Craniofacial and upper airway cephalometrics in hypohidrotic ectodermal dysplasia

Arslan, Seher; Kama, Jalen Devecioğlu; Özer, Törün; Yavuz, İzzet

doi:10.1259/dmfr/28789331

Cited by 16 publications

(18 citation statements)

References 17 publications

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“…On the contrary, a more anteriorly positioned hyoid bone was seen in class III subjects when compared to class I [ 7 ]. Literature also supports more the posteriorly placed hyoid bone in class I horizontal patterns as compared to anteriorly placed in the class III pattern [ 8 ].…”

Section: Introductionmentioning

confidence: 98%

Position of the Hyoid Bone in Anteroposterior Skeletal Patterns

Bilal

2021

Journal of Healthcare Engineering

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Introduction. Hyoid bone plays a vital role in the craniofacial dynamics. From the maintenance of pharyngeal airway to phonation, deglutition, and mandibular movements, it has multifactorial roles to fulfill. The hyoid bone has a spatial relationship with cranial base, mandible, and cervical vertebrae. This study was performed to determine the position of the hyoid bone in different anteroposterior growth patterns. Materials and Methods. 75 lateral cephalograms belonging to all three sagittal skeletal patterns were evaluated in the study. The angular and linear measurements using 10 variables were made to determine the relationship of the hyoid bone with different craniofacial structures. SPSS was used to calculate the descriptive statistics, mean, and standard deviation of all the linear and angular variables. The analysis of variance (ANOVA) and post hoc test were used to compare the means of linear and angular measurement and multiple comparisons, respectively, at p < 0.05 significance level. Results. There was no significant difference in the mean angular measurements (NSH and MPH) between the 3 groups ( p > 0.05 ). Also, the multiple comparisons showed no significant difference between the linear measurements (H-FH, H-MP, H-H′, and H-N). However, there was a significant difference in three linear measurements: H-Rgn, RGN-C3, and C3-H. The values were more in class III subjects, compared to class I and class II. Conclusion. The linear millimetric distance between points H-RGN, RGN-C3, and C3-H which determine the relationship and spatial position of the hyoid bone with the mandible and third cervical vertebrae was statistically significant among groups. The values were more in class III subjects inferring the anterior position of the hyoid bone.

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Section: Introductionmentioning

confidence: 98%

Position of the Hyoid Bone in Anteroposterior Skeletal Patterns

Bilal

2021

Journal of Healthcare Engineering

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“…Craniofacial and anthropometric changes are also described in XLHED and consist in reduced and retrognathic maxilla, frontal prominence, cranial base modifications, reduced facial convexity and facial height with deficiencies in sagittal and transverse skeletal growth (Johnson et al, 2002; Arslan et al, 2007; Clauss et al, 2008). Global reduction of facial growth, especially in the middle third of the facial skeleton, and mandibular growth retardation are integrated in HED craniofacial phenotype (Dellavia et al, 2010).…”

Section: Introductionmentioning

confidence: 99%

Subtle Morphological Changes in the Mandible of Tabby Mice Revealed by Micro-CT Imaging and Elliptical Fourier Quantification

Bornert¹,

Choquet²,

Gros³

et al. 2011

Front. Physio.

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X-linked hypohidrotic ectodermal dysplasia (XLHED) is a genetic disorder due to a mutation of the EDA gene and is mainly characterized by an impaired formation of hair, teeth and sweat glands, and craniofacial dysmorphologies. Although tooth abnormalities in Tabby (Ta) mutant mice – the murine model of XLHED – have been extensively studied, characterization of the craniofacial complex, and more specifically the mandibular morphology has received less attention. From 3D micro-CT reconstructions of the left mandible, the mandibular outline observed in lateral view, was quantified using 2D elliptical Fourier analysis. Comparisons between Ta specimens and their wild-type controls were carried out showing significant shape differences between mouse strains enabling a clear distinction between hemizygous Ta specimens and the other mouse groups (WT and heterozygous EdaTa/+ specimens). Morphological differences associated with HED correspond not only to global mandibular features (restrained development of that bone along dorsoventral axis), but also to subtle aspects such as the marked backward projection of the coronoid process or the narrowing of the mandibular condylar neck. These modifications provide for the first time, evidence of a predominant effect of the Ta mutation on the mandibular morphology. These findings parallel the well described abnormalities of jugal tooth row and skeletal defects in Ta mice, and underline the role played by EDA-A in the reciprocal epithelial–mesenchymal interactions that are of critical importance in normal dental and craniofacial development.

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“…It may be inherited by autosomal dominant, autosomal recessive, or X-linked genetic transmission. 5,6 The most common form of ectopic dysplasia, HED, is usually inherited as an X-linked recessive genetic trait; in such cases the disorder is fully expressed in males only. However, females who carry a single copy of the mutant allele (heterozygote carriers) may exhibit some of the signs and findings associated with the disorder.…”

Section: Discussionmentioning

confidence: 99%

“…The characteristic craniofacial features of patients with HED are maxillary retrusion and deficiency in vertical, transversal, and sagittal growth of the jaw and soft tissues. These patients have smaller pharyngeal and upper airway dimensions 5 and the absence of sweat glands, resulting in very smooth, dry skin, and/or hyperkeratosis of the hands and feet. In addition, anodontia, hypodontia, and conical teeth can be seen in these patients.…”

Section: Introductionmentioning

confidence: 99%

Ectodermal Dysplasia: Otolaryngologic evaluation of 23 Cases

et al. 2012

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The aim of this prospective study was to improve the quality of life of and reduce morbidity for patients with ectodermal dysplasia by assessing their actual and potential ENT pathologies, and offering methods of prevention and treatment. The study was conducted between 2006 and 2008 and included 23 patients diagnosed with ectodermal dysplasia. The major symptoms of ectodermal dysplasia were evaluated. Patient histories were obtained in all cases, and a complete head and neck examination was carried out. Of the 23 patients (11 males and 12 females, aged 5 to 45 years) diagnosed with ectodermal dysplasia, 22 had hypohidrotic ectodermal dysplasia and 1 had ectrodactylyectodermal dysplasia-clefting syndrome. In all patients diagnosed with hypohidrotic ectodermal dysplasia, the salivary glands were examined by ultrasonography and, when necessary, by scintigraphy. Hearing defects in patients with otologic problems were determined by audiometric examination: 39.1% of the patients had hearing loss, 43.5% had otitis media, and 39.1% had impacted cerumen. The most common rhinologic findings were saddle nose deformity in 56.5%, nasal obstruction and nasal dryness (52.2% each), and chronic rhinitis/rhinosinusitis (34.8%). The most common oral and oropharyngeal findings were difficulty chewing in 82.6% and dry mouth in 78.3%. All 23 patients had required dental work. Because this disorder affects several aspects of the body, its treatment requires a multidisciplinary approach, with the otolaryngologist being a vital part of the management team.

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Craniofacial and upper airway cephalometrics in hypohidrotic ectodermal dysplasia

Cited by 16 publications

References 17 publications

Position of the Hyoid Bone in Anteroposterior Skeletal Patterns

Position of the Hyoid Bone in Anteroposterior Skeletal Patterns

Subtle Morphological Changes in the Mandible of Tabby Mice Revealed by Micro-CT Imaging and Elliptical Fourier Quantification

Ectodermal Dysplasia: Otolaryngologic evaluation of 23 Cases

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