Associations between severity of clefting and maxillary growth in patients with unilateral cleft lip and palate treated with infant orthopedics.

OBJECTIVE: The purpose of this study was to examine possible associations between severity of clefting in infants and maxillary growth in children with complete unilateral cleft lip and palate. DESIGN: This was a retrospective study of measurements made on infant maxillary study casts and maxillary cephalometric variables obtained at 5 to 6 years of follow-up. SETTING: The study was performed at the Institute of Reconstructive Plastic Surgery of New York University Medical Center, New York, New York. PATIENTS: Twenty-four consecutive nonsyndromic unilateral complete cleft lip and palate patients treated during the years 1987 to 1994. INTERVENTIONS: All the patients received uniform treatment (i.e., presurgical orthopedics followed by gingivoperiosteoplasty to close the alveolar cleft combined with repair of the lip and nose in a single stage at the age of 3 to 4 months). Closure of the palate was performed at the age of 12 to 14 months. RESULTS: Infant maxillary study cast measurements correlated in a statistically significant manner with maxillary cephalometric measurements at age 5 to 6 years. CONCLUSIONS: The results demonstrate the large variation in the severity of unilateral cleft lip and palate deformity at birth. Patients with large clefts and small arch circumference, arch length, or both demonstrated less favorable maxillary growth than those with small clefts and large arch circumference or arch length at birth.

Single-follicular-unit hair transplantation to correct cleft lip moustache alopecia.

OBJECTIVE: To present the case of an 18-year-old boy with a cleft lip scar and an obligatory need for facial hair who underwent single-follicular-unit graft hair transplantation that resulted in significant moustache hair restoration in a single procedure. SETTING: The surgery was performed in an outpatient private practice setting using oral sedation and local anesthesia. RESULTS: Advances in instrumentation technology and an increased understanding of the anatomical clustering of hair follicles into so-called "follicular units" containing one to six hairs per unit has resulted in a rapid expansion of hair restoration surgery into new areas including female-pattern alopecia, scarring alopecias, and cosmetic surgery scars. These new techniques can be employed to create natural-looking hair lines in front of artificial hair replacement systems; to improve unnatural looking, old "large-plug" hair transplants; and to correct discontinuity of eyebrows and hairlines in patients with congenital facial clefts. Increased awareness is needed to incorporate follicular-unit graft hair transplant surgery into the family of corrective surgery subspecialties.

Presurgical nasoalveolar orthopedic molding in primary correction of the nose, lip, and alveolus of infants born with unilateral and bilateral clefts.

This addendum to the "State of the Art Dental Treatment of Predental and Infant Patients With Clefts and Craniofacial Anomalies," by Prahl-Andersen (Cleft Palate Craniofac J. 2000;37:528532), offers an extended perspective on this controversial subject. This article reviews the role of combined nasal and alveolar (nasoalveolar) molding in the primary correction of the nose, lip, and alveolus of infants born with unilateral and bilateral clefts. The background of presurgical nasoalveolar orthopedic molding, the technique, and the literature are presented. The proposed benefits of treatment from the traditional techniques of presurgical orthopedics have been shown to be unsubstantiated (Kuijpers-Jagtman and Prahl, 1996). A close comparison of the proposed benefits of earlier forms of presurgical orthopedics, along with those of the current technique of nasoalveolar molding, is presented.

Development of a device for the delivery of agents to bone during distraction osteogenesis.

Various agents have been theoretically and experimentally implicated as mediators of distraction osteogenesis (DO). The purpose of this study was to develop a vehicle for the potential delivery of these factors to the region of the distraction site in an attempt to manipulate this biologic process. Three adult mongrel dogs (12 months old) had oblique osteotomies performed bilaterally through the gonial regions. In group I, the external distracter was affixed to the right hemimandible of two dogs (n = 2 hemimandibles) with cannulated pins (external diameter = 1.5 mm; lumen diameter = 1.0 mm; length = 60 mm), whereas the distracter on the left was affixed with standard, noncannulated pins of the same dimensions. In group II, cannulated pins were used to affix the external distracter to both hemimandibles (n = 2 hemimandibles) of a dog. The devices were activated after a 5-day latency period and were lengthened at a rate of 1 mm/day for 20 days. During the distraction period, 0.1 ml/d of sterile india ink was injected into the cannulated pins, after which the sterile stylet was replaced. The activation protocol was followed by 28 days of fixation (consolidation period). The hemimandibles from group I underwent removal of soft tissues, acetone fixation, and gross examination/photography, whereas the hemimandibles from group II were prepared for histologic evaluation (whole mount, hematoxylin and eosin staining). All dogs survived to the end of the study and demonstrated successful DO without evidence of complications. Hemimandibles in group I displayed evidence of india ink on both the lingual and buccal cortex around the cannulated pin site, in the regenerate and on the neocortices of the distracted segment. Hemimandibles of group II showed histologic evidence of the india ink being deposited densely around the cannulated pin site and extending in a radial fashion around the pin site into the regenerate. This study demonstrates for the first time a vehicle device for the delivery of an inert dye to the regenerate site during distraction osteogenesis. This vehicle offers the potential of delivery of various factors implicated in distraction osteogenesis (i.e., mitogens) in an attempt to alter this process and also substances (i.e., chemotherapy, antibiotics, etc.) for use in the treatment of various osteopathies.

A rat model of gingivoperiosteoplasty.

The ability to avoid a subsequent bone graft makes the use of gingivoperiosteoplasty (GPP) at the time of cleft lip repair an attractive technique. The use of GPP, in combination with presurgical orthodontics, has been shown to result in successful bony union in the majority of patients. However, secondary bone grafting is still necessary in 30% to 40% of patients due to persistent alveolar bony defects. The elucidation of methods to improve the success rates of these procedures has been hampered by the lack of reproducible animal models. The purpose of this study was, therefore, to develop a rodent model of GPP that would facilitate the investigation of methods to improve osteogenesis in alveolar defects. We report a surgically produced rat model (9 x 5 x 3-mm alveolar defect) that is reproducible, inexpensive (relative to large-animal models), and simple technically. In addition, healing in this model occurs in a predictable manner during a 12-week period, thus enabling analysis of methods designed to accelerate or facilitate osseous regeneration.

Controlled multiplanar distraction of the mandible. Part III: Laboratory studies of sagittal (anteroposterior) and horizontal (mediolateral) movements.

Distraction osteogenesis has proven to be an effective technique for the correction of mandibular deficiencies. However, problems have been encountered in achieving a final, idealized form of the mandible when using distraction devices capable of moving the bone segments in only one dimension (uniplanar). Specifically, occlusal irregularities and deficiencies in lower facial contour have been seen following uniplanar distraction. To address these problems, a distraction device capable of independent movements in three planes (multiplanar) was developed. Previously reported studies in a canine model have demonstrated that this device can successfully distract the mandible along both the sagittal axis (anteroposterior or z-axis) and the vertical axis (superoinferior or y-axis). This study examines the ability of the multiplanar device to distract along the sagittal and horizontal axes (mediolateral or x-axis). A total of 12 dogs were included in the study. All animals underwent unilateral or bilateral mandibular distraction using an external multiplanar device. After a latency period of 5 days, primary distraction along the anteroposterior axis at a rate of 1 mm/day for 10 days (10 mm total) was performed. During the following 10 days, along with an additional 11 mm to 20 mm of anteroposterior axis distraction, concomitant secondary distraction was performed along the horizontal (mediolateral) axis at a rate of 5 degrees/day (50 degrees total). Cephalometric radiographs were obtained preoperatively and at the conclusion of both anteroposterior and combined anteroposterior-mediolateral distraction. Computed tomography (CT) scans were obtained preoperatively and at the end of consolidation (28 days), after which all animals were sacrificed and the dry skulls examined. In all animals, distraction along the mediolateral or x-axis was found to change the anteroposterior projection of the mandible. Varus angulation of the device with respect to the midline of the mandible caused compression of the distracted segments and reduced the anteroposterior thrust of the mandible. In contrast, valgus positioning of the device, with respect to the midline of the mandible, created the opposite effect, increasing the distracted length in the anteroposterior direction. The bone (mandibular) segments being distracted assumed the orientation of the device only for valgus positioning of the device (producing a decrease in the bigonial distance). Conversely, there was no effect from the mediolateral angulation on the distracted segments during varus positioning of the device. A possible explanation for this finding may be a greater resistance to an increase in the bigonial distance (varus positioning of the device) posed by obstruction of lateral movement of the condyle. This stands in contrast to a decrease in the bigonial distance observed following valgus positioning of the device. These findings confirm the clinical impression that distraction along the anteroposterior or sagittal axis remains the critical or keystone therapeutic maneuver in distraction of the mandible. Mediolateral or horizontal axis distraction is best used only as a supplementary movement; in essence, it only affects the anteroposterior dimension with little impact on clinically relevant changes to the bigonial distance.

Presurgical nasoalveolar molding in infants with cleft lip and palate.

Presurgical infant orthopedics has been employed since the 1950s as an adjunctive neonatal therapy for the correction of cleft lip and palate. In this paper, we present a paradigm shift from the traditional methods of presurgical infant orthopedics. Some of the problems that the traditional approach failed to address include the deformity of the nasal cartilages in unilateral as well as bilateral clefts of the lip and palate and the deficiency of columella tissue in infants with bilateral clefts. The nasoalveolar molding (NAM) technique we describe uses acrylic nasal stents attached to the vestibular shield of an oral molding plate to mold the nasal alar cartilages into normal form and position during the neonatal period. This technique takes advantage of the malleability of immature cartilage and its ability to maintain a permanent correction of its form. In addition, we demonstrate the ability to nonsurgically construct the columella through the application of tissue expansion principles. This construction is performed by gradual elongation of the nasal stents and the application of tissue-expanding elastic forces that are applied to the prolabium. Use of the NAM technique has eliminated surgical columella reconstruction and the resultant scar tissue from the standard of care in this cleft palate center.

Long-term effects of nasoalveolar molding on three-dimensional nasal shape in unilateral clefts.

OBJECTIVE: This objective of this study was to determine the effect of presurgical nasoalveolar molding on long-term nasal shape in complete unilateral clefts. DESIGN: The study was retrospective, and the subjects were chosen at random. Nasal casts of the subjects were scanned in three dimensions. Each nose was best fit to its mirror image, and a numerical asymmetry score was determined. SETTING: All patients were treated at the Institute of Reconstructive Plastic Surgery, NYU Medical Center, New York, New York. PATIENTS: The study subjects (n = 10) were selected from a group that had undergone presurgical nasal molding in conjunction with alveolar molding. The control subjects (n = 10) were selected from a group that had undergone presurgical alveolar molding alone. INTERVENTIONS: All subjects underwent presurgical orthopedic treatment until the age of approximately 4 months at which time the primary surgery was performed. MAIN OUTCOME MEASURE: The nasal shape following nasal molding should be more symmetrical than if molding had not been done. RESULTS: The mean asymmetry index for the nasoalveolar molding group was 0.74, and the control group was 1.21. This difference was statistically significant (p < .05). CONCLUSIONS: Presurgical nasoalveolar molding significantly increases the symmetry of the nose. The increase in symmetry is maintained long term into early childhood. The limitations of this study include (1) asymmetry alone is not an adequate shape result in most situations, (2) the children evaluated in this study were not fully grown, and (3) the control group was not age matched.

Distraction osteogenesis of the mandible: a ten-year experience.

Mandibular distraction has been performed at the authors' institution for the past 10 years on a variety of craniofacial anomalies. This article reviews the experience with distraction and outlines the authors' treatment algorithms based on patient age and pathology. The roles of distraction versus conventional orthognathic surgery are reviewed. The need for preoperative surgical planning and postoperative orthodontic therapy is emphasized.

Treatment planning and biomechanics of distraction osteogenesis from an orthodontic perspective.

As in traditional combined surgical and orthodontic procedures, the orthodontist has a role in the planning and orthodontic support of patients undergoing distraction osteogenesis. This role includes predistraction assessment of the craniofacial skeleton and occlusal function in addition to planning both the predistraction and postdistraction orthodontic care. Based on careful clinical evaluation, dental study models, photographic analysis, cephalometric evaluation, and evaluation of three-dimensional computed tomographic scans, the orthodontist, in collaboration with the surgeon, plans distraction device placement and the predicted vectors of distraction. Both surgeon and orthodontist closely monitor the patient during the active distraction phase, using intermaxillary elastic traction, sometimes combined with guide planes, bite plates, and stabilization arches, to mold the newly formed bone (regenerate) while optimizing the developing occlusion. Postdistraction change caused by relapse is minimal. Growth after mandibular distraction is variable and appears to be dependent on the genetic program of the native bone and the surrounding soft tissue matrix. A significant advantage of distraction osteogenesis is the gradual lengthening of the soft tissues and surrounding functional spaces. Distraction osteogenesis can be applied at an earlier age than traditional orthognathic surgery because the technique is relatively simple and bone grafts are not required for augmentation of the hypoplastic craniofacial skeleton. In this new technique, the surgeon and the orthodontist have become collaborators in a process that gradually alters the magnitude and direction of craniofacial growth.

Early decannulation with bilateral mandibular distraction for tracheostomy-dependent patients.

Obstructive sleep apnea in the neonatal period may originate from a hypoplastic mandibular framework causing retroposition of the base of the tongue and an inadequate hypopharyngeal space. A tracheotomy in childhood is an effective treatment for obstructive sleep apnea, but it is associated with increased morbidity, management problems, and difficulties in social interaction. Tracheostomy-dependent pediatric patients who underwent mandibular distraction were reviewed to determine the effectiveness of this technique in achieving decannulation. A clinical review was completed to determine the status of the tracheostomy after external, unidirectional distraction in tracheostomy-dependent patients. Expansion of the mandibular framework was analyzed using traditional bony landmarks on predistraction and postdistraction lateral cephalograms. The area of the lower face was analyzed, and changes in the position of the hyoid bone were determined. Four patients with tracheostomies underwent an average of 21.3 mm and 20.8 mm of distraction on the left and right hemimandibles, respectively. The average age at the time of distraction was 2.7 years (range, 2.2 to 3.2 years). All patients underwent successful decannulation at an average of 3.8 months (range, 1.5 to 5.5 months) after completion of distraction. The area of the lower face increased 26.9 percent (range, 12.2 to 53.5 percent) after distraction, and the hyoid bone advanced an average of 14.5 mm (range, 8 to 25 mm). Bilateral mandibular distraction is an effective method of expanding the mandibular framework and concomitantly advancing the base of the tongue. The technique provides a tool for early intervention and decannulation in pediatric patients with indwelling tracheostomies secondary to mandibular deficiencies.

Controlled multiplanar distraction of the mandible: device development and clinical application.

Distraction osteogenesis has been shown to be an effective method of lengthening and augmenting endochondral bone. It has also been applied effectively in the reconstruction of the membranous bones of the craniofacial skeleton. With the accumulation of clinical experience in mandibular distraction, the differences between endochondral and membranous bone distraction have become apparent, especially in the limitations of uniplanar distraction for the three-dimensional reconstruction of the deficient mandible. Distraction of the mandible in a single plane cannot satisfy fully the functional and structural requirements of the patient with malocclusion as well as deficiency of the skeletal and soft tissue. This study reports the development and clinical use of a multiplanar mandibular distraction device with the ability to achieve linear distraction (Z-plane or sagittal), angular distraction (Y-plane or vertical), and transverse distraction (X-plane or coronal). The device contains two independent gear arrangements attached to two arms that extend from the central unit. Therefore, the trajectory of the regenerated bone may be changed during the distraction process. The device also allows manipulation of the various planes of movement independent of each other. Furthermore, the rotational points for the multiplanar distraction devices are located at a single point; therefore only a single osteotomy and two pin sites are required. The multiplanar distraction device allows the surgeon to customize and contour the dimensions of the distraction process by controlling the trajectory of the translation of the regenerated bone.

Hypertrophy and asymmetry of the facial muscles: a previously unrecognized association.

We present 6 patients who do not satisfy the criteria for inclusion into any known diagnostic group, although they share similar physical findings. All had facial findings suggestive of either mild deformational plagiocephaly or craniofacial microsomia; however, hypertrophic and occasionally hyperactive muscles of the face appear to be the unifying and distinctive feature of the group. Contraction of hypertrophic postauricular muscles on the affected side resulted in movement and contraction of the pinna. Hypertrophy of the mentalis and masseter muscles was also frequently observed. Additionally, anomalous neck musculature was seen in 4 of the 6 patients. In 2 patients, the hypertrophy of certain of these muscles produced enough deformity to warrant excision. This is the first report of such an association in the literature. Possible explanations are discussed.

Reduced need for alveolar bone grafting by presurgical orthopedics and primary gingivoperiosteoplasty.

OBJECTIVE: The purpose of this study was to evaluate if narrowing and approximation of the alveolar cleft through presurgical alveolar molding followed by gingivoperiosteoplasty (GPP) at the time of lip repair reduces the need for a bone-grafting procedure. DESIGN: This was a retrospective blind study of patients with unilateral or bilateral alveolar clefts who underwent presurgical infant alveolar molding and GPP by a single surgeon. Alveolar bone formation was assessed prior to the eruption of the maxillary lateral incisor or canine by clinical examination, panoramic and periapical radiographs, and/or a dental CT scan. The criterion for bone grafting was inadequate bone stock to permit the eruption and maintenance of the permanent dentition. SETTING: This study was performed at the Institute of Reconstructive Plastic Surgery by the members of the Cleft Palate Team. PATIENTS: All patients with unilateral (n = 16) or bilateral (n = 2) alveolar clefts who underwent presurgical infant alveolar molding and GPP by a single surgeon from 1985 to 1988 were studied. The control population consisted of all alveolar cleft patients (n = 14) who did not undergo alveolar modeling or GPP during the same time period. INTERVENTIONS: Presurgical alveolar modeling was performed with an intraoral acrylic molding plate. This plate was modified on a weekly basis to align the alveolar segments and close the alveolar gap. The surgical intervention consisted of a modified Millard GPP. MAIN OUTCOME MEASURES: The primary study outcome measure was the elimination of the need for a secondary bone graft in patients who underwent presurgical alveolar molding and GPP. RESULTS: Of the 20 sites in the 18 patients who underwent GPP, 12 sites did not require an alveolar bone graft. Of the 8 sites requiring a bone graft, 4 presented minimal bony defects. All 14 patients in the control group required bone grafts. CONCLUSIONS: In this series of 20 alveolar cleft sites treated with presurgical orthopedics and GPP, 60% did not need a secondary alveolar bone graft in the mixed dentition.

Controlled multiplanar distraction of the mandible, Part II: Laboratory studies of sagittal (anteroposterior) and vertical (superoinferior) movements.

The application of distraction osteogenesis in craniofacial surgery has significantly altered the treatment of congenital mandibular deficiencies. However, evaluation of results in both animal studies and clinical cases has revealed deficiencies, particularly in two areas. First, distraction using a uniplanar device in an anteroposterior direction can result in a persistent anterior open bite. Second, the lateralization of the distracted hemimandible was often limited, with insufficient incremental gain in the bigonial distance. To overcome these shortcomings, a multiplanar distraction device was developed and tested in the canine model. This report details canine studies addressing the first problem: combined anteroposterior or sagittal (z-axis) and superoinferior or vertical (y-axis) movements. Six dogs underwent bilateral mandibular distraction with an external (extraoral), multiplanar device and completed sagittal plus vertical distraction. Evaluation included clinical examination (facial form, jaw position, and occlusion), photography, cephalograms (posteroanterior, basilar, and lateral), three-dimensional computed tomography reconstructions, and examination of dry skulls. The dogs averaged 18.5 mm (range, 15-20 mm) of sagittal distraction and 41.0 degrees (range, 21-50 degrees) of vertical distraction. Marked anterior open bites were produced after vertical distraction secondary to premature contact of the maxillary and mandibular molars. Distraction in the vertical direction also had the additive effect of increasing the sagittal gains by approximately 5% to 10%. In conclusion, a multiplanar distraction device (with the potential for distraction in three planes) was effective in increasing mandibular anteroposterior thrust (sagittal distraction) and also in creating an anterior open bite (vertical or superoinferior distraction). Vertical distraction probably requires bilateral osteotomies to obtain optimal results. The preliminary gains in sagittal length are modified (reduced or increased) after distraction in a second plane (vertical and horizontal). Specifically, vertical distraction in the inferior direction (creating an open bite) also leads to isolated increases in the anteroposterior plane. Conversely, vertical distraction in the superior direction (closing an open bite), as seen in a human malocclusion, may lead to isolated decreases in the anteroposterior plane, but this question remains to be investigated in the laboratory.

A CT scan technique for quantitative volumetric assessment of the mandible after distraction osteogenesis.

Distraction osteogenesis has become an accepted method of treatment for patients requiring reconstruction of hypoplastic mandibles. We present a quantitative analysis of volumetric changes after distraction osteogenesis in a series of 10 patients. Group I (n = 5 patients, 3 unilateral craniofacial microsomia, 1 Goldenhaar syndrome, and 1 bilateral craniofacial microsomia) underwent unilateral distraction of the mandible. Group II (n = 5 patients, 1 Nager syndrome, 1 bilateral craniofacial microsomia, 1 developmental micrognathia, and 2 Treacher Collins syndrome) underwent bilateral distraction of the mandible. Predistraction and postdistraction axial and three-dimensional computed tomographic (CT) scans were digitized and transferred to a computer for analysis with image-processing software to determine the changes in volume of the mandible and bony regenerate. The CT-derived volume method was validated by scanning three dry cadaver mandible specimens and comparing the volume data with those derived from a water-displacement method. The difference between the two methods was less than 5 percent. The mean distracted length, as recorded from the calibrated device, was 22.6 mm in the 10 patients. In the unilateral distraction group, the mean increase in hemimandibular bone volume was 2.8 cc, with a mean percentage increase of 27 percent in the distracted hemimandible. In the bilaterally distracted patients, the mean increase in total mandibular volume was 7.9 cc, with a mean percentage increase in bone volume of 25 percent. This study represents the first attempt to quantify the increase in bone volume resulting from distraction osteogenesis. Quantitative volumetric analysis of CT scans is an accurate method to measure the amount of bone regenerate in patients undergoing distraction osteogenesis of the mandible or the extremities. The concept and utility of quantifying the volumetric changes in bone following distraction osteogenesis may become more important as multiplanar devices are developed and used in other areas of the craniofacial skeleton.

Gingivoperiosteoplasty and midfacial growth.

The objective of this study was to report the effect of gingivoperiosteoplasty on growth of the midfacial skeleton 6 years following primary surgical repair. Patients with complete unilateral cleft lip and palate who underwent primary cleft lip and nose repair with and without gingivoperiosteoplasty (GPP) were retrospectively compared by means of a lateral cephalogram. Mean age at the time of evaluation was 5.7 years. All patients were treated at the Institute of Reconstructive Plastic Surgery, New York University Medical Center. All surgery and presurgical orthopedics was performed by the same surgeon and the same orthodontist. Twenty-five consecutively treated patients who presented with complete unilateral clefts of the primary and secondary palate were included in the study. Of these, 20 patients were available for 6-year follow-up cephalometric documentation and review. All patients received preoperative orthopedics with passive molding appliances, followed by repair of the lip, alveolus, and nose in a single stage at the age of 3 months. The repair was performed using the rotation/advancement technique. The difference between the two groups was whether or not gingivoperiosteoplasty was performed. The reason for not performing gingivoperiosteoplasty was incomplete approximation of the alveolar segments usually due to a late start in beginning therapy. Lateral cephalograms (68.5 months post primary surgery) were obtained and traced. Cranial base (S-N), maxilla (ANS-PNS), and mandible (Go-Pg) were digitized for shape coordinate analysis. No significant difference in the mean position of ANS-PNS was found between groups (with or without gingivoperiosteoplasty). There was, however, a significant difference in the variance of position for the points ANS-PNS between the groups (p < .002). We were unable to observe any difference (anteroposterior or supero-inferior) in the average position of the hard palate (ANS-PNS) between groups. We conclude that gingivoperiosteoplasty results in a more uniform position of the hard palate (ANS-PNS) relative to patients that did not receive gingivoperiosteoplasty. We were unable to demonstrate any clear impairment of maxillary growth in the patients treated with gingivoperiosteoplasty when compared to patients treated without gingivoperiosteoplasty.