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A different look: 3-dimensional facial imaging of a child with Binder syndrome

2007; Elsevier BV; Volume: 132; Issue: 5 Linguagem: Inglês

10.1016/j.ajodo.2007.01.020

1097-6752

Chung How Kau, Lindsay Hunter, Emma J. Hingston,

Cleft Lip and Palate Research

Binder syndrome, or maxillonasal dysplasia, is an uncommon developmental anomaly characterized by an unusually flat, underdeveloped midface, with an abnormally short nose and a flat nasal bridge. In this article, we review the literature and describe how 3-dimensional laser scanning was used to evaluate the facial morphology of a 12-year-old boy with this syndrome. Binder syndrome, or maxillonasal dysplasia, is an uncommon developmental anomaly characterized by an unusually flat, underdeveloped midface, with an abnormally short nose and a flat nasal bridge. In this article, we review the literature and describe how 3-dimensional laser scanning was used to evaluate the facial morphology of a 12-year-old boy with this syndrome. The essential features of maxillonasal dysplasia were first described by Noyes.1Noyes F.B. Case report.Angle Orthod. 1939; 9: 160-165Google Scholar However, it was over 20 years before Binder2Binder K.H. Dysostosis maxillo-nasalis, ein arhinencephaler missbildungskomplex Deutsh.Zahnaerztl Z. 1962; 17: 438-444Google Scholar recorded a distinct syndrome of maxillonasal dysplasia, characterized by a short nose with a flat bridge, a short columella, an acute nasolabial angle, perialar flatness, a convex upper lip, and a tendency to Class III malocclusion. There is little published data on the incidence of Binder syndrome, in part because patients in some ethnic groups, such as the Japanese, are undiagnosed. However, several authors have attempted to determine its etiology. Olow-Nordenram and Valentin3Olow-Nordenram M. Valentin J. An etiologic study of maxillonasal dysplasia—Binder's syndrome.Scand J Dent Res. 1988; 96: 69-74PubMed Google Scholar studied 60 families to see whether there was an autosomal recessive inheritance pattern. They concluded that the syndrome either is inherited as an autosomal recessive trait with incomplete penetrance or a threshold characteristic with a genetically multi-factorial background. In contrast, Gross-Kieselstein et al4Gross-Kieselstein E. Har-Even Y. Navon P. Branski D. Familial variant of maxillonasal dysplasia?.J Craniofac Genet Dev Biol. 1986; 6: 331-334PubMed Google Scholar suggested a dominant mode of inheritance after examining an affected mother and daughter. Several studies have investigated the craniofacial morphology of patients with Binder syndrome by using lateral cephalometric radiographs. One such study compared the lateral cephalometric radiographs of 14 patients with Binder syndrome who had no prior surgical or orthodontic treatment with published age- and sex-matched norms.5Carach B. Woods M. Scott P. Maxillonasal dysplasia (Binder syndrome): a lateral cephalometric assessment.Aust Orthod J. 2002; 18: 82-91PubMed Google Scholar They found that the anteroposterior dimensions of the anterior cranial base and the maxilla were reduced, with most patients having a Class III skeletal relationship. Despite the prominence of the mandibular incisors, both overjet and overbite were within normal ranges. In addition to the facial features of Binder syndrome, several authors presented evidence of cervical spine abnormalities. One study involved radiographic examinations of 43 patients with maxillonasal dysplasia and found that 44% had malformations of the cervical vertebrae.6Olow-Nordenram M.A. Radberg C.T. Maxillo-nasal dysplasia (Binder syndrome) and associated malformations of the cervical spine.Acta Radiol Diagn. 1984; 25 (Stockh): 353-360PubMed Google Scholar The atlas and the axis are most frequently affected.7Hopkin G.B. Hypoplasia of the middle third of the face associated with congenital absence of the anterior nasal spine, depression of the nasal bones, and Angle class III malocclusion.Br J Plast Surg. 1963; 16: 146-153Abstract Full Text PDF PubMed Scopus (37) Google Scholar In addition, 6 of the patients in the series had dysplasia of the vertebral bodies related to persistence of the chorda dorsalis. The incidence or theseverity of the malformations did not appear to be related to the degree of maxillonasal dysplasia. In a study of 28 cases of maxillonasal dysostosis, it was found that 54% had craniospinal and cervicospinal malformations.8Resche F. Tessier P. Delaire J. Tulasne J.F. Craniospinal and cervicospinal malformations associated with maxillonasal dysostosis (Binder syndrome).Head Neck Surg. 1980; 3: 123-131Crossref PubMed Scopus (31) Google Scholar Much of the published material on Binder syndrome is in orthodontic, head and neck surgery, and plastic surgery journals, and focuses on facial reconstruction. The management of these patients depends on the level of complexity because of variations in the midface discrepancy and the occlusal relationship. Some lesser-affected patients can be managed by using orthodontic camouflage alone, whereas others require multi-disciplinary input. The effects of posteroanterior traction, used to improve the facial esthetics of an 11-year-old boy with maxillonasal dysplasia, were analyzed; improvement in maxillary retrusion vis-à-vis the mandible only was accomplished, and the recorded advancement of the maxillary bones was slight (0.6 mm).9Rune B. Sarnas K.V. Selvik G. Jacobsson S. Posteroanterior traction in maxillonasal dysplasia (Binder syndrome) A roentgen stereometric study with the aid of metallic implants.Am J Orthod. 1982; 81: 65-70Abstract Full Text PDF PubMed Scopus (20) Google Scholar Osteotomy and grafts (bone or cartilage) are frequently used in the management of Binder syndrome. Grafts, when limited to the nasal sill, do not address the "dish face" deformity characteristic of the syndrome. LeFort II osteotomy, although correcting the midface deficiency, can overcorrect the occlusal relationship if good dental camouflage has occurred, making a LeFort I osteotomy or orthodontic therapy necessary. It has been suggested that mild cases of Binder syndrome, without severe malocclusion, can be managed by using a cartilage graft around the nostril sill, dorsum, and the pyriform aperture, correcting the paranasal and infraorbital deformity without osteotomy.10Watanabe T. Matsuo K. Augmentation with cartilage grafts around the pyriform aperture to improve the midface and profile in Binder's syndrome.Ann Plast Surg. 1996; 36: 206-211Crossref PubMed Scopus (17) Google Scholar In more severe cases, however, a LeFort II osteotomy can correct the midface deficiency and the Class III occlusion seen in 15% of patients with Binder syndrome, but it does not always fully address the nasal flatness. Twenty-seven patients with maxillonasal dysplasia of various degrees were examined on both short- and long-term bases.11Monasterio F.O. Molina F. McClintock J.S. Nasal correction in Binder's syndrome: the evolution of a treatment plan.Aesthetic Plast Surg. 1997; 21: 299-308Crossref PubMed Scopus (35) Google Scholar For some patients, surgical treatment began at age 3, but others were treated as teenagers or young adults. The overall goals were to augment the skeletal deficiencies of the midface and begin the soft-tissue expansion process as early as possible. Fifteen percent of the patients had a Class III malocclusion and were managed with osteotomies; the remainder were managed with various grafts. Long-term follow-up, up to 15 years, showed that surgical treatment should begin early to take advantage of greater skin elasticity and to aid self-image. In a young patient, sequential lengthening procedures of the dorsum and the columella are beneficial. Paranasal and midfacial augmentation is reserved until midfacial growth is near complete, when patients are in their midteen years. In this article, we present a 12-year-old boy with Binder syndrome. Three-dimensional laser scanning, a noninvasive tool, was used to study his facial morphology and to aid in planning the management of his facial deformity.12Kau C.H. Zhurov A.I. Scheer R. Bouwman S. Richmond S. The feasibility of measuring three-dimensional facial morphology in children.Orthod Craniofac Res. 2004; 7: 198-204Crossref PubMed Scopus (74) Google Scholar The patient, aged 12 years 3 months, came to the Paediatric Department of the University Dental Hospital, Cardiff, United Kingdom, for a regular recall appointment (Fig 1). A local community dental officer had initially referred him to this department for complete care at age 2, at the request of his parents. His medical history showed that he had the Binder type of chondroplasia punctata, mild pulmonary stenosis, and a previous atrial septal defect, which closed spontaneously in infancy. His mother reported that he did not suffer from shortness of breath, and there were no concerns over his general development. The patient's height was normal for his age. He had a flat midface with half-moon–shaped nasal apertures, a broad philtrum, and a deep fold between his nose and lip, typical of the Binder type of chondroplasia punctata.13McCollum A.G. Wolford L.M. Binder syndrome: literature review and long-term follow-up on two cases.Int J Adult Orthod Orthognath Surg. 1998; 13: 45-58PubMed Google Scholar In addition, he had a markedly Class III skeletal base. He was well adjusted and was not experiencing teasing at school, due at least in part to his outgoing personality; he did, however, express emerging concern about his facial appearance, especially in relation to the flatness of his nose. The intraoral examination showed he was in the late mixed dentition and had excellent oral hygiene. The maxillary first premolars were extracted for orthodontic purposes, and the patient wore a removable space maintainer for 1 year postextraction to allow eruption of the impacted maxillary permanent canines. There was a reverse overjet of 6 mm and an anterior open bite. A dental panoramic radiograph taken before extraction of the maxillary first premolars showed that a full complement of permanent teeth was developing (with the mandibular deciduous molars still present), and there were no obvious bony abnormalities. New bitewing radiographs were obtained, confirming a caries-free dentition. A lateral cephalometric radiograph (Fig 2) demonstrated the absence of the anterior nasal spine and the thin bony plate overlying the maxillary central incisors. These findings are in line with those of other authors. However, our patient had apparently normal frontal sinuses, whereas these have been described as small or absent in other patients. Because cervical spine abnormalities are a relatively common feature of Binder syndrome, anteroposterior and lateral views of the cervical spine were requested, because the patient will require surgical intervention soon. The films showed deficiencies of the anterosuperior aspects of the cervical vertebrae, typical of the syndrome. The arch of C1 appeared to be intact but did not articulate normally with the hypoplastic dens. It was difficult to determine whether C1 was attached to the base of the skull, but the alignment was considered normal (Fig 3). The patient will require flexion and extension views before general anesthetic. Three-dimensional laser scanning was originally used in industry, but it now gives us a noninvasive, fast, and reproducible means of evaluating the effects of growth and surgical and nonsurgical interventions in the facial region.14Aung S.C. Ngim R.C. Lee S.T. Evaluation of the laser scanner as a surface measuring tool and its accuracy compared with direct facial anthropometric measurements.Br J Plast Surg. 1995; 48: 551-558Abstract Full Text PDF PubMed Scopus (167) Google Scholar It has been shown to be appropriate and reliable when used to study the facial morphology of children.15Kau C.H. Hartles F.R. Knox J. Zhurov A.I. Richmond S. Measuring facial morphology in young subjects.in: Middleton J. Shrive M.G. Jones M.L. Computer methods in biomechanics and biomedical engineering. First Numerics, Cardiff, United Kingdom2005Google Scholar, 16Moss J.P. The use of three-dimensional imaging in orthodontics.Eur J Orthod. 2006; 28: 416-425Crossref PubMed Scopus (32) Google Scholar The technique used in this study has been reported previously,15Kau C.H. Hartles F.R. Knox J. Zhurov A.I. Richmond S. Measuring facial morphology in young subjects.in: Middleton J. Shrive M.G. Jones M.L. Computer methods in biomechanics and biomedical engineering. First Numerics, Cardiff, United Kingdom2005Google Scholar, 17Kau C.H. Richmond S. Zhurov A.I. Application of 3-dimensional soft tissue imaging in orthodontics.in: McNamara Jr, J.A. Kapila S. Digital radiography and three-dimensional imaging Monograph 43. University of Michigan, Ann Arbor: Center for Human Growth and Development2006: 159-179Google Scholar, 18Kau C.H. Richmond S. Savio C. Mallorie C. Measuring adult facial morphology in three dimensions.Angle Orthod. 2006; 76: 771-776Google Scholar, 19Kau C.H. Hartles F.R. Knox J. Zhurov A.I. Richmond S. Natural head posture for measuring three-dimensional soft tissue morphology.in: Middleton J. Shrive M.G. Jones M.L. Computer methods in biomechanics and biomedical engineering. First Numerics, Cardiff, United Kingdom2005Google Scholar, 20Kau C.H. Zhurov A. Richmond S. Cronin A. Savio C. Mallorie C. Facial templates: a new perspective in three dimensions.Orthod Craniofac Res. 2006; 9: 10-17Crossref PubMed Scopus (51) Google Scholar, 21Kau C.H. Zhurov A.I. Bibb R. Hunter M.L. Richmond S. The investigation of the changing facial appearance of identical twins employing a three-dimensional laser imaging system.Orthod Craniofac Res. 2005; 8: 85-90Crossref PubMed Scopus (51) Google Scholar, 22Kau C.H. Zhurov A.I. Knox J. Richmond S. The validity and reliability of a portable 3-dimensional laser scanner for field studies.in: Giuliani R. Galliani E. Proceedings of the 7th European Craniofacial Congress; 2003 Nov 20-22. Monduzzi Editore, International Proceedings Division, Bologna, Italy2004: 41-45Google Scholar Briefly, 2 high-resolution Minolta Vivid VI900 3D (Osaka, Japan) cameras, with a reported manufacturer's accuracy of 0.3 mm, operating as a stereo pair, were used. The cameras emit an eye-safe Class I laser that reflects from a mirror, spreading the beam to measure and capture the surface of the face. Positions of the data points were calculated with triangulation, and the results were transferred to a desktop workstation with a computer. A custom-made studio facilitated standardized light conditions, and natural head posture was used to allow for a clinically reproducible pose for each scan.23Kau C.H. Richmond S. Zhurov A.I. Knox J. Chestnutt I. Hartles F.R. et al.Reliability of measuring facial morphology with a 3-dimensional laser scanning system.Am J Orthod Dentofacial Orthop. 2005; 128: 424-430Abstract Full Text Full Text PDF PubMed Scopus (154) Google Scholar The patient sat on a self-adjustable stool and was asked to look into a mirror with standard horizontal and vertical lines simulating a cross on it. He was then asked to adjust the height of the stool so that he could achieve natural head position with his eyes level with the horizontal line and the midline of his face coincident with the vertical line on the mirror. The scan lasted 7.5 seconds and enabled the capture of the subject's facial morphology. The scans of 42 nonsyndromic boys at age 11.5 years with Class I skeletal relationship, who had not received orthodontic treatment, were combined to produce an average face.20Kau C.H. Zhurov A. Richmond S. Cronin A. Savio C. Mallorie C. Facial templates: a new perspective in three dimensions.Orthod Craniofac Res. 2006; 9: 10-17Crossref PubMed Scopus (51) Google Scholar The 3-dimensional scan of the patient was manually aligned to the inner and outer cantus of the eyes and the corners of the mouth. The images were then aligned by using the iterative closest point algorithm without the need for further magnifications. This produced a superimposition that allowed visualization of the Binder deformity and highlighted the gross deficiency of the nose and the maxilla. The lateral view (Fig 4) shows the Binder profile superimposed on the average profile; the mandible is not prognathic and might be a little small. The three-quarter view is color coded to highlight the regions of deficiency of the Binder face. The scale to the left of the image enables measurement of the differences between the average and the Binder faces in millimeters. The tip of the nose of the child with Binder syndrome is deficient by about 15 to 17 mm (Fig 5).Fig 5Three-quarter view scan, aligned over average image and color coded to highlight deficiency.View Large Image Figure ViewerDownload Hi-res image Download (PPT) The patient's future treatment will greatly depend on his wishes. For some lesser-affected patients, management can be relatively simple and consist solely of orthodontic camouflage. However, these procedures will address only the dental occlusion and not the facial profile.24Aguirre I.U. Dogliotti P.L. Transmaxillary osteogenesis distraction combined with orthopedics and orthodontics in the correction of a severe retrusion of the upper maxilla.J Craniofac Surg. 2004; 15: 785-791Crossref PubMed Scopus (2) Google Scholar This patient has a flat midface, and this area is difficult to augment; improving his profile will require a combination of surgical procedures. The extent of the deficiency should be reassessed about age 16, when growth is almost complete. Surgery before the midteen years can produce scarring that might inhibit potentially favorable growth. It is anticipated that the surgical management of this patient will involve an osteotomy and nasal augmentation with an L-shaped bone strut. Cartilage is occasionally used, but the degree of uplift required in this patient prohibits its use. Surgery was delayed because the patient has received excellent parental support and is extremely well adjusted. Early treatment risks scarring but, more importantly, can result in multiple procedures that risk reduced compliance. In addition, this patient has adequate soft tissue over the bridge of his nose, and tissue expansion should not be required. There has been much debate about the extent of the Binder deformity. Many authors believe that it is restricted to the nasal area, whereas others state that it affects the entire midface. Three-dimensional scans highlight the extent of the area affected in a patient with Binder syndrome. The information from the scan will enable enhanced planning of any future surgical intervention to create an ideal facial balance and allow the patient to be better informed about his options. It serves as a useful 3-dimensional soft-tissue template from which a treatment plan and an objective final point can be made. However, further evaluation methods (computed tomography or cone-beam computed tomography scans) will be required for this patient's definitive treatment planning.