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.

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.

Secondary cleft lip nasal reconstruction: state of the art.

OBJECTIVE: This paper summarizes the state of the art in secondary cleft lip nasal reconstruction, distilled from the many papers written on the subject and from the author's experience with many of those procedures over the past 25 years. METHODS: The evaluation starts with the skeletal base and the need for LeFort 1 or alveolar bone grafting is discussed. The boney dorsum is next evaluated and a "monobloc" osteotomy considered. The cartilaginous dorsum follows and a "spreader-strut" graft is entertained. The tip cartilages are approached with either an open Potter or Dibbell preferred or replacement conchal graft if the tip has been destroyed by previous surgery. The skin envelope is then adjusted using methods described by Tajima, Dibbell, and Bardach.

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.

Cerclage suture method for closed-tip rhinoplasty.

This article presents a method for passing a cerclage suture around the nasal tip complex to narrow it. The method does not require elevation of the skin from the lower lateral cartilages. The method requires a double-pointed straight needle with the suture swaged on in the center.

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.

Average African American three-dimensional computed tomography skull images: the potential clinical importance of ethnicity and sex.

The production of average 'normative' three-dimensional (3D) computed tomography surface images of the bony skull has only recently been explored. The authors wish to determine the effect of using sex- and ethnicity-specific adult average 3D skull images for comparisons with patient images at various stages of craniofacial surgical management (i.e., diagnosis, treatment planning, prosthetic design, image-guided operative procedures, and outcomes assessment). Craniofacial surgical reconstruction for abnormal patterns of development, cancer resection, or trauma are most likely to benefit from these comparisons. To morphometrically test the significance of separating normative 3D skull data by sex and ethnicity, the authors collected 52 3D, anatomical landmarks from 3D computed tomography scans of dry skulls of 20 Americans of European ethnicity and 20 Americans of primarily African (i.e., primarily African and some European) ethnicity. A Procrustes-based morphometric analysis of shape detects 1.2 times as much interethnic variance as intersex variance. The African American sample presents 4.2% more dolichocephaly, wider orbits, flatter nasal area, larger gnathic anatomy, and more procumbent dentition. Pooling the sexes across both groups, it is seen that men tend to have less bulbous crania, more protruding brows, noses, and masticatory muscle attachments, and relatively less protrusive palettes and anterior mandibles. Despite a small sample size (N = 40), the authors' results are statistically significant (P approximately 0.001 overall) for both of the main factors, sex and ethnicity, separately.

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.

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.

Septoplasty for obstructive sleep apnea in infants after cleft lip repair.

A neonate with a unilateral cleft lip and palate usually presents with a deviated nasal septum due to the asymmetric bony base associated with cleft palate. Prior to repair, the facial cleft offers a wide open breathing passage despite the septal deviation. Cleft lips are traditionally repaired in neonates at about 3 months of age. These patients usually do not present with significant symptoms of nasal obstruction following repair, except in unusual cases. Severe septal deviation may cause obstructive sleep apnea. Repair of septal deformities in children is controversial due to the potential alteration of facial growth. We present two patients with documented obstructive sleep apnea that began after cleft lip repair. Conservative surgical correction of the septal deviation resulted in relief of the sleep apnea.

A classification of plagiocephaly utilizing a three-dimensional computer analysis of cranial base landmarks.

Plagiocephaly is a term commonly used to describe congenital forehead asymmetry. Previous classification systems based on the various etiologies of dysmorphic crania have been used in an effort to categorize the patients into groups and to assist in treatment planning. The system most commonly used today was described by Bruneteau and Mulliken in 1992. The authors separated frontal plagiocephaly into three types: synostotic, compensational, and deformational. The present study was undertaken in order to define a simple system for classifying plagiocephaly based on Bruneteau and Mulliken's system using the patients' preoperative craniofacial computed tomography scans. The involvement of the entire coronal ring in synostotic plagiocephaly led to the choice of 20 skull base landmarks as the basis of the analysis. Nine lateral landmarks (the superior orbital fissure, the optic foramen, the zygomatic arch, the greater palatine foramen, the foramen ovale, the mastoid tip, the hypoglossal canal, the external auditory canal, and the internal auditory canal) and two midline landmarks (the crista galli and the internal occipital protuberance) were used. The changes that occurred in these landmarks were analyzed in 30 patients. The results demonstrated that Bruneteau and Mulliken's classification system underestimated the number of different subtypes of plagiocephaly. As a result, three major types of frontal plagiocephaly and several different subtypes based on the different etiologies were described. Type I plagiocephaly includes plagiocephaly resulting from cranial suture synostosis. Type II includes those with a nonsynostotic etiology. Type III describes patients with craniofacial microsomia-associated plagiocephaly. Statistical analysis was unavailable because of the small number of patients in each subtype. With a larger number of patients, we hope to refine this system for use by the surgeon in preoperative diagnosis and surgical planning. The analysis is unique in its ability to quantitate changes from normal on the x-, y-, and z-coordinates, and therefore allows for identification of both horizontal (frontal bone deviation) and vertical (ear shear) growth disturbances.

Introduction of an intraoral bone-lengthening device.

It has been demonstrated that distraction osteogenesis is an effective clinical tool with applications for the entire human skeleton. Therapeutic exploitation in the correction of the asymmetrical and hypoplastic mandible has been reported previously. However, the main criticism of this technique has been the residual cutaneous scars resulting from the surgical incision and the path of the expansion device. In order to obviate the need for skin incisions, we have developed and demonstrated the feasibility of a miniaturized mandibular bone lengthener that is suitable for intraoral placement. Ten growing mongrel dogs were studied. Under general anesthesia, a buccal mucosal incision was made along the oblique line, and the body and ramus of the mandible were exposed in a supraperiosteal plane. Two 2.0-mm half-pins were placed percutaneously in the area of the angle of the mandible, and two other pins were placed 3.5 cm anteriorly. The clamps of the lengthening device were then attached to the half-pins in an intraoral position. An osteotomy was made by connecting percutaneously made drill holes between the clamps in a line positioned posterior to the third molar. The mucosa was closed loosely over the device. Distraction was commenced on the eighth postoperative day. The results were assessed with pre- and post-lengthening photographs, cephalograms, and CT scans with three-dimensional reconstruction. All animals demonstrated a contralateral cross-bite. The newly developed bone also was examined histologically. The intraoral method of mandibular lengthening offers the same advantages of extraoral lengthening but without the need for a cutaneous incision and resulting scar.

Twenty-year experience with early surgery for craniosynostosis: I. Isolated craniofacial synostosis--results and unsolved problems.

Early surgery for isolated craniosynostosis is designed to improve morphology, to prevent functional disturbances, and equally important, to enhance the psychosocial development of the child. As the first of a two-part series, 104 patients with isolated craniofacial synostosis were retrospectively analyzed. Diagnoses included bilateral coronal (10), unilateral coronal (57), metopic (29), and sagittal synostosis (8). All patients underwent primary fronto-orbital advancement-calvarial vault remodeling procedures at less than 18 months of age (mean 8.1 months). Thirteen percent of patients (14) required a secondary cranial vault operation (mean age 22.6 months) to address residual deficits in craniofacial form. Perioperative complications were minimal (5.0 percent), and there was no mortality. Average length of postoperative follow-up was 46.0 months. By the classification of Whitaker et al., which assesses surgical results, 87.5 percent of patients were considered to have at least satisfactory craniofacial form (category I-II) at latest evaluation. Overall rates of hydrocephalus, shunt placement, and seizures (3.8, 1.0, and 2.9 percent, respectively) were low. Among the isolated craniosynostoses, unilateral coronal synostosis/plagiocephaly poses the most complex problems, including vertical orbital dystopia, nasal tip deviation, and residual craniofacial asymmetry; there is also a wide spectrum of findings and growth patterns in this subgroup.

Twenty-year experience with early surgery for craniosynostosis: II. The craniofacial synostosis syndromes and pansynostosis--results and unsolved problems.

As the second of a two-part series, 76 patients with pansynostosis and craniofacial synostosis syndromes were retrospectively analyzed. Diagnoses included pansynostosis (7), craniofrontonasal dysplasia (8), and Apert (24), Crouzon (15), and Pfeiffer (15) syndromes. All patients underwent primary fronto-orbital advancement-calvarial vault remodeling procedures at less than 18 months of age (mean 6.1 months). Twenty-eight patients (36.8 percent) required a secondary cranial vault operation (mean age 28.4 months). Additionally, a major tertiary procedure was necessary in 5 patients to deal with persistent unacceptable craniofacial form. To address the associated finding of midface hypoplasia, 64.8 percent (n = 35) of patients underwent Le Fort III midface advancement or had that procedure recommended for them. The remainder were awaiting appropriate age for this reconstruction. The more extensive pathologic involvement of the pansynostosis and craniofacial syndrome group is illustrated. As compared with the isolated craniofacial synostosis group previously reported, the incidence of major secondary procedures (36.8 versus 13.5 percent), perioperative complications (11.3 versus 5.0 percent), follow-up complications (44.7 versus 7.7 percent), hydrocephalus (42.1 versus 3.9 percent), shunt placement (22.4 versus 1.0 percent), and seizures (11.8 versus 2.9 percent) was significantly increased. Complex problems including those of increased intracranial pressure, airway obstruction, and recurrent turricephaly or cranial vault maldevelopment are repeatedly encountered. In addition, that early fronto-orbital advancement-cranial vault remodeling failed to promote midface development and hypoplasia of this region is almost a consistent finding in the craniofacial syndromic group. The average length of postoperative follow-up was 6 years. According to the classification of Whitaker et al., which assesses surgical results, 73.7 percent of patients were considered to have at least satisfactory craniofacial form (category I-II) at latest evaluation. An algorithmic approach to the treatment of all patients with craniosynostosis is presented utilizing early surgical intervention as the key element.

An analysis of extradural dead space after fronto-orbital surgery.

This study was undertaken to evaluate several concerns regarding the extradural space resulting from elective fronto-orbital advancement or frontal sinus cranialization techniques. The questions are (1) Do infants undergoing these techniques have the potential to obliterate this space at an accelerated rate, e.g., within 1 or 2 days? (2) Do adults have any potential to obliterate the space? (3) Do children obliterate the space like infants or like adults? (4) What is the specific time sequence for dead-space obliteration? Twenty patients ranging in age from 6 months to 35 years were studied before and after fronto-orbital advancement. The patients were divided into three groups: (1) infants (up to 15 months), (2) children (up to 9 years), and (3) adults (9 years and beyond). Postoperative intracranial dead space was assessed by serial CT scans. Ten patients had CT scans more than 14 days after surgery. These data demonstrate that intracranial dead space in infants is obliterated in a delayed fashion. Children tend to obliterate intracranial dead space in a manner similar to that of infants. Adults are able to obliterate the space over a longer, but finite, period of time as compared with infants and children. Part of the mechanism responsible for obliteration of the postoperative space may be enlargement of the ventricular system.

Landmarks in three dimensions: reconstruction from cephalograms versus direct observation.

A method for generating three-dimensional landmark locations from routine biplane pairs of cephalograms was previously introduced. In this article we compare the locations thus reconstructed to the same configurations as directly recorded through a redundant network of interpoint distances measured with calipers. Six mandibular landmarks were located by both methods on each of 10 dry skulls. With the caliper distances is associated a standard error that can be estimated without explicit remeasurement by the method of "adjustment of networks" familiar from surveying. These standard errors are consistent with the remeasurement error both of the caliper measurements and of the stereolocation from cephalograms; the methods appear to have the same precision, about 0.4 mm per distance. The bias (systematic shift) of the biplane reconstructions with respect to the points used for laying down the calipers may be estimated by regressions of distance discrepancies on the direction cosines of the separations between pairs of landmarks. The caliper tips placed condylion approximately 10 mm medially and a bit forward of where we chose to reconstruct it from biplane cephalograms. The caliper locations of gonion average about 1.6 mm back of their cephalometric position, while those at menton and lower incisal edge are forward by some 1.4 mm. We conclude that the biplane reconstruction (the "three-dimensional cephalogram") is sufficiently accurate for routine clinical and surgical application.

Aplasia cutis congenita of the scalp: issues in its management.

Two cases of aplasia cutis congenita with associated bony defects are presented to highlight the dangers of delayed coverage of exposed dura. Management of one case was complicated by repeated local and systemic sepsis and the other by repeated, life-threatening hemorrhage. Early, definitive coverage of these defects using full thickness skin flaps avoids the risks of hemorrhage and should, if the recipient bed is clean, lessen the incidence of complicating sepsis.