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A new approach to repairing cleft palate and acquired palatal defects with distraction osteogenesis.

Cleft palate (CP) is one of the most common human congenital deformities, and acquired palate defects after trauma or tumour resection are also common. In this study, distraction osteogenesis (DO) for CP and other palatal bone defects was evaluated. Twenty cats were assigned randomly to 3 groups of (1) 15, (2) 3 and (3) 2 cats. In groups 1 and 2, a rectangular ostectomy, in the posterior of the palatal bone shelf, was performed in the sagittal axis to establish the CP defect model. At the same time, a pure titanium intraoral distractor was fixed to molar teeth with brackets and to the palatal bone shelf across the defect with titanium miniscrews bilaterally. Four weeks later, a secondary transport disc (TD) osteotomy was performed, and gradual DO treatment started at 0.4mm twice a day, after 6 days of latency. DO was performed until the TD reached the opposite margin over the gap in 5-6 days. Three cats each of group 1 were killed at 2, 4, 6, 8 and 12 weeks after completion of DO. In group 2, the bone and soft-tissue defects were untreated until death 6 weeks later. Group 3 cats (control) were killed after 6 weeks. The TD successfully recombined with the opposite palatal bone stump, and proportional expansion of the overlay mucoperiosteal flap was achieved. Intramembranous bone formation was revealed: parallel collagen bundles gradually deposited on new bone trabeculae while the proliferative osteoblasts produced bone matrix. The bone defect was finally reconstructed by de novo osteogenesis. The control group was observed to have no spontaneous repairing. These results suggest that the CP defect was reconstructed by osteogenesis in situ, and the soft tissues expanded simultaneously to achieve functional correction. The intraoral distractor provided both effective distraction and stability.

Animals↗

Neonatal palatal cysts and their morphology in cleft lip and palate.

Palatal cysts (Epstein pearls) are occasionally noticed as white papules on the palatal mucosa of a neonate whilst having its 6-week postnatal check. We describe five cleft patients and the distribution characteristics of their palatal cysts, including the previously unrecorded observation of a submucous cleft palate with a double row of palatal cysts, one row on each side of the zona pellucida. The role of spatial morphology of palatal cysts in interpreting cleft pathogenesis is discussed.

Biopsy↗

Maxillary dental arch and occlusion in patients with unilateral cleft lip and palate treated with different delays in closure of the hard palate after early velar repair.

We wanted to find out if growth of the maxilla in 26 patients with unilateral cleft lip and palate (UCLP) was adversely affected by having the residual cleft of the hard palate repaired earlier than had been done previously in a 2-stage palatal closure protocol. The ages at repair of the hard palate of the present patients ranged from 38 to 89 months. Dental casts from ages about 3 years (before any repair of the hard palate), 5, 7, and 10 years of age were analysed. The results indicated that earlier repair of the cleft in the hard palate did not influence maxillary growth differently from the later repair.

Child↗

Distraction osteogenesis for lengthening of the hard palate: Part II. Histological study of the hard and soft palate after distraction.

To correct velopharyngeal incompetence, a new treatment concept was proposed in Distraction Osteogenesis for Lengthening of the Hard Palate: Part I (using lengthening of the hard and soft palate by distraction osteogenesis). Cephalometry and computed tomography showed successful elongation of the posterior hard palate with gradual calcification. Here the sequential use of fluorochrome markers (oxytetracycline, xylenol orange, DCAF [2,4-bis-N-N'-dicarboxymethyl aminomethyl fluorescein], and alizarin complexone) during the distraction and retention period is reported together with the histologic investigations using light and laser scan microscopy without prior demineralization. The experimental gap showed de novo osteogenesis in all dogs. The new bone was always in continuity with the original anterior and posterior palatal bone margins. It either bridged the experimental gap fully or left a small central zone of fibrous tissue, in which eventual ossification occurred. Several distinct zones could be distinguished: A small central zone was found with parallel strains of collagen fibers, oriented longitudinally in the direction of the distraction. Next to this zone a layer of undifferentiated mesenchymal precursor cells was seen in direct contact to newly formed bone. The next zone was coarse woven bone, showing a transition to mature lamellar bone through remodeling. No evidence of endochondral bone formation was found, i.e., all dogs showed exclusively intramembranous bone formation. The soft tissues showed no signs of alteration: in particular, there was no necrosis or scar formation. The soft tissues were not thinned but appeared to have followed the longitudinal displacement. In conclusion, gradual distraction osteogenesis of the hard palate could be a possible method for lengthening the palate to treat velopharyngeal incompetence.

Animals↗

Palatal length in cleft palate as a predictor of speech outcome.

In an attempt to predict which patients might benefit from primary posterior pharyngeal flaps done at the time of palatal repair, palatal length was assessed before palatal repair and the patient was placed in one of four categories. Patients with longer palates preoperatively had statistically better speech outcomes than patients with shorter palates. Statistical significance was found for most speech parameters. Information on presurgical palatal length can be useful in predicting which patients might profit from primary "pharyngoplasties."

Adolescent↗

Inhibition of human embryonic palatal mesenchymal cell cycle by secalonic acid D: a probable mechanism of its cleft palate induction.

OBJECTIVE: To assess the mechanism(s) of cleft palate induction by secalonic acid D (SAD) in human embryonic palatal mesenchymal (HEPM) cells and compare them with those evaluated in the murine embryonic palate. DESIGN: Effect of SAD on HEPM cell proliferation was studied by obtaining dose response curves for cell numbers, uptake of 3H-thymidine and the expression of proliferating cell nuclear antigen (PCNA). Effects of SAD on cell cycle were assessed by flowcytometry. Cell-labeling with 3H-glucosamine and immunoblot analysis were conducted to study SAD effects on the synthesis of glycosaminogycans (GAG) and the expression of fibronectin and tenascin, respectively. RESULTS: SAD induced a concentration-dependent decrease in HEPM cell number and 3H-thymidine uptake beginning at 0.1 microg of SAD/ml. Expression of PCNA and progression of cell cycle from G1 to S phase were inhibited following SAD exposure. Cell viability was significantly reduced only at 7.5 microg/ml of SAD or higher indicating that the reduction in cell numbers by SAD at lower concentrations is likely due to reduced proliferation and at higher concentrations due to both reduced proliferation and cell death. Synthesis of extra cellular matrix components (GAGs, fibronectin or tenascin) by HEPM cells, however, was not inhibited by SAD. CONCLUSION: The results of these studies confirmed those of our previous studies with mice and the MEPM cells that SAD may induce cleft palate by reducing numbers of palatal mesenchymal cells by inhibition of their proliferation thereby leading to a reduction in the size of the developing palate shelves.

Animals↗

H-2 and non-H-2 determined strain variation in palatal shelf and tongue adenosine 3':5' cyclic monophosphate: a possible role in the etiology of steroid-induced cleft palate.

The concentration of cAMP was measured in palatal shelves and tongues of 14.5-day old foetuses, 14.5-day old foetuses from steroid treated mothers, and 15.5-day old foetuses from four inbred lines of mice which represent the four possible combinations of two H-2 alleles and two residual genetic backgrounds. The incidence of spontaneous and steroid-induced cleft palate in these four strains was also determined. Analyses of variance of the cAMP data reveal that both the H-2 region and residual genetic background determine cAMP concentrations in both tissues and on both days of development. Similar analyses of cAMP concentrations after steroid treatments of the mother indicate that the interaction between H-2 and residual genetic background is significantly different in the injected than in the uninjected mice in both palatal shelves and tongues. The incidence of steroid-induced cleft palate parallels the palatal shelf concentration of cAMP before steroid treatment of the mother with one exception. These data suggest that a portion of the H-2 controlled component of susceptibility to steroid-induced cleft palate is mediated through alterations in the metabolism of cAMP.

Animals↗

Developmental mechanisms in normal and abnormal palate formation with particular reference to the aetiology, pathogenesis and prevention of cleft palate.

Palatal development was studied macroscopically, microscopically and ultrastructurally in foetuses of inbred Wistar rats and Alligator mississippiensis. In the rat, elevation of the palatal shelves from a vertical position lateral to the tongue to a horizontal position above the tongue, occurs very rapidly. This reorientation is postulated to be caused by an intrinsic turgor shelf force generated by the hydration of mesenchymal mucopolysaccharides (predominantly hyaluronic acid). Cleft palate was induced in rat foetuses using 5-fluoro-2-desoxyuridine and was associated with greatly decreased mucopolysaccharide synthesis. The alligator is the only animal which develops in an external egg and which possesses a true mammal-like secondary palate: it is therefore a useful animal model system because longitudinal studies and direct surgical and pharmacological manipulations can be performed. The palatal shelves of alligators grow horizontally above the dorsum of the tongue from their first appearance. This de novo horizontal shelf growth is associated with an increase amount of space in the alligator oronasal cavity due to the small, fatty, alligator tongue. It is postulated that the evolution of the large muscular mammalian tongue constrains the palatal shelves to grow vertically until sufficient space can be created to form the common nasal passage simultaneous with shelf elevation.

Alligators and Crocodiles↗

Hard palate repair timing and facial growth in cleft lip and palate: a systematic review.

OBJECTIVE: To evaluate the effect of timing of hard palate repair on facial growth in patients with cleft lip and palate, with special reference to cranial base, maxilla, mandible, jaw relation, and incisor relation. DESIGN: A systematic review. METHODS: The search strategy was based on the key words "facial growth," "cleft lip palate," and "timing of (hard) palate repair." Case reports, case-series, and studies with no control or comparison group in the sample were excluded. RESULTS: Fifteen studies met the selection criteria. All the studies were retrospective and nonrandomized. Five studies used cephalometry and casts, seven used cephalometry, and three used casts. Methodological deficiencies and heterogeneity of the studies prevented major conclusions. CONCLUSION: The review highlights the importance of further research. Prospective well-designed, controlled studies, especially targeting long-term results, are required to elucidate the effect of timing of hard palate repair on facial growth in patients with cleft lip and palate.

Cleft Lip↗

Dentoalveolar growth inhibition induced by bone denudation on palates: a study of two isolated cleft palates with asymmetric scar tissue distribution.

OBJECTIVE: This report presents two cases of isolated cleft palate with asymmetric distribution of postsurgical scar tissue determined by laser Doppler flowmetry. To determine the effect of mucoperiosteal denudation of the bone on maxillary alveolar growth, the analysis of dentoalveolar structures compared the affected side to the unaffected side of each case. METHOD: Two Japanese girls with isolated cleft palates were examined. Both subjects had undergone pushback operations (a modified version of the procedure of Wardill) for palatal repair at 18 months of age. Palatal blood flow was examined by laser Doppler flowmetry when the girls were 12 years old to determine the extent of postsurgical scar tissue over the denuded bone. To analyze the maxillary dentoalveolar structures three dimensionally, the whole surface of the upper dental cast was measured and recorded by an optical measuring device when the girls were 7 years old. RESULTS AND CONCLUSIONS: Analysis via flowmetry showed that the palatal scar tissue area was limited to the anterior tooth region on the right (unaffected) side but extended posteriorly to the premolar region on the left (affected) side in both subjects. The two girls had similar dentoalveolar structures, with the dental and alveolar arches deflected lingually at the deciduous molar area on the affected side. There were no differences in the buccolingual inclination of deciduous molars or in the vertical growth of the alveolar processes between the affected and unaffected sides. In both girls, bone denudation in the premolar region appeared to result in less than 3 mm of displacement of the teeth palatally, with no change in lingual inclination. Any effects of scar tissue on the vertical development of the alveolus were not substantiated.

Alveolar Process↗

An electropalatographic investigation of middorsum palatal stops in an adult with repaired cleft palate.

OBJECTIVE: Middorsum palatal stops are compensatory articulations that occur relatively frequently in cleft palate speech. This study used electropalatographic (EPG) and acoustic data to investigate /t/ and /k/ targets produced as middorsum palatal stops ([c]) by an adult with an articulation disorder associated with a repaired cleft palate. RESULTS: Two novel observations were made from the instrumental data. First, although /t/ and /k/ targets were judged by phonetically trained listeners as homophonous (i.e., both produced as [c]), the EPG data revealed that the place of articulation for the [c] produced for /t/ was more anterior than the place of articulation for the [c] produced for /k/. Second, production of palatal stops involved lateral release followed by a variable period of lateral friction. Measurements made from the instrumental data quantified the temporal extent of lateral friction during the aspiration period. CONCLUSIONS: These observations merit further systematic investigation in cleft palate speech, and the procedures reported in this study are considered appropriate for such future research.

Adult↗

Palate height: another indicator of surgical outcome in unilateral cleft lip and palate?

OBJECTIVE: To determine whether palate height and maxillary arch depth are systematically related to the surgical center at which primary repair in unilateral cleft lip and palate (UCLP) was carried out. DESIGN: A retrospective comparison based on study casts of consecutive cases of UCLP obtained at age 9 years from six different centers. The observer who conducted measurements was blinded to the source of individual records. SETTING: The patients whose records were analyzed received all their surgical care in a national health service setting in six different northern European centers and regions. PATIENTS: Patients were consecutively treated Caucasian children with non-syndromic complete UCLP born in the period 1976 to 1979. MAIN OUTCOME MEASURES: The main outcome measures for the original study were craniofacial form, dental arch relationships, nasolabial appearance, and speech. This report focuses on measurements of anterior maxillary arch depth and palate height. RESULTS: Anterior arch depth and anterior palate height showed some variation among the centers. There was a tendency for anterior arch depth and palate height to also be reduced at centers at which patients showed unfavorable dental arch relationships. CONCLUSIONS: Anterior arch depth and palate height might be considered in future studies of surgical outcome and in their possible relationship to problems of articulation.

Analysis of Variance↗

Palatal width growth rates as the genetic determinant of cleft palate induced by vitamin A.

While many studies have demonstrated sexual dimorphism and racial variation (Mongoloid greater than Caucasian greater than Black) in the incidence of palatal clefting, only generalized statements have been made concerning the causal mechanism (eg, "genetic propensity"). The present study tests the hypothesis that the more rapid the growth rate of the width of the facial skeleton, the higher the incidence of palatal clefting, given a uniform environmental insult. Two strains of pregnant rats with differing rates of craniofacial development (Wistar-fast and Sprague-Dawley-slow) were injected prenatally with high doses of vitamin A (300,000 IU/kg) on days 10-13 gestation. Fetuses were examined at 20 d prenatally and the incidence of palatal clefting was recorded. A significantly higher (P less than .05) incidence of palatal clefting was noted in the more rapidly developing strain (66% vs 81%). These results support statements by Fraser and others who suggest that head width and palatal clefting are causally related. The "genetic propensity" in this case, then, is the rate of palatal width development.

Aging↗

Palatal slit: a new spontaneous defect of the palate of C57BL/6 mice.

A hitherto undescribed palatal defect, here named "palatal slit," was observed during a teratological study of C57BL/6 fetuses. The defect, involving a failure of fusion of the premaxilla and palatal shelves, corresponds to stage 7 in normal palate closure. Adult C57BL/6 mice have been observed with the defect, so it does not represent a developmental delay that is repaired postnatally. Genetic factors of an unknown nature seem to be involved in the occurrence of palatal slit, which does not appear to be related developmentally to cleft palate. Some preliminary information on the defect is reported here.

Animals↗

Development of the object permanence concept in cleft lip and palate and noncleft lip and palate infants.

Studies of language acquisition in cleft lip and palate infants have not investigated the development of specific cognitive concepts, such as object permanence, which may be related to early linguistic skills. This study obtained comparative data on the development of the object permanence concept in cleft lip and palate and noncleft lip and palate infants to determine whether there were significant differences in rate or sequence of development. Infants were tested for the object permanence concept from 12 through 18 months of age. Results revealed significant improvement in all infants' scores with age, indicating progressive development of the concept. Further, while scores were not significantly different between the cleft lip and palate groups, scores for these groups were significantly better than scores for noncleft lip and palate infants. Superior performance of the cleft lip and palate infants may have resulted from increased environmental stimulation provided by their parents. Implications for intervention and future research in this area are presented.

Attention↗

Analysis of the posterior palatal seal and the palatal form as related to the retention of complete dentures.

Dislodging forces were applied at three different locations--anterior, posterior, middle--on acrylic resin bases of uniform thickness. The influence of different posterior palatal seals and the form of the palate were evaluated. A Pearson statistical correlation analysis was performed to correlate these factors. The findings were as follows: 1. The anterior attachment needed for greatest amount of force to dislodge the base; the posterior attachment needed the least. 2. The middle attachment showed more variability of the forces needed to dislodge the bases. It was influenced by the form of the palate, the type of posterior palatal seal, and the weight of the bases. 3. The middle location is the most reliable region for testing the retention of complete dentures. 4. The form of the palate has direct influence on the retention of complete dentures and will aid in the selection of the type of posterior palatal seal needed.

Denture Bases↗

Role of free-tissue transfer in the treatment of recalcitrant palatal fistulae among patients with cleft palates.

Recurrent palatal fistulae present a particularly vexing problem for patients with cleft lips and palates and their surgeons. When primary closure fails, conventional wisdom and the standard of care suggest local flap techniques for defect closure. For the large majority of patients, this approach is successful. There is, however, a small subset of patients who undergo multiple surgical procedures in unsuccessful attempts to close recalcitrant fistulae, particularly at the anterior, densely scarred, hard palate. In this setting, repair calls for the introduction of well-vascularized pliable tissue to close the defect and to avoid hampering further palatal growth. Local muscle flaps and oral axial pattern flaps have been advocated and used successfully. However, those approaches have their own drawbacks, such as multiple surgical interventions, patient compliance, and intraoral scarring. In an effort to avoid the problems associated with local flaps, distant microvascular tissue transfers were investigated. During a 6-year period, six free-tissue transfers were performed as a primary means of treating recalcitrant palatal fistulae. Three dorsalis pedis flaps and three osseous angular scapular flaps were used. The conditions of all patients improved, with five patients achieving complete long-term closure of the palatal defect. This experience indicates that modern microvascular techniques have reached a level of success commensurate with that of other flap techniques; therefore, it is concluded that free-tissue transfer should be considered as a primary means of addressing these difficult cleft problems.

Adolescent↗

The middle ear in cleft palate children pre and post palatal closure.

A multicentre prospective trial was commenced in July 1984 to establish the incidence of otitis media with effusion (OME) in children born with a cleft of the palate. Additionally, the data recorded would allow an assessment of the effect of palatal closure on middle ear function. Prior to palatal closure, 97% of ears in a group of 50 patients had otitis media with effusion (OME). The insertion of a long-term ventilation tube provided a means of aeration of one ear with the non-ventilated ear acting as a control. Eighty percent of control ears had persistent OME during a 24-month follow-up period post palatal repair. It would seem that OME is universally present in children with a cleft palate prior to 4 months of age and this incidence is only marginally diminished by palatal surgery. The liaison between plastic surgical and ENT units should be even closer than before in order to manage these patients satisfactorily.

Cleft Palate↗