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1.
Orphanet J Rare Dis ; 19(1): 204, 2024 May 18.
Artigo em Inglês | MEDLINE | ID: mdl-38762603

RESUMO

BACKGROUND: Trigonocephaly occurs due to the premature fusion of the metopic suture, leading to a triangular forehead and hypotelorism. This condition often requires surgical correction for morphological and functional indications. Metopic ridges also originate from premature metopic closure but are only associated with mid-frontal bulging; their surgical correction is rarely required. Differential diagnosis between these two conditions can be challenging, especially in minor trigonocephaly. METHODS: Two hundred seven scans of patients with trigonocephaly (90), metopic rigdes (27), and controls (90) were collected. Geometric morphometrics were used to quantify skull and orbital morphology as well as the interfrontal angle and the cephalic index. An innovative method was developed to automatically compute the frontal curvature along the metopic suture. Different machine-learning algorithms were tested to assess the predictive power of morphological data in terms of classification. RESULTS: We showed that control patients, trigonocephaly and metopic rigdes have distinctive skull and orbital shapes. The 3D frontal curvature enabled a clear discrimination between groups (sensitivity and specificity > 92%). Furthermore, we reached an accuracy of 100% in group discrimination when combining 6 univariate measures. CONCLUSION: Two diagnostic tools were proposed and demonstrated to be successful in assisting differential diagnosis for patients with trigonocephaly or metopic ridges. Further clinical assessments are required to validate the practical clinical relevance of these tools.


Assuntos
Craniossinostoses , Humanos , Craniossinostoses/diagnóstico por imagem , Craniossinostoses/patologia , Craniossinostoses/diagnóstico , Feminino , Masculino , Lactente , Imageamento Tridimensional/métodos , Crânio/diagnóstico por imagem , Crânio/patologia
2.
J Morphol ; 284(8): e21609, 2023 08.
Artigo em Inglês | MEDLINE | ID: mdl-37458086

RESUMO

We present a novel method for the morphometric analysis of series of 3D shapes, and demonstrate its relevance for the detection and quantification of two craniofacial anomalies: trigonocephaly and metopic ridges, using CT-scans of young children. Our approach is fully automatic, and does not rely on manual landmark placement and annotations. Our approach furthermore allows to differentiate shape classes, enabling successful differential diagnosis between trigonocephaly and metopic ridges, two related conditions characterized by triangular foreheads. These results were obtained using recent developments in automatic nonrigid 3D shape correspondence methods and specifically spectral approaches based on the functional map framework. Our method can capture local changes in geometric structure, in contrast to methods based, for instance, on global shape descriptors. As such, our approach allows to perform automatic shape classification and provides visual feedback on shape regions associated with different classes of deformations. The flexibility and generality of our approach paves the way for the application of spectral methods in quantitative medicine.


Assuntos
Craniossinostoses , Animais , Tomografia Computadorizada por Raios X , Imageamento Tridimensional/métodos
3.
Plast Reconstr Surg ; 152(3): 612-622, 2023 09 01.
Artigo em Inglês | MEDLINE | ID: mdl-36847681

RESUMO

BACKGROUND: Crouzon syndrome is characterized by complex craniosynostosis and midfacial hypoplasia. Where frontofacial monobloc advancement (FFMBA) is indicated, the method of distraction used to achieve advancement holds an element of equipoise. This two-center retrospective cohort study quantifies the movements produced by internal or external distraction methods used for FFMBA. Using shape analysis, this study evaluates whether the different distraction forces cause plastic deformity of the frontofacial segment, producing distinct morphologic outcomes. METHODS: Patients with Crouzon syndrome who underwent FFMBA with internal distraction [Hôpital Necker-Enfants Malades (Paris, France)] or external distraction [Great Ormond Street Hospital for Children (London, United Kingdom)] were compared. Digital Imaging and Communications in Medicine files of preoperative and postoperative computed tomographic scans were converted to three-dimensional bone meshes and skeletal movements were assessed using nonrigid iterative closest point registration. Displacements were visualized using color maps and statistical analysis of the vectors was undertaken. RESULTS: Fifty-one patients met the strict inclusion criteria. Twenty-five underwent FFMBA with external distraction and 26 with internal distraction. External distraction provides a preferential midfacial advancement, whereas internal distractors produce a more positive movement at the lateral orbital rim. This confers good orbital protection but does not advance the central midface to the same extent. Vector analysis confirmed this to be statistically significant ( P < 0.01). CONCLUSIONS: Morphologic changes resulting from monobloc surgery differ depending on the distraction technique used. Although the relative merits of internal and external distraction still stand, it may be that external distraction is more suited to addressing the midfacial biconcavity seen in syndromic craniosynostosis. CLINICAL QUESTION/LEVEL OF EVIDENCE: Therapeutic, III.


Assuntos
Acrocefalossindactilia , Disostose Craniofacial , Craniossinostoses , Osteogênese por Distração , Criança , Humanos , Estudos Retrospectivos , Osteogênese por Distração/métodos , Ossos Faciais/diagnóstico por imagem , Ossos Faciais/cirurgia , Disostose Craniofacial/diagnóstico por imagem , Disostose Craniofacial/cirurgia , Craniossinostoses/diagnóstico por imagem , Craniossinostoses/cirurgia , Acrocefalossindactilia/diagnóstico por imagem , Acrocefalossindactilia/cirurgia
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