ORIGINAL ARTICLE

M. T. Abeleira M. Outumuro M. Diniz J. Limeres I. Ramos P. Diz

Morphometry of the hard palate in Down’s syndrome through CBCTimage analysis

Authors' affiliations: M. T. Abeleira, M. Outumuro, M. Diniz, J. Limeres, I. Ramos, P. Diz, OMEQUI Research Group, School of Medicine and Dentistry, Santiago de Compostela University, Santiago de Compostela, Spain

Abeleira M. T., Outumuro M., Diniz M., Limeres J., Ramos I., Diz P.

Correspondence to: P. D. Diz Departamento de Estomatologıa Facultad de Medicina y Odontologıa Universidad de Santiago de Compostela c/ Entrerrıos sn, 15782 Santiago de Compostela Spain E-mail: [email protected]

Morphometry of the hard palate in Down’s syndrome through CBCTimage analysis Orthod Craniofac Res 2015; 18: 212–220. © 2015 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd Structured Abstract Objectives – To define the morphometry of the hard palate in Down’s syndrome (DS) on cone beam computed tomography (CBCT) images. Setting and Sample Population – Santiago de Compostela University (Spain). The study group included 40 white DS individuals aged 10 to 40 years (mean = 18.8  7.3 years), 25 males and 15 females. The control group consisted of 40 individuals matched for age and sex were selected. Material & Methods – Nine measurements were taken on the CBCT images. Axial plane: anteroposterior length (aAPL) and arch length (aARL); sagittal plane: anteroposterior length (sAPL), maximum height (sMH) and sagittal arch (sAR); coronal plane: interdental width (cIDW), height (cHE), skeletal width (cSW) and coronal arch (cAR). Results – aAPL, aARL, sAPL, sMH, sAR, cMH and cAR were comparable in the two groups. cIDW and cSW were greater in controls than in DS. We found no statistically significant differences between males and females with DS. In the controls, sAPL and sAR were greater in males than females. In DS, age only had a statistically significantly increasing effect on aAPL and sAPL. In the controls, age significantly affected sAR and cHE. Conclusion – The hard palate is narrower in DS than in controls, but the anteroposterior measurements and the height of the vault are comparable in both groups.

Date: Accepted 6 April 2015 DOI: 10.1111/ocr.12097 © 2015 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd

Key words: anatomy; cone syndrome; hard palate

beam

computed

tomography;

Down’s

Abeleira et al. Hard palate morphometry in Down’s syndrome

Introduction Nearly ever since the first descriptions of Down’s syndrome (DS) reported in the literature, the morphology of the palate has been described as part of the characteristic phenotype. Oster (1) considered a narrow palate to be one of the 10 cardinal signs of the syndrome, and Benda (2) included the palate among the regions with greatest developmental delay in DS. Most reports published up to that time had been observational and, therefore, susceptible to criteria of subjectivity. One of the most important studies on palatal morphometry in DS was published by Shapiro et al. in 1967 (3) in the prestigious New England Journal of Medicine. Their most relevant conclusion was that, in DS, the anteroposterior axis of the palate was narrower and significantly shorter than in the general population. Based on this narrow morphology and the apparent greater height of the palate than normal, Benda (2) described a morphological pattern that he termed ‘steeple palate’. Four decades later, after qualitative analysis of the hard palate in individuals with DS, Carlstedt et al. (4) described marked variability in the morphological characteristics (flat, staired, round, pointed, high and narrow), with a predominance of staired palates. Skrinjaric et al. (5) also reported a high prevalence of palates with a staired or shelf-like morphology, particularly among younger individuals with DS. Anecdotally, Shapiro et al. (6) designed an instrument to perform in vivo measurements directly on the palatal surface, although there were obviously some major difficulties with its use, such as the extent of collaboration of the individual and a certain degree of subjectivity of the examiner in the location of the anatomical landmarks. As a result, this technique did not become popular for use in research, and direct measurement on plaster casts using calibrated callipers became the most widely used method in the literature for the analysis of palatal morphology (5, 7). The technique has been perfected over the years, with the incorporation of new recording instruments such as measurement

banks, the vernier calliper and digital callipers (8). In parallel, indirect analyses have been performed on two-dimensional projections, using photographs or radiographs (9, 10). These techniques have been shown to be valid for the study of cephalometric measurements or morphology of the dental arches, but they are very limited for the evaluation of palatal morphology (11). Digitization of the reference points avoids some of the difficulties of two-dimensional techniques, but it does not make up for the loss of the third dimension, which is essential to perform a full quantitative analysis of the palate. The incorporation of three-dimensional digitization has made it possible to record reference points as Cartesian coordinates directly on plaster casts (11, 12). Various types of instruments have been used to perform these measurements, including optical (13, 14), electromechanical (13) and electromagnetic (15); the electromagnetic instruments have the incorporated advantage of computerization (11, 15–18). In recent years, a number of studies have been published on measurement of dimensions of the hard palate using 3D-imaging techniques applied directly to the patient, such as computed tomography, cone beam computed tomography (CBCT) and magnetic resonance imaging (19– 21). Stereophotogrammetry and laser surface scanning have also been employed, but these techniques are mainly used for the measurement of facial surfaces (21, 22). CBCT imaging could be the preferred method for assessing palatal bone volume, as well as for surgical planning (21). The objective of the present study was to define the morphometry of the hard palate in individuals with DS using CBCT-image analysis in the three orthogonal anatomical planes.

Patients and methods All the images used in the study were obtained by an I-CATâ scanner (Imaging Sciences International, Hatfield, PA, USA) and were drawn from the historical archive of the Radiology Unit of the Faculty of Medicine and Dentistry of the

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University of Santiago de Compostela in Spain. All the imaging studies were performed by a single operator, following the manufacturer’s instructions and standardizing patient position. The studies selected were those in which the entire maxilla was visible. Images were obtained at 120 kVp and 5.0 mA; voxel size was 0.3 mm, with a field of view of 10–20 cm and an exposure time of 8.9 s. All images were reconstructed using the I-CAT VISIONâ software (Imaging Sciences International) and were exported in the DICOM (Digital Imaging Communication in Medicine) format to a MacBook 27 personal computer (Mac OsX 10.6; Apple Inc, Cupertino, CA, USA). All measurements were taken using the open-source OsiriX image processing software (Pixmeo, Geneva, Switzerland; www.osirixviewer.com). The only inclusion criterion was the availability of images of acceptable quality of the palatal vault in the three spatial orthogonal planes, and the only exclusion criterion was previous orthopaedic/orthodontic treatment or maxillofacial surgery affecting the jawbones. No pre-established exclusion criteria were applied regarding age, sex, comorbid disease or alterations of the orofacial region, which permitted the selection of a heterogeneous study group that could be considered to be more representative of the DS population than if, for example, we had only included individuals with significant malocclusions. The study group was formed of 40 dentate white individuals with DS with a mean age of 18.8  7.3 years (range, 10–40 years). Twentyfive were male, with a mean age of 18.6  6.2 years (range, 10–40 years), and 15 were female, with a mean age of 17.6  5.3 years (range, 10–29 years). The age distribution of the study group was the following: 10–15 years, 12 individuals; 16–20 years, 19 individuals; 21–25 years, four individuals; 26–30 years, one individual; 31–35 years, two individuals; and 36–40 years, two individuals. Using the same selection criteria applied to the study group, CBCT images of dentate persons without severe maxillofacial malformations, performed by the same operator, were selected 214 |

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from the historical archive of the Radiology Unit to form a control group of 40 individuals that was matched for age (mean age, 18.8  8.5 years; range, 10–40 years) and sex (25 males and 15 females) with the study group. Individuals with congenital syndromes that gave rise to orofacial alterations and individuals with past or present systemic diseases that could affect orofacial development or skeletal maturation directly or as a result of their treatment were excluded. In relation to the maxillary teeth, the permanent canines had still not erupted in nine individuals of the DS group and in seven individuals of the control group. No incisor or canine agenesis was detected in any individuals of either group; one maxillary pre-molar was absent in two DS individuals; one maxillary molar was absent in two DS and two control individuals; and two maxillary molars were absent in one DS individual. A specific informed consent was not required as all patients or, as appropriate, their legal representatives routinely authorize the use of images obtained in the Radiology Unit for training or research purposes. To perform the measurements, the CBCT images were orientated by aligning the hard palate with the x-axis (in the sagittal and coronal planes) and the medial palatine suture with the y-axis (in the axial and coronal planes). The cursor could then be moved along the z-axis of the plane under study (the coronal plane selected was the one that coincided with the mesial surface of the upper first molars in an axial view). Axial plane

The anteroposterior length (aAPL) was the segment between the posterior border of the nasopalatine duct and the transverse palatine suture (Fig. 1). The arch length (aARL) was measured by drawing a line from the mesial surface of the permanent right upper first molar to its contralateral counterpart, passing through the interproximal contact points of the other teeth in the arch (Fig. 1).

Abeleira et al. Hard palate morphometry in Down’s syndrome

A

B

Fig. 1. Dimensions of the hard palate in the axial plane: (A) anteroposterior length (aAPL) and (B) arch length (aARL).

Fig. 2. Dimensions of the hard palate in the sagittal plane: anteroposterior length (sAPL), maximum height (sHE) and sagittal arch (sAR).

Fig. 3. Dimensions of the hard palate in the coronal plane: interdental width (cIDW), height (cHE), skeletal width (cSW) and coronal arch (cAR).

Sagittal plane

sides (Fig. 3). The coronal arch (cAR) was measured by drawing a line across the palatal curvature between the points of dentoskeletal union of the permanent upper first molars. All the measurements of the analysed variables are expressed in centimetres. The statistical analysis of the results was performed using R software, version 2.12.0 (R Development Core Team, Vienna, Austria). Logistic regression models were employed to determine whether statistically significant differences existed between the morphometric variables of the palate in the study and control groups, describing the p value and the odds ratio (OR) with its 95% confidence interval. The same tool was used to evaluate whether differences existed due to sexual dimorphism. Linear regression models and a mixed additive model were employed to determine the influence of age after logarithmic transformation to achieve a normal distribution. Additive models are extensions of linear regression models and enable us to model the nonlinear effects of continuous covariates on a response of interest. The following packages, included in the R software, were used: ‘mgcv’ for fitting mixed additive models and ‘nlme’ for fitting linear models.

The anteroposterior length in the sagittal plane (sAPL) was measured by drawing a straight line from the prosthion to the transverse palatine suture (Fig. 2). A parallel line that passed through the highest point of the palatal vault was then drawn; the perpendicular distance between the two parallel lines gave the maximum height in the sagittal plane (sHE) (Fig. 2). The sagittal arch (sAR) was measured by drawing a line along the palatal curvature from the prosthion to the transverse palatine suture (Fig. 2). Coronal plane

The interdental width (cIDW) was defined as the shortest distance between the points of dentoskeletal union of the permanent upper first molars in the coronal plane (Fig. 3). Drawing a perpendicular line from the line used to measure the cIDW to the midline of the palate gave the coronal height (cHE) (Fig. 3). The skeletal width in the coronal plane (cSW) was taken as the distance between the most prominent points of the vestibular alveolar border on the left and right

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The measurements of the palatal variables were taken by two investigators. Reproducibility of the results was tested by repeating the measurements on 10 randomly selected cases 1 month after the first measurement. Intraclass coefficients of correlation between 0.89 and 0.99 were achieved in all the variables (a = 0.05; power = 80%).

Results Table 1 shows the mean values of the morphometric palatal variables obtained from individuals with DS and from the control group. Comparison of these measurements reveals sta-

Table 1. Palatal morphometric variables in individuals with Down’s syndrome and in non-syndromic controls* Statistical significance p value Odds ratio

Down’s syndrome

Controls

(95% confidence

mean  SD

mean  SD

interval)

Axial plane aAPL 2.645  0.240 2.551  0.269 ns aARL 6.301  0.525 6.461  0.523 ns Sagittal plane sAPL 3.549  0.299 3.542  0.530 ns sHE

0.544  0.127 0.524  0.121 ns

sAR

4.608  0.444 4.730  0.521 ns

Coronal plane cIDW 3.020  0.317 3.241  0.254 0.003 1.248 (1.078; 1.444) cHE

1.283  0.157 1.187  0.268 ns

cSW

5.010  0.369 5.536  0.326

Morphometry of the hard palate in Down's syndrome through CBCT-image analysis.

To define the morphometry of the hard palate in Down's syndrome (DS) on cone beam computed tomography (CBCT) images...
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