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Maxillonasal dysplasia or Binder's syndrome is an uncommon congenital condition characterized by a retruded mid-face with an extremely flat nose. We report here six patients with maxillonasal dysplasia whose noses were corrected with onlay costal cartilage grafts using a combined oral vestibular and external rhinoplasty approach for nasal dorsal au...
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Citations
... Binder syndrome can also be known as maxillonasal dysplasia and is a congenital malformation characterized by nasomaxillary hypoplasia due to underdevelopment of the midfacial skeleton 1 . In 1962, Binder reported three cases and six features: (1) arrhynoid face; (2) abnormal position of the nasal bones; (3) intermaxillary hypoplasia with consecutive malocclusion; (4) reduced or absent anterior nasal spine; (5) atrophy of the nasal mucosa and (6) absence of the frontal sinus (not mandatory) 1,2 . ...
... Binder syndrome can also be known as maxillonasal dysplasia and is a congenital malformation characterized by nasomaxillary hypoplasia due to underdevelopment of the midfacial skeleton 1 . In 1962, Binder reported three cases and six features: (1) arrhynoid face; (2) abnormal position of the nasal bones; (3) intermaxillary hypoplasia with consecutive malocclusion; (4) reduced or absent anterior nasal spine; (5) atrophy of the nasal mucosa and (6) absence of the frontal sinus (not mandatory) 1,2 . The phenotypic appearance is characteristic, with the hypoplastic midface profile, the flat nose, the convex upper lip with a wide philtrum, and the typically crescent or semilunar nostrils giving a half-moon appearance 1 . ...
... In 1962, Binder reported three cases and six features: (1) arrhynoid face; (2) abnormal position of the nasal bones; (3) intermaxillary hypoplasia with consecutive malocclusion; (4) reduced or absent anterior nasal spine; (5) atrophy of the nasal mucosa and (6) absence of the frontal sinus (not mandatory) 1,2 . The phenotypic appearance is characteristic, with the hypoplastic midface profile, the flat nose, the convex upper lip with a wide philtrum, and the typically crescent or semilunar nostrils giving a half-moon appearance 1 . ...
... Some authors prefer to use cartilage graft instead of bone graft to augment the nose [11]. Rune et al [12] studied the properties of bone graft in Binder's syndrome. ...
Binder’s Syndrome or Maxillonasal dysplasia is an uncommon condition that presents as retrusion of the midface. This varies in severity, ranging from minor involvement of the nose to severe hypoplasia of the maxillary complex. Depending upon the severity, different types of procedures are required. In this paper we present our experience with managing such patients.
... The primary objective is to reconstruct nasal deformities, including tip correction and dorsal and columellar lengthening. 35,57,58 However, the timing of surgery has been a point of contention for many years. Many surgeons advocate early corrections due to the considerable psychological effect on the growing child. ...
... It can be avoided by immersing the graft in a 0.9% NaCl and gentamicin solution for approximately 30 min. 58 A K-wire may also stabilise larger grafts and limit the risk of warping. In the case of failure of cartilage grafts, titanium mesh or Silastic implants have been used. ...
Aim:
The classical features of Binder's syndrome (BS) have been widely reported, yet there is a lack of information on diagnostic and treatment challenges. Therefore, we aimed to review the literature on various aspects, including the role of a multidisciplinary approach in the management of BS.
Methods:
A thorough literature search was performed on PubMed, Scopus, Embase, Ovid, Web of Science, EBSCOhost, and Google Scholar using the keywords"Maxillonasal dysplasia", "Maxillonasal dysostosis", "nasomaxillary hypoplasia", "Binder type", "Binder syndrome", "Binder phenotype", and "OMIM 155050". In addition, the articles published in the English language from the inception of the database until June 2021 were considered.
Results:
The search of different databases yielded 958 publications, out of which 145 relevant articles were reviewed. The studies were categorised by year of publication, study design, and theme. Most of the studies were case reports (42%) followed by case series (19%). The most reported topic was based on different surgical techniques (42%). Finally, the results were synthesised into a narrative review.
Conclusions:
The need for a multidisciplinary approach involving clinicians from different specialities is highlighted. Future research is warranted to develop concrete clinical guidelines for the management of this syndrome.
... It can be differentiated from Binder syndrome as nasal bones are not abnormally positioned nor the nasal spine is absent, there is no atrophy of nasal mucosa, absence of maxillary hypoplasia and frontal sinus is present. [7] Although the molecular basis of all types of OFD's are not known till date, molecular study, in this case, was not done due to personal reasons. Considering the varied presentation of this syndrome, diagnosing a particular subtype is difficult. ...
Oro‑facial‑digital syndrome (OFD) is a disorder with varied inheritance patterns. They deal mainly
with abnormalities of the face, digits, i.e., fingers and toes, and oral cavity. Hypoplasia of nasal alar
cartilage, broad nasal bridge, frontal bossing, and micrognathia are the common facial abnormalities
observed in its multiple potential forms. Among the oral features, lobulated tongue, multiple frenulae,
and cleft lip/palate are the common findings. The subject presented in our case manifested cleft of
the hard palate at the time of presentation, along with other diverse features, which could not match
any of the defined OFD types in literature. Furthermore, follow‑up of the infant observed closure of
the palate spontaneously, without any intervention. Hence, the purpose is to add to the knowledge
the typical features of this unspecified type of OFD, along with this unique unprompted cleft palate
closure, which aroused the need of close watch in such cases.
... • Absence of the frontal sinus (not obligatory). 4,5 Individuals with Binder's syndrome have a typical facial appearance with a retruded mid-face, flat nose, underdeveloped upper jaw, relatively protruding lower jaw, half-moon shaped nostrils, and perialar flattening resulting in concave midfacial profile. There may be malocclusion of teeth as well. ...
... There may be malocclusion of teeth as well. [5][6][7] We hereby report to you a rare case of Binder's syndrome and its surgical correction in collaboration with our plastic surgery department. ...
... Our work is similar to his using costal cartilages for single-stage premaxillary and nasal augmentation along with columellar lengthening in an adult patient. 5 Osteotomies and autologous bone and cartilage grafts have been traditionally used through various internal and external approaches by different authors mainly in adolescents. 5,7 Ragnell used iliac cancellous onlay bone chips to augment premaxilla through a median incision at the columellar base and angulated block bone graft for nose in a single stage in teenagers. ...
... • Gorlin et al. suggested that maxillonasal dysplasia is a nonspecific abnormality of the nasomaxillary complex [3,4]. ...
... • Olow-Norderam reported the positive family history in 36% of his patients inherited in an autosomal recessive manner [4]. • Noguchi et al. investigated the correlation between transthyretin gene mutation and facial growth retardation in an animal studies. ...
... The onlay grafting technique like costal cartilage grafts for dorsal nasal augmentation, columellar lengthening, and premaxillary augmentation helps in facial growth [4]. ...
... Binder's syndrome is a rare genetic condition that is characterised by under-development of the middle third of the face leading to hypoplasia of the nasomaxillary complex. 1 Zuckerkandl in 1882 first described the abnormality of the anterior nasal f loor where the anterior nasal spine was replaced by the fossa prenasalis which constituted the inferior margin of the piriform aperture. Later Noyes in 1939 noted a case with flat nasal tip sitting on a retruded maxillonasal base. ...
... Since the vertebral column also develops during the same time period, a large number of such patients also present with vertebral anomalies. 1 Other causative agents such as birth trauma, vitamin K deficiency in the expectant mother, alcohol and drug abuse, use of lithium, ethanol, warfarin or phenytoin based drugs have been indicated. 1,3 At the fifth and sixth week of pregnancy, inhibition of the centre of ossification which gives rise to the inferior and lateral borders of piriform aperture is seen. ...
... 1 Other causative agents such as birth trauma, vitamin K deficiency in the expectant mother, alcohol and drug abuse, use of lithium, ethanol, warfarin or phenytoin based drugs have been indicated. 1,3 At the fifth and sixth week of pregnancy, inhibition of the centre of ossification which gives rise to the inferior and lateral borders of piriform aperture is seen. This leads to localised hypoplasia of the maxilla causing retraction of the columella-lip junction and a lack of the triangular flare of columella seen in normal subjects. ...
... The viability of the tissue sections was also significantly higher in the SL-treated group compared to the control group ( Figure 3). [16]. There are several techniques that can increase the survival of diced cartilage grafts, including the Turkish delight method described by Erol, which has proven effective and popular around the world [3]. ...
Purpose: To evaluate the effect of scopoletin (SL) on fascia-wrapped diced cartilage grafts in rhinoplasty surgery. Methods: Cartilage grafts (2 × 2 cm) from the ears of New Zealand rabbits were diced into sections (1 mm³) and then wrapped in muscle fascia taken from the right rear leg. Each graft was placed on the back of the animal after measuring its weight, and then the rabbits were separated into two groups, viz, control and the SL-treated groups {10 mg/kg, per os (p.o.)}. The treatments were administered for 3 months, the rabbits were sacrificed, and the histopathological features and weight of the grafts were examined. Results: The weight of the grafts in the two groups did not significantly (p < 0.05) differ but the histopathological results suggested that there was a pronounced increase in the viability of the graft tissues in the SL-treated group compared to the control group. Treatment with SL decreased the resorption rate and enhanced basophilia relative to the control group. However, fibrosis, inflammation, and bone metaplasia- and calcification-like factors did not significantly differ (p < 0.05). Conclusion: Treatment with SL significantly enhances the viability of the grafts, and thus may have a beneficial effect on fascia-wrapped diced cartilage grafts. © Pharmacotherapy Group, Faculty of Pharmacy, University of Benin, Benin City, 300001 Nigeria. All rights reserved.
... 24 Bhatt et al. proposed L-shaped costal cartilage grafts for nasal columella and nasal dorsum enhancement, and recommended immersing the graft in a saline solution of gentamicin for 30 min prior to implantation in order to prevent it from warping. 29 Chummun et al. described the use of costochondral grafts for reconstructing craniofacial disorders, including 46 cases of Binder syndrome. 11 They concluded that this kind of procedure resulted in long-lasting correction of underdeveloped noses. ...
... In the authors' opinion, cartilage and dermal grafts show a lower incidence of resorption; this coincides with other authors' observations. 11,[28][29][30] In the present study, no alloplastic materials were used to augment the nasolabiomaxillary region. Other authors report the use of silicone or high-density porous polyethylene implants in the nasal dorsum and columella. ...
Background:
Binder syndrome (BS) is an uncommon congenital underdevelopment of the maxilla and nasal skeleton. Other clinical features include a hypoplastic or absent anterior nasal spine; a short, flat nose with short columella; an acute nasolabial angle; a convex upper lip and class III malocclusion.
Objectives:
The aim of the study was to outline the major characteristics of BS and to present a variety of surgical treatment methods.
Material and methods:
The study included 18 patients treated in the authors' department from 1989 to 2013.
Results:
The patients were predominantly women, aged 6 months to 34 years. Nine patients did not present any co-morbidities, but in the other 9 the most common co-morbidities were a unilateral cleft lip and palate, followed by a cleft palate, a bilateral cleft lip and palate, a cleft lip, GERD, gluten intolerance, oligophrenia, goiter and foot malformation. Most of the patients had not been operated on previously. The most common procedure carried out was an iliac crest bone graft. In 4 patients, no procedures other than cleft lip and palate repair were undertaken. In 3 cases a Le Fort I osteotomy was performed to correct the patients' orthognathic status. No major or life-threating complications were noted. In 2 cases, due to a high degree of resorption of bone grafts, multiple secondary grafting of bone, cartilage and deepithelialized skin was necessary to obtain satisfactory results.
Conclusions:
In BS surgical treatment is the treatment of choice. It results in adequate correction of facial retrusion. However, due to various degrees of bone resorption, the results are not lifelong. No unequivocally superior surgical strategy in BS has been presented so far. Most disagreement among authors is related to the need for and timing of maxillary osteotomy, the choice between bone and cartilage grafting in nose reconstruction and columella lengthening. Although alloplastic materials offer the tempting advantage of fast and simultaneous augmentation of deficient tissues, their use may risk prolonged infections and extrusion, resulting in exacerbations of deformities.
... Augmentation is enough to give an aesthetically appealing and acceptable facial profile in mild to moderate cases (10). ...
