (phi) were measured, and the mandibular index was calculated. Method error was estimated by duplicate measurements. Growth was calculated by regressing measured variables on age. RESULTS: Mandibular measurements were found to be reliable [precision: 0.5-1.2 mm (1SD)]. Mean mandibular variable values at week 11: B = 5.2 +/- 2.5 mm; H = 2.7 +/- 1.3 mm; L = 7.7 +/- 3.2 mm; phi = 149 +/- 6.0 degrees; and at week 26: B = 22.6 +/- 2.5 mm; H = 12.3 +/- 1.3 mm; L = 33.1 +/- 3.2 mm; phi = 135 +/- 6.0 degrees. A linear model described growth (B(g)/H(g)/L(g)/phi(g)) giving B(g) = 1.2; H(g) = 0.64; L = 1.7 mm/week; phi(g) = - 0.9 degrees/week. CONCLUSION: Normative 3D data values for the human mandible in 11- to 26-week-old fetuses were presented. All measured mandibular parameters could be described using a linear increasing model from 11 to 26 weeks. All linear dimensions increased in size, while the mandibular angle decreased steadily during the observation period.

Deep orofacial phenotyping of population-based infants with isolated cleft lip and isolated cleft palate.

Isolated orofacial clefts (OFC) are common with poorly understood aetiology. Heterogeneous phenotypes and subphenotypes confound aetiological variant findings. To improve OFC phenome understanding, population-based, consecutive, pre-treatment infants with isolated unilateral cleft lip (UCL, n = 183) and isolated cleft palate (CP, n = 83) of similar ancestry were grouped for deep phenotyping. Subphenotypes stratified by gender and cleft severity were evaluated for primary dental malformations and maturation using radiographs. We found that cleft severity and tooth agenesis were inadequate to distinguish heterogeneity in infants with UCL and CP. Both groups featured slow dental maturity, significantly slower in males and the UCL phenotype. In 32.8% of infants with UCL, supernumerary maxillary lateral incisors were present on the cleft lip side, but not in infants with CP, suggesting a cleft dental epithelium and forme fruste cleft dentoalveolus of the UCL subphenotype. The findings underscored the importance of deep phenotyping to disclose occult OFC subphenotypes.

Phenotypic variability in Muenke syndrome-observations from five Danish families.

Muenke syndrome is a craniosynostosis syndrome associated with the p.Pro250Arg mutation in FGFR3. An increasing number of individuals with this mutation are reported to not have craniosynostosis. The purpose of this report is to increase awareness of the high phenotypic variability seen in Muenke syndrome. DNA testing for the p.Pro250Arg mutation is routinely performed in Denmark, in children presenting with isolated coronal synostosis. Verified diagnosis entails detailed family history, drawing of family pedigree, DNA testing of the parents, genetic counseling, skull radiographs, clinical photographs, and follow-up. Sixteen individuals from 5 Danish families with Muenke syndrome are presented. Large phenotypic variation was seen both within and across families. The most striking observations were that 6/16 (38%) cases did not have craniosynostosis and one individual presented with a normal phenotype. In addition, 3 unrelated cases had incomplete cleft palate, submucous cleft palate, and bifid uvula, respectively. There is strong evidence for reduced penetrance of the craniosynostosis trait in Muenke syndrome. We argue that many studies on Muenke syndrome have been influenced by ascertainment bias in regard to craniosynostosis. In addition, it is suggested that oral clefting might be part of the clinical spectrum seen in Muenke syndrome.

Quantifying Normal Craniofacial Form and Baseline Craniofacial Asymmetry in the Pediatric Population.

BACKGROUND: Restoring craniofacial symmetry is an important objective in the treatment of many craniofacial conditions. Normal form has been measured using anthropometry, cephalometry, and photography, yet all of these modalities have drawbacks. In this study, the authors define normal pediatric craniofacial form and craniofacial asymmetry using stereophotogrammetric images, which capture a densely sampled set of points on the form. METHODS: After institutional review board approval, normal, healthy children (n = 533) with no known craniofacial abnormalities were recruited at well-child visits to undergo full head stereophotogrammetric imaging. The children's ages ranged from 0 to 18 years. A symmetric three-dimensional template was registered and scaled to each individual scan using 25 manually placed landmarks. The template was deformed to each subject's three-dimensional scan using a thin-plate spline algorithm and closest point matching. Age-based normal facial models were derived. Mean facial asymmetry and statistical characteristics of the population were calculated. RESULTS: The mean head asymmetry across all pediatric subjects was 1.5 ± 0.5 mm (range, 0.46 to 4.78 mm), and the mean facial asymmetry was 1.2 ± 0.6 mm (range, 0.4 to 5.4 mm). There were no significant differences in the mean head or facial asymmetry with age, sex, or race. CONCLUSIONS: Understanding the "normal" form and baseline distribution of asymmetry is an important anthropomorphic foundation. The authors present a method to quantify normal craniofacial form and baseline asymmetry in a large pediatric sample. The authors found that the normal pediatric craniofacial form is asymmetric, and does not change in magnitude with age, sex, or race.

Prenatal diagnosis of autosomal recessive Robinow syndrome using 3D ultrasound.

This article hypothesizes that it is possible to detect and diagnose both the autosomal recessive and dominant forms prenatally using ultrasound. By focusing on the characteristic phenotypical presentation, the examinator is able to diagnose the syndrome prenatally, which is of clinical importance to the parents and counseling for the consideration of terminating the pregnancy.

Sagittal synostosis: I. Preoperative morphology of the skull.

The aim of this study was to characterise the preoperative morphology of the skull in sagittal synostosis in an objective and quantified way. The shapes of the skulls of 105 patients with isolated premature synostosis of the sagittal suture (SS group) were studied and compared with those of a control group of 72 children with unilateral incomplete cleft lip (UICL). A standardised radiocephalometric technique was used to obtain the images. A modification of a method developed by Kreiborg was used to analyse the radiocephalograms, which included the digitisation of 88 landmarks in the calvaria, skull base, and orbit (42 in the lateral and 46 in the frontal projections), the production of plots of mean shape for each group, and the intergroup comparison of a series of 81 variables (linear distance between selected landmarks, and angles defined by groups of three landmarks). Data from a subgroup of 66 patients aged 5 to 8 months were further compared to age-matched normative data in terms of seven angular and linear calvarial, cranial base and orbital variables. In a comparative analysis of the mean lateral plots, the foreheads of the study group (SS) had a more pronounced anterior slope and were also more convex. The vertex area was located more anteriorly, and was less convex. The occipital curvature was more prominent. Analysis of the mean frontal plots revealed a lack in convexity and lateral projection of the upper parietal regions, as well as a lower location of the line of maximum skull width. Comparison of the mean values of an SS subgroup to age-matched normative data showed a longer (p<0.001) and narrower skull (p<0.001) and a greater interorbital distance (p<0.001). The cranial base angle, the sella to nasion, and sella to basion lengths did not differ significantly. Sagittal synostosis is characterised by an extensive deformity of the cranial vault, with an essentially normal cranial base. The widened interorbital distance is probably related to compensatory metopic hyperactivity.

Sagittal synostosis: II. Cranial morphology and growth after the modified pi-plasty.

The aim of this study was to characterise the postoperative cranial growth and morphology after a modified pi-plasty for sagittal synostosis. The shape of the skull of 82 patients with isolated premature synostosis of the sagittal suture (SS group) operated on with a modified pi-plasty was studied longitudinally. Forty-five children with unilateral incomplete cleft lip (UICL), evaluated longitudinally at the ages of 2.4 and 23.2 months were used as controls. A standardised radiocephalometric technique was used for image acquisition. The radiocephalograms were analysed using a modification of a method developed by Kreiborg, which included the digitisation of 89 landmarks of the calvaria, cranial base, and orbit (43 in the lateral and 46 in the frontal projections), the production of mean shape plots for each group, and the intergroup comparison of a series of 78 variables (linear distance between selected landmarks, and angles defined by groups of three landmarks). Paired and unpaired t tests were used to assess the differences between the variables studied. These were accepted as significant for values of p<0.01 and were presented as coloured segments or areas in the respective plots. In a comparative analysis with the mean UICL lateral plots, the mean preoperative lateral plots of the study group (SS) showed that the anterior slope of the forehead was more pronounced and it was also more convex. The vertex area was located more anteriorly and was less convex. The occipital curvature was more prominent. Comparison of the mean frontal plots showed a deficiency in convexity and lateral projection of the upper parietal regions, and the line of maximum skull width was lower. The postoperative mean lateral plots of the study group showed a correction of the exaggerated anterior inclination of the forehead and a reduction of the abnormal occipital convexity. However, there was little change in the vertex region and it remained flatter than in the control group. In the mean frontal plots, the increase in convexity and in the lateral projection of the upper parietal areas led to a shape that was similar to that of the UICL group. The mean (SD) cephalic index changed from 64.9% (1.8%) to 71.4% (3.5%) (p<0.001). The longitudinal comparison between the mean postoperative plots at 3 and 5 years of age showed that there had been little change in cranial shape. In conclusion, after a modified pi-plasty for sagittal synostosis, significant objective changes in cranial shape towards normality were produced. The postoperative profile cranial shape was improved except in the vertex area, which remained flatter than normal. In the frontal projection an almost normal shape was obtained. The postoperative cranial shape obtained at 3 years of age had remained stable at the age of 5 years.

Helmet versus active repositioning for plagiocephaly: a three-dimensional analysis.

BACKGROUND AND PURPOSE: Orthotic helmets and active repositioning are the most common treatments for deformational plagiocephaly (DP). Existing evidence is not sufficient to objectively inform decisions between these options. A three-dimensional (3D), whole-head asymmetry analysis was used to rigorously compare outcomes of these 2 treatment methods. PATIENTS AND METHODS: Whole-head 3D surface scans of 70 infants with DP were captured before and after treatment by using stereophotogrammetric imaging technology. Helmeted (n=35) and nonhelmeted/actively repositioned (n=35) infants were matched for severity of initial deformity. Surfaces were spatially registered to a symmetric template, which was deformed to achieve detailed right-to-left point correspondence for every point on the head surface. A ratiometric asymmetry value was calculated for each point relative to its contralateral counterpart. Maximum and mean asymmetry values were determined. Change in mean and maximum asymmetry with treatment was the basis for group comparison. RESULTS: The helmeted group had a larger reduction than the repositioned group in both maximum (4.0% vs 2.5%; P=.02) and mean asymmetry (0.9% vs 0.5%; P=.02). The greatest difference was localized to the occipital region. CONCLUSIONS: Whole-head 3D asymmetry analysis is capable of rigorously quantifying the relative efficacy of the 2 common treatments of DP. Orthotic helmets provide statistically superior improvement in head symmetry compared with active repositioning immediately after therapy. Additional studies are needed to (1) establish the clinical significance of these quantitative differences in outcome, (2) define what constitutes pathologic head asymmetry, and (3) determine whether superiority of orthotic treatment lasts as the child matures.