Modified technique of presurgical infant maxillary orthopedics for complete bilateral cleft lip and palate

This article introduces technical modifications to the conventional presurgical infant maxillary orthopedics device for newborns with complete bilateral cleft lip and palate, providing procedural simplicity and efficiency as well as therapeutic efficacy. The modifications incorporate a wax block-out on the stone model prior to device fabrication in a manner that the need for periodic acrylic addition and removal is not required, and thus eliminates the risk of natural maxillary growth restriction during infant maxillary orthopedics treatment. The premaxilla is completely excluded from the acrylic palatal plate and is repositioned primarily by the bilateral labial tape alone. In addition, nasal stent wires are installed on the same day of the palatal plate delivery to establish a tripod-like retention mechanism for the intraoral device to be able to replace the conventional mechanical lock-type retention methods. Applying these modifications, infant maxillary orthopedics treatment objectives for bilateral cleft lip and palate can be successfully achieved within 8 weeks of treatment, and the definitive primary cleft lip repair can be performed within 3-4 months of infant maxillary orthopedics treatment at our Center.     

Orthognathic surgery in cleft patients treated by early bone grafting

For the past 25 years at Children's Memorial Hospital in Chicago a protocol has been followed for complete clefts that involves placement of an infant maxillary orthopedic appliance prior to lip closure, surgical closure of the lip, autogenous split-rib grafts to the alveolus to stabilize maxillary segments, and palatal closure, generally within the first year of life. The oldest 36 patients whose skeletal growth was for all practical purposes finished have been followed to determine the need for and type of orthognathic surgery. Of the total sample, 8 patients (22.2 percent) required some type of sagittal orthognathic surgery (1 patient in this group also required vertical maxillary alignment) and 2 patients required maxillary augmentation only in the form of an onlay graft. This report may serve as a baseline for others who wish to report on the incidence and type of orthognathic surgery in their cleft palate centers.     

新生儿唇腭裂术前矫治方法与技术的临床探讨

目的:评价新生儿唇腭裂术前进行鼻-牙槽突-腭畸形矫治方法的疗效。方法:对28例单双侧唇腭裂新生儿进行术前鼻-牙槽突-腭畸形矫治治疗,在面部确定基点,利用数码相机拍射照片,通过image-Pro Plus5.1软件测量相关距离及角度,测量治疗前后的鼻小柱倾斜度、鼻小柱长度、鼻孔宽度和鼻孔高度。治疗前后取上颌石膏模型进行牙槽骨裂隙宽度的测量,比较矫治治疗前后腭部裂隙最大处及牙槽突裂隙的变化。结果:鼻小柱倾斜度平均减小27.11°,鼻孔宽度平均减小4.39 mm(单)或5.29 mm(双),鼻孔高度平均增加2.56 mm(单)或3.57 mm(双),牙槽突裂隙平均减少3.18 mm,腭部裂隙最大处平均减少5.77 mm。治疗前后的各项差异均有统计学意义(P0.05),鼻塌陷畸形程度也得到显著改善。结论:术前进行鼻-牙槽突-腭畸形矫治治疗可为唇腭裂患者手术治疗创造有利条件,提高其整复效果。     

A comparison of the effects of the Latham-Millard procedure with those of a conservative treatment approach for dental occlusion and facial aesthetics in unilateral and bilateral complete cleft lip and palate: part I. Dental occlusion

The purpose of this study was to compare the effect of the Latham-Millard presurgical orthopedics, gingivoperiosteoplasty, and lip adhesion protocol with conservative treatment (nonpresurgical orthopedics without gingivoperiosteoplasty) for palatal and dental occlusion in complete bilateral and complete unilateral cleft lip and palate. All patients were from the South Florida Cleft Palate Clinic. A retrospective dental occlusal study was conducted using serial dental casts that had been taken of patients from birth to 12 years of age. All surgical procedures, except for the secondary alveolar bone grafts in the conservative, nonpresurgical orthopedics group, were performed by D. Ralph Millard, Jr. Ralph Latham supervised the presurgical orthopedics cases. Samuel Berkowitz collected and analyzed all the serial records from 1960 to 1996. Among the patients with complete unilateral cleft lip and palate, 30 patients were treated with presurgical orthopedics, gingivoperiosteoplasty, and lip adhesion (the Latham-Millard protocol) and 51 patients were treated conservatively (i.e., nonpresurgical orthopedics without gingivoperiosteoplasty). Among the patients with complete bilateral cleft lip and palate, 21 patients were treated with the Latham-Millard protocol and 49 patients were treated conservatively. Conservative treatment was performed between 1960 and 1980. In patients with bilateral cleft lip and palate, a head bonnet with an elastic strip was used to ventroflex the protruding premaxilla. In all patients (unilateral and bilateral cleft), lip adhesion was performed at 3 months followed by definitive lip surgery at 6 to 8 months and palatal cleft closure between 18 and 24 months of age, in most cases. The Latham-Millard procedure was performed from 1980 to 1996; in bilateral cleft patients, it involved the use of a fixed palatal orthopedic appliance to bodily retract the protruding premaxilla and align it within the alveolar segments soon after birth. In all patients (unilateral and bilateral cleft), palatal alignment was also followed by gingivoperiosteoplasty and lip adhesion. Definitive lip surgery was performed between 6 and 8 months of age, and palatal closure was performed between 8 and 24 months of age using the von Langenbeck procedure with a modified vomer flap. All of the study participants had cleft lips and palates of either the unilateral or bilateral type; the unilateral and bilateral groups were further subdivided based on whether they had received the Latham-Millard protocol or the conservative treatment. It was then determined how many in each of these four basic groups had either anterior or buccal crossbites at four different age levels, when they were approximately 3, 6, 9, and 12 years of age. Although several children entered the study at or just before age 6, every patient in the 9-year-old and 12-year-old sample groups had been in the 6-year-old group and all of the 12-year-olds had been included in the immediate preceding age sample. Two-by-two chi-square tests were carried out within each cleft type (unilateral or bilateral) at each of the four age levels separately, to test whether the treatment groups (protocol versus conservative) differed in the frequency of cases with a given kind of crossbite (rather than not having that kind of crossbite). At every age level, a greater percentage of patients treated with the Latham-Millard protocol developed crossbites than did those treated more conservatively. This difference existed for both the anterior and buccal crossbites and for both unilateral and bilateral clefts. Chi-square tests of the treatment differences in crossbite frequency showed that in three quarters of the Latham-Millard protocol versus conservative treatment comparisons (12 out of 16), a significantly greater frequency of crossbite cases occurred after the Latham-Millard protocol treatment as compared with after the conservative procedure. The chi-square values for the differences in outcome between the two kinds of treatment procedures were greater for the anterior crossbites than for the buccal crossbites, suggesting that the Latham-Millard protocol, relative to the conservative method, was more likely to have an adverse effect on the anterior crossbites than on the buccal crossbites. For those patients born with a bilateral cleft, the differences in crossbite frequency between the protocol and the conservative treatment were statistically significant for patients with an anterior crossbite but not for patients with a buccal crossbite. The analysis shows that in complete bilateral and unilateral cleft lip and palate, the frequency of the anterior crossbite and (except for ages 3 and 12) the buccal crossbite is significantly higher with the Latham-Millard presurgical orthopedics, gingivoperiosteoplasty, and lip adhesion protocol compared with the conservative, nonpresurgical orthopedics without gingivoperiosteoplasty treatment. The exception in the bilateral buccal case may be attributed to the small experimental sample size, which brings down the confidence level.     

Orthopedic intrusion of premaxilla with distraction devices before alveolar bone grafting in patients with bilateral cleft lip and palate

Surgical repositioning of the downward displaced premaxilla in bilateral cleft lip and palate patients remains a controversial and perplexing issue because of its detrimental effects on the growth of the premaxilla. The purpose of this prospective clinical study was to introduce and evaluate the treatment results of an innovative technique for nonsurgically intruding the downward displaced premaxilla. Eight consecutive cases of bilateral cleft lip and palate at the age of mixed dentition were included for the correction of their premaxillary deformities. A pair of intraoral tooth-borne distraction devices was used for the orthopedic intrusion. Serial lateral and posteroanterior cephalometric radiographs were taken periodically for evaluating the growth of the premaxilla 1 year before the intrusion, changes during the intrusion, and growth/relapse up to 1 year after the intrusion. There was no overgrowth of the premaxilla or overeruption of the maxillary incisors during the 1-year observing period before the orthopedic intrusion. The treatment results revealed that the downward displaced premaxillae were all corrected within 1 month. Cephalometrically, 46 percent of the correction resulted from a true orthopedic intrusion and another 54 percent from a dentoalveolar effect in which the maxillary incisors were intruded and the premaxillary dentoalveolus was shortened. The cephalometric evaluations also implied that what occurred during the orthopedic intrusion was mostly the sutural contraction osteogenesis/osteolysis in the vomeropremaxillary suture combined with slightly mechanical upward displacement of the vomeronasal septum complex and nasal bones. The orthopedic intrusion of the premaxilla with distraction devices is an effective nonsurgical method for correcting the downward displaced premaxilla before alveolar bone grafting in patients with bilateral cleft lip and palate, and the results remained stable after 1 year.     

A long-term retrospective outcome assessment of facial growth,secondary surgical need,and maxillary lateral incisor status in a surgical-orthodontic protocol for complete clefts

In 1965, the cleft palate team at Children's Memorial Hospital embarked on a new surgical-orthodontic protocol in the habilitation of newborn complete cleft lip and palate cases. It brought the orthodontic effort into focus at birth and in planned sequence to correspond with the surgical procedures of lip closure, maxillary alveolar stabilization by means of an autogenous graft of the authors' design, and complete palate closure, all within the first year of life. The purpose of this investigation is threefold: first, to review the authors' previous publications and assess growth, secondary surgical need, and lateral incisor status of teeth adjacent to the cleft in a series of patients who have all followed a precise, early surgical/orthodontic protocol; second, to compare these cases with other collaborative studies wherein this protocol was not used; and third, to report on an additional 82 cases with regard to secondary surgical need and the status of teeth adjacent to the cleft. Methods of assessment have included cephalometric radiography, periapical and occlusal dental radiography, computer-assisted tomography, plaster cast analysis, and intraoral and extraoral photography. The authors have demonstrated, along with other collaborative studies, that there is growth as good as other similar samples wherein there was no primary osteoplasty. In addition, the authors found their incidence of orthognathic surgery to be 18.29 percent; pharyngoplasty, 3.65 percent; and oronasal fistulas requiring surgical closure, 29.27 percent. In the case of unilateral complete clefts, 53.13 percent of those lateral incisors present adjacent to the cleft area were usable, and in bilateral cases, 57.77 percent were usable. The authors remain convinced after more than 35 years of following this successful protocol that early maxillary orthopedics and their technique of primary osteoplasty in planned sequence with lip and palate closure can produce a more favorable alignment of maxillary growth potential and, with comprehensive orthodontic treatment, can lead to teeth in a better overall occlusion than if these procedures had not been undertaken.     

The case for early bone grafting in cleft lip and palate: a second report

In 1982, the first long-term study of our early bone-grafting and infant maxillary orthopedic approach to newborn complete clefts of the lip, alveolus, and palate was published. The protocol and sequence of procedures were shown on the first 16 consecutively treated orthodontic patients, with a mean age of 14 years. Cephalometric analysis evaluated anteroposterior and vertical facial growth. This report follows the next 37 consecutively treated individuals in a similar manner and includes not only 20 complete unilateral clefts, but also 17 complete bilateral clefts. Results, when evaluated alone and in comparison with the original series, show once again that there are no adverse growth restraints and that early primary bone grafting in our protocol leads to teeth in better overall occlusion than if it had not been undertaken.     

The progressive changes of nasal symmetry and growth after nasoalveolar molding: a three-year follow-up study

The purpose of this study was to assess the progressive changes of nasal symmetry, growth, and relapse after presurgical nasoalveolar molding and primary cheiloplasty in unilateral complete cleft lip/palate infants. Twenty-five consecutive complete unilateral cleft lip/palate infants were included. All the infants underwent nasoalveolar molding before primary cheiloplasty. Standard 1:1 ratio basilar photographs were taken before and after nasoalveolar molding, 1 week after cheiloplasty, and yearly for 3 years. Linear measurements were made directly on the photographs. The results of this study revealed that the nasal asymmetry was significantly improved after nasoalveolar molding and was further corrected to symmetry after primary cheiloplasty. After the primary cheiloplasty, the nasal asymmetry significantly relapsed in the first year postoperatively and then remained stable and well afterward. The relapse was the result of a significant differential growth between the cleft and noncleft sides in the first year postoperatively. To compensate for relapse and differential growth, the authors recommend (1) narrowing down the alveolar cleft as well as possible by nasoalveolar molding, (2) overcorrecting the nasal vertical dimension surgically, and (3) maintaining the surgical results using a nasal conformer.     

Nasoalveolar Molding in Cleft Care—Experience in 40 Patients from a Single Centre in Germany

Nasoalveolar molding (NAM) has gained wide acceptance and evidence in cleft therapy. However, standardized treatment protocols and experiences recorded from European centres are lacking. The results of 40 infants with cleft lip and palate treated with presurgical NAM according to the Grayson technique were analyzed. Standardized parameters of cleft width and nasal symmetry were measured in pre- and posttreatment plaster casts and in digitalized 3-dimensional STL models. Statistical analyses were performed by using Student´s t-test in a per-protocol manner. 27 out of 40 infants completed NAM and were analyzed. In 13 patients NAM was either temporarily interrupted or terminated prematurely due to skin irritations or lack of parental support. These cases were excluded from statistical analysis, resulting in a drop-out rate of 32.5%. Intersegmental alveolar distance (ISAD), intersegmental lip distance (ISLD), nostril height (NH), nostril width (NW) and columella deviation angle (CDA) were significantly changed in unilateral cleft lip and palate (UCLP) (n = 8). In unilateral cleft lip (UCL) (n = 9), only ISLD, NH and CDA were significantly changed. ISAD of the right and left side, ISLD of the right and left side, premaxilla deviation angle, nostril height and columella length were changed significantly in bilateral cleft lip and palate (BCLP) cases (n = 10). NAM is a suitable presurgical treatment modality. A positive effect has been seen in UCLP and BCLP infants, as compared with their birth status.     

Radiographic analysis of the midface of a stillborn infant with a unilateral cleft lip and palate

The midface of a full-term stillborn infant with a right complete unilateral cleft lip and palate was studied with plain-film radiography and tomography, xeroradiography, and computerized axial tomography. Gross skeletal and soft-tissue deficiencies on the cleft side were evident as compared to the noncleft side and involved the entire bony maxillary complex and antrum, the orbit, and the nasal pyramid and intranasal structures. The area on the cleft side was 19 percent less than the noncleft side, and the maximal anteroposterior dimension was 16 percent less. The cleft bony palatal shelf was 12.5 mm wide compared to 20 mm on the noncleft side. The findings demonstrate the deficient and abnormal functional matrix inherent in the cleft condition.     

Binderoid complete cleft lip/palate

A small subset of infants with complete cleft lip/palate look different because they have nasolabiomaxillary hypoplasia and orbital hypotelorism. The authors' purpose was to define the clinical and radiographic features of these patients and to comment on operative management, classification, and terminology. The authors reviewed 695 patients with all forms of incomplete and complete cleft lip/palate and identified 15 patients with nasolabiomaxillary hypoplasia and orbital hypotelorism. All 15 patients had complete labial clefting (5 percent of 320 patients with complete cleft lip/palate), equally divided between bilateral and unilateral forms. The female-to-male ratio was 2:1. Of the seven infants with unilateral complete cleft lip/palate, one had an intact secondary palate and all had a hypoplastic septum, small alar cartilages, narrow basilar columella, underdeveloped contralateral philtral ridge, ill-defined Cupid's bow, thin vermilion-mucosa on both sides of the cleft, and a diminutive premaxilla. Of the eight infants with bilateral complete cleft lip, one had an intact secondary palate. The features were the same as in patients with unilateral cleft, but with a more severely hypoplastic nasal tip, conical columella, tiny prolabium, underdeveloped lateral labial elements, and small/mobile premaxilla. Central midfacial hypoplasia and hypotelorism did not change during childhood and adolescence. Intermedial canthal measurements remained 1.5 SD below normal age-matched controls. Skeletal analysis (mean age, 10 years; range, 4 months to 19 years) documented maxillary retrusion (mean sagittal maxillomandibular discrepancy, 13.7 mm; range, 3 to 17 mm), absent anterior nasal spine, and a class III relationship. The mean sella nasion A point (S-N-A) angle of 74 degrees (range, 65 to 79 degrees) and sella nasion B point (S-N-B) angle of 81 degrees (range, 71 to 90 degrees) were significantly different from age-matched norms ( = 0.0007 and = 0.004, respectively). The ipsilateral central and lateral incisors were absent in all children with unilateral cleft, whereas a single-toothed premaxilla was typically found in the bilateral patients. Several modifications were necessary during primary nasolabial repair because of the diminutive bony and soft-tissue elements. All adolescent patients had Le Fort I maxillary advancement and construction of an adult nasal framework with costochondral or cranial graft. Other often-used procedures were bony augmentation of the anterior maxilla; cartilage grafts to the nasal tip and columella; and dermal grafting to the median tubercle, philtral ridge, and basal columella. Infants with complete unilateral or bilateral cleft lip/palate in association with nasolabiomaxillary hypoplasia and orbital hypotelorism do not belong on the holoprosencephalic spectrum because they have normal head circumference, stature, and intelligence, nor should they be referred to as having Binder anomaly. The authors propose the term cleft lip/palate for these children. Early recognition of this entity is important for counseling parents and because alterations in standard operative methods and orthodontic protocols are necessary.     

Risk factors for poor dental arch relationships in young children born with unilateral cleft lip and palate

To identify risk factors for poor dental arch relationships in children with unilateral cleft lip and palate in the United Kingdom, the authors performed a cross-sectional outcome study with retrospective data capture of treatment histories in children under the care of 44 cleft teams in the United Kingdom. The study sample comprised 238 children born with nonsyndromic complete unilateral cleft lip and palate between April 1, 1989, and March 31, 1991, who were between 5.0 and 7.7 years of age (mean age, 6.5 years) at the time of data collection. The Five-Year-Old Index was used to rank dental arch relationships from dental study models. Velopharyngeal insufficiency was assessed with the use of the Cleft Audit Protocol for Speech. An independent panel recorded surgical treatment histories from the clinical notes. There was no association between the technique and the timing of primary repair, the experience of the surgeon, or presurgical orthopedics and dental arch relationships. Secondary velopharyngeal surgery was independently associated with poor outcome (OR, 4.14; 95 percent CI, 1.6 to 10.7; p = 0.003). Primary nasal repair was protective (OR, 0.47; 95 percent CI, 0.23 to 0.93; p = 0.031) against poor dental arch relationships. Secondary velopharyngeal surgery and primary nasal repair were found to be independently associated with dental arch relationship outcomes in young children with unilateral cleft lip and palate in the United Kingdom.     

Cleft palate repair by double opposing Z-plasty

In an attempt to improve speech results following palate repair while allowing adequate maxillary growth, a palatoplasty using two opposing Z-plasties of the soft palate, one of the oral and one of the nasal layers, has been used in 22 infants. Eight patients had unilateral cleft lip and palate, eight had bilateral cleft lip and palate, and six had cleft palate. The Z-plasties facilitate effective dissection and redirection of the palatal muscles to produce an overlapping muscle sling and lengthen the velum without using tissue from the hard palate, which permits hard palate closure without pushback or lateral relaxing incisions. Of the 20 children old enough for speech evaluation, 18 have no velopharyngeal insufficiency. Two have very mild velopharyngeal insufficiency. None has required a pharyngeal flap.     

A model for fetal cleft lip repair in lambs.

Fetal wounds heal without inflammation and scar formation. This phenomenon may, in the future, be applicable to human cleft lip and palate repair. However, extensive experimental work must first be done to document the benefits of in utero repair. We developed a large animal model for creation and repair of a complete cleft lip and alveolus using fetal lambs. The cleft lip and alveolus deformity was created in eight 75-day-gestation fetuses (term = 145 days) and either repaired in three layers or left unrepaired. There were four sham-operated fetuses, and all animals were alive at harvest. Repaired, unrepaired, and control fetuses were harvested at 7, 14, 21, and 70 days following surgery. The unrepaired fetuses demonstrated a complete cleft lip and alveolus with an oronasal fistula. The maxilla was asymmetrical, with the greater segment deviated toward the cleft and with decreased anterior maxillary width. In contrast, repaired cleft lip and alveolus animals showed no scar, normal thickness of the lip, and a symmetrical maxilla. Histologic analysis of the repaired wounds showed evidence of tissue regeneration without scar formation. The results of this preliminary study indicate that the fetal lamb cleft lip and alveolus model is technically feasible with an excellent survival rate. Healing occurs without scar formation. In the repaired animals, the maxilla was symmetrical. This model will be used to document facial growth following in utero repair of a cleft lip and alveolus.     

Effect of Veau-Wardill-Kilner type of cleft palate repair on long-term midfacial growth

There is a common concern that the Veau-Wardill-Kilner type of cleft palate repair causes extensive denudation of the palate, resulting in inhibition of maxillary growth. The evidence for this belief is equivocal in the literature. The authors present some long-term results of this technique from a pure sample of nonsyndromic complete unilateral cleft lip and palate patients operated on by a single intermediate-volume cleft surgeon over a period of 25 years. Twenty-five patients, all born between 1977 and 1989, met the above inclusion criteria. Their age at the time of collection of study models and cephalograms was 9 to 17 years (average, 12 years). Midfacial growth was studied using 12-year dental models and lateral cephalograms taken before definitive orthodontic treatment. These were evaluated using the GOSLON Yardstick and digital cephalometric analysis. The final GOSLON results show that 72 percent of the patients had a good or satisfactory outcome, with a GOSLON score of 1, 2, or 3, and only 28 percent ended with a poor score of 4 or 5. The poor sensitivity of cephalometrics in discerning statistically significant differences was highlighted by the huge overlaps observed in the 95 percent confidence interval graph of mean sella-nasion-subspinale angle (S-N-A) values when comparing the results of the Eurocleft centers with those of the authors' center. The results suggest that satisfactory long-term midfacial growth can be obtained with Veau-Wardill-Kilner cleft palate repair.     

Early palatal changes in complete and incomplete cleft lip and/or palate.

Early palatal development in various complete and incomplete forms of cleft lip and/or palate (CLP) was studied from birth to 3 months of age by means of dental casts. Palatal morphology (shape) and dimensions--based on reproducible reference points--were determined in a group of 128 CLP children and 68 normal children who served as controls. Substantial normal palatal growth during the first 3 months of life was observed. Round arch forms changed into oval arch forms. Growth mainly takes place in the sagittal direction (+4 mm) (transverse: +1 mm). Palates of CLP children differed significantly dependent on the type of cleft and whether the cleft was complete or incomplete. Cleft lip and alveolus children and bilateral cleft lip and palate children had more elongated palatal arches, whereas unilateral cleft lip and palate children and cleft palate children had wider palatal arches than the control group. Incomplete clefts differed from the control group in the same direction as their complete cleft forms, though less distinctly. Preoperative orthopedics used in CLP patients does not stimulate growth. On the contrary, it even restricts growth.     

From birth to maturity: a group of patients who have completed their protocol management. Part I. Unilateral cleft lip and palate

The optimal management of the cleft lip and palate patient from birth to completion of treatment presents a formidable challenge to the plastic surgeon and the associated health care system. The multidisciplinary team approach for the management of these patients is widely accepted. However, a paucity of literature exists discussing specific protocol management, interventions, and the long-term outcomes of patients who have completed a strict treatment protocol with a consistent multidisciplinary team. The aim of this study was to present the details of the specific management protocol at the Australian Craniofacial Unit for cleft lip and palate patients and to present a group of patients who have completed this specific protocol and discuss the details of their long-term care. During a 28-year period from 1974 to 2002, the records of 337 patients treated for unilateral cleft lip and palate were evaluated. Of these 337 patients, 22 have completed the same specific protocol management. The same surgeon (David, the senior author) has been responsible for performing all operative interventions and for overseeing the care of each of the 22 patients, ensuring that the treatment protocol has been executed appropriately and without deviation. The interventions and outcomes were analyzed on the basis of speech, hearing, nasal airway, occlusion, psychosocial adjustment, and appearance. Because of the large volume of data and potential differences in outcomes, the authors' intention is to present this as part I of a four-part series beginning with unilateral cleft lip and palate. The results of isolated cleft palate, isolated cleft lip, and bilateral cleft lip and palate will be presented as parts II, III, and IV, respectively. Speech results were assessed as normal speech, mild abnormality, or severe abnormality by objective measures, and intervention for velopharyngeal insufficiency was noted. Seventeen patients were rated as having normal speech. Four patients were rated as having mild speech abnormality, one patient was rated as having severe speech abnormality, and seven patients required surgery for velopharyngeal insufficiency. Hearing results were measured objectively, and good hearing results were obtained in 18 cases. Five patients required tympanoplasty. All patients required alveolar bone grafting. The high Le Fort I osteotomy was performed in six cases. Bimaxillary surgery was performed in one case. Of all the patients assessed from birth to maturity, 13 required between three and five surgical interventions, and nine required six operations or more. Further details and photographs of preoperative and postoperative examples are provided.     

Cleft lip and palate treated by presurgical orthopedics, gingivoperiosteoplasty, and lip adhesion (POPLA) compared with previous lip adhesion method: a preliminary study of serial dental casts.

Comparing the treatment protocols for cleft lip and palate requires a study of facial growth and development. Serial orthodontic study models aid in delineating the effects of surgery on maxillary mandibular relationships. In 1978, a new protocol, POPLA (presurgical orthopedics followed by periosteoplasty and lip adhesion), was developed and put into practice. This article compares the results obtained using POPLA (group I) with those using the previous method (lip adhesion alone) (group II) for cases of unilateral and bilateral clefts. Complete orthodontic study models were available for 124 patients, 63 in group I and 61 in group II. Specific timing sequences and surgical details are discussed. Dental cast analyses evaluated alveolar gap, arch width, anteroposterior distance, incisor crossbite (single tooth and multiple tooth), and buccal crossbite (single and multiple tooth) at birth and at 3, 6, and 9 years of age. X-ray studies evaluated the bony bridge. Additional comments are made regarding the incidents and effects of pharyngeal flaps and bone grafts on maxillary-mandibular relationships. Results included less buccal crossbite in the POPLA group, with a wider transverse distance of the upper dental arch. There was a greater frequency of anterior crossbite in the POPLA group, and the anteroposterior distance was shorter at 6 years of age but less so by age 9. This may be because of the different orthodontic care received by the patients in the two groups. In conclusion, the POPLA approach achieves the main goal of moving the palate into a normal position and stabilizing the arch with a bony bridge that attracts teeth. It avoids the difficult anterior fistulae and presents a more symmetrical platform upon which the lip can be united and the nose can be corrected early.     

Accelerated Wound Closure In Vitro by Fibroblasts from a Subgroup of Cleft Lip/Palate Patients: Role of Transforming Growth Factor-α

In a fraction of patients surgically treated for cleft lip/palate, excessive scarring disturbs maxillary growth and dento-alveolar development. Since certain genes are involved in craniofacial morphogenesis as well as tissue repair, a primary defect causing cleft lip/palate could lead to altered wound healing. We performed in vitro wound healing assays with primary lip fibroblasts from 16 cleft lip/palate patients. Nine foreskin fibroblast strains were included for comparison. Cells were grown to confluency and scratch wounds were applied; wound closure was monitored morphometrically over time. Wound closure rate showed highly significant differences between fibroblast strains. Statistically, fibroblast strains from the 25 individuals could be divided into three migratory groups, namely “fast”, “intermediate”, and “slow”. Most cleft lip/palate fibroblasts were distributed between the “fast” (5 strains) and the “intermediate” group (10 strains). These phenotypes were stable over different cell passages from the same individual. Expression of genes involved in cleft lip/palate and wound repair was determined by quantitative PCR. Transforming growth factor-α mRNA was significantly up-regulated in the “fast” group. 5 ng/ml transforming growth factor-α added to the culture medium increased the wound closure rate of cleft lip/palate strains from the “intermediate” migratory group to the level of the “fast”, but had no effect on the latter group. Conversely, antibody to transforming growth factor-α or a specific inhibitor of its receptor most effectively reduced the wound closure rate of “fast” cleft lip/palate strains. Thus, fibroblasts from a distinct subgroup of cleft lip/palate patients exhibit an increased migration rate into wounds in vitro, which is linked to higher transforming growth factor-α expression and attenuated by interfering with its signaling.     

A long-term, controlled-outcome analysis of in utero versus neonatal cleft lip repair using an ovine model.

Successful open repair of a cleft lip in utero has the advantage of scarless wound healing in the fetus. Unfortunately, no long-term outcome studies have been performed to evaluate the efficacy of these repairs. Moreover, no study to date has compared the long-term results of an in utero cleft lip repair to a similar, control-matched, newborn cleft repair. This study was performed to evaluate the 9-month outcome of in utero cleft lip surgery compared with an identical cleft lip repair performed on infant lambs. In utero epithelialized cleft lips were created through an open hysterotomy in sixteen 65-day-old fetal lambs (term = 140 days) using methods described by Longaker et al. Eight of 16 animals underwent subsequent in utero repair of these clefts at 90 days gestational age. The repair of the remaining eight animals was delayed until 1 week postpartum. At 9 months, the animals were analyzed for changes in lip contour and for the degree of scarring by hematoxylin and eosin and Masson's trichrome collagen staining. Two animals in each group died from preterm labor. Of the animals that survived to term, all repaired lips had some degree of abnormality postoperatively. One of six lips repaired in utero dehisced before delivery. Three of six neonatal repairs dehisced in the first postoperative month. In the remaining animals with intact lip repairs, the vertical lip height on the repaired side was an average of 9 to 12 mm shorter than the normal lip in both the in utero and neonatally repaired animals. Phenotypically, the postnatally repaired animals had more lip distortion and visible notching. Histologically, the in utero repair was scarless and the neonatal repairs had scar throughout the entire vertical height of the lip with an associated loss of hair in this region. Maxillary growth was also evaluated. There was no inhibition of maxillary growth in the animals that underwent in utero cleft lip repair. However, in the neonatal repair group, significant maxillary retrusion was evident. Compared with the cleft side of the maxilla, horizontal growth was decreased by 11 percent (p = 0.01). Compared with the intrauterine repair group, there was a 17-percent decrease in horizontal maxillary width (p = 0.01). Straight-line in utero repair of a cleft lip produces a better long-term result in terms of maxillary growth than a similar repair performed postnatally in the ovine model. There was no diminution in maxillary growth in the animals treated in utero. Histologically, in utero repair of clefts was indeed scarless. However, both lip repairs produced lips that were significantly shorter than their contralateral noncleft sides. This degree of lip shortening would require a secondary lip revision, thereby defeating the purpose of performing an intrauterine repair. Comparisons now need to be made between in utero and neonatal repairs using a Millard-type rotation advancement technique before intrauterine treatment can be considered to be more beneficial than our current treatment modalities.