bluuebunny/crossref_metadata_2025 huggingface dataset:
Responsiveness of Single Afferents in the Infraorbital Nerve to Oral Air Pressures Generated by Consonants
Authors: Furusawa Kiyofumi , Yamaoka Minoru , Ichikawa Norihiko
Published: October 2018

The responsiveness of receptors supplying the oral mucosa to air pressures generated during consonant production was investigated to obtain information about hypothetical mechanisms underlying speech deficits. The delay between the onset of the neural discharge and the pop puff of phonation (mouth-exit pressure) for /pa/ production was significantly shorter and less variable than it was for /ta/ and /ka/ production, suggesting that the discharge is more closely coupled to the onset of /pa/ production. The data were interpreted to imply that single fibers of the Infraorbital nerve respond to the build-up of oral air pressure during /pa/ production. This, and similar sensory information, may be used by the central neural mechanisms which monitor and control the air pressures required for phonation.
Anterior Paraseptal Cartilage Development in Normal and Cleft Lip and Palate Human Fetal Specimens
Authors: Mooney Mark P. , Siegel Michael I. , Kimes Kyle R. , Todhunter John S. , Smith Timothy D.
Published: October 2018

As part of an ongoing study of age-related variation in fetal cleft lip and palate nasal capsule morphology, anterior paraseptal cartilage development was examined histologically in a sample of nine complete cleft lip and palate human fetuses, and 20 without clefts ranging in age from 8 to 21 weeks. A computer reconstruction technique was used to quantify lengths, volumes, and generate growth curves from various regression equations. Anterior paraseptal cartilage length was seen to change in a slgmoldal fashion while volume changes were best described by a logarithmic curve for both groups, with the steepest increases from 15 to 21 weeks. Cleft specimens exhibited significantly different growth rates (line slopes) for both length and volume dimensions compared with the normal specimens. The larger, for age, cartilages were associated with an enlarged, Inferior border of the nasal septal cartilage and vomer in the cleft sample. These results are consonant with previous findings in this cleft fetal sample of other enlarged midline structures (i.e., nasal septal cartilage and vomer). Results suggest that growth abnormalities in one or all of these structures may be yet another mechanism for early nasal airway impingement and reduction in cleft lip and palate neonates.
Recurrent Trismus: Twenty-Year Follow-Up Result
Authors: Hirano Akiyoshi , Iio Yoshimi , Murakami Ryuichi , Fujii Tohru
Published: October 2018

The authors encountered an unusual case of recurrent trismus in a patient who had oral synechia, cleft lip and palate, digital anomalies, and external genital anomaly. Mandibular movement was severely restricted by congenital soft-tissue adhesion in the retromolar region. Despite aggressive surgical efforts and postoperative manipulations, limitation of mouth opening recurred soon after each operation. Furthermore, congenital and postoperative soft-tissue adhesion resulted in severe subsequent jaw deformities, which needed combined maxillary and mandibular advancement.
Atypical Cleft of the Palate: A Report
Authors: Hughes Lowell A. , Zuker Ronald M.
Published: October 2018

Our early experience with a rare atypical cleft of the soft palate in a 15-month-old female infant is presented. The cleft extended obliquely across the soft palate from the retromolar region on the left to the base of an intact uvula. Surgical management included a three-layer closure and lengthening of the left side of the soft palate. Close follow-up and reporting of the patient's progress are planned.
Induction of Cleft Palate by Gamma-Irradiation of Prenatal CD-1 Mice
Authors: Saad A. Yousef , Abdelazim Ali A. , El-Khashab Mostafa M. , Mansour Mohamed A.
Published: October 2018

Fetuses of pregnant mice exposed to whole body γ-irradiation (400 rads) on the 12th gestational day were compared with control fetuses to assess the effect of radiation on palatogenesis. Experimental and control groups were sacrificed on day 18 of gestation. The fetuses were removed via laparotomy and counted to determine the litter size. Animals were decapitated and the maximum head height, width, and circumference recorded. For light microscopic examination of palatogenesis, heads were fixed in Bouin's solution and prepared for paraffin sectioning and staining with hematoxylin and eosin. A significant reduction in litter size (p < .0025) and head measurements (p < .005) of Irradiated fetuses was observed when compared to those of the control group. Histologic examination of serial coronal sections demonstrated that 100% of the experimental fetuses had palatal clefts. It was concluded that γ-rays have adverse effects on the litter size, head measurements, and palatal closure of mouse fetuses.
Midline Defects of the Orofaciodigital Syndrome Type VI (Váradi Syndrome)
Authors: Wey Philip D. , Neidich Julie A. , Hoffman Lloyd A. , Latrenta Gregory S.
Published: October 2018

The orofaciodigital syndromes (OFDS) represent a spectrum of anomalies of the palate, cranium, hands, and feet. Váradi syndrome, designated OFDS type VI, is a rare disorder that is additionally characterized by cerebellar anomalies. The following report is of a patient with OFDS VI and characteristic multiple midline defects: median cleft lip and palate, lingual cleft with nodules, and midline brain malformation. In addition, this case is uniquely associated with the presence of midline (metopic and sagittal) craniosynostoses as well. It is unusual that deformities which result from premature fusion of cranial vault sutures would appear synchronously in a syndrome based on the concept of failure of fusion or coalescence of facial growth centers. The midline represents an independent developmental field, whereby CNS defects and midline anomalies can present concurrently.
Anterior Cranial Base Morphology in Mice with Midfacial Retrusion
Authors: Lozanoff Scott , Jureczek Stefan , Feng Tracey , Padwal Raj
Published: October 2018

The role of the anterior cranial base in the morphogenesis of class III malocclusions remains uncertain. This study was conducted to determine whether morphologic deficiencies occur in the anterior cranial base in the Brachyrrhine (Br) mouse mutant showing severe midfacial retrusion, which is characteristic of a class III malocclusion. Crania from three groups of C3H/Hej, 3H1 Br/+, and 3H1+/+ mice, each consisting of 15 animals, were collected at 1, 3, and 5 days of age (total = 135). The anterior cranial base from each specimen was subjected to computerized reconstruction and ten landmarks were digitized from each model. The landmark configurations were compared using Procrustes analysis. Significant differences between models were determined at each age. In order to localize differences between forms, average landmark configurations derived from Procrustes analysis were subjected to finite-element analysis. Size-change values for the 3H1 Br/+ animals showed magnitudes that increased in an anteroposterior direction when compared to the 3H1 +/+ and C3H/Hej animals at all ages. The largest values were located posteriorly along the ossifying front of the presphenoid. In five of six comparisons, the size-change values separated into two distinct clusters. The posterior region of the anterior cranial base was divisible into two subclusters, one located superiorly and the other interiorly. These data suggest that midfacial retrusion in the Br mouse may be caused, in part, by growth deficiencies in the posterior region of the anterior cranial base, particularly the presphenoidal and sphenoethmoidal regions.
Effects of Visible and Invisible Orofacial Defects on Self-Perception and Adjustment across Developmental Eras and Gender
Authors: Broder Hillary L. , Smith Fraser B. , Strauss Ronald P.
Published: October 2018

Self-ratings of satisfaction with appearance and accomplishment of psychosocial tasks were examined by age and gender among school aged children with visible defects (cleft lip and/or palate, n = 272), or Invisible defects (cleft palate only, n = 159), and dental patients (n = 128) without clefts. Using weighted least squares ANOVA and logistic regressions, the results revealed that subjects with visible defects expressed greater dissatisfaction with their appearance than those subjects with invisible defects (p < .001). Subjects with Invisible defects consistently expressed lower problem solving ability than subjects with visible defects (p < .001) and dental patients with no defects (p < .05). Both groups with clefts expressed less social independence (p < .001); and subjects with clefts reported having more friends than other children (p < .01). Implications for clinicians and further research are discussed.
Presurgical Orthopedic Premaxillary Alignment in Cleft Lip and Palate Reconstruction
Authors: Papay Frank A. , Morales Louis , Motoki David S. , Yamashiro Duane K.
Published: October 2018

Premaxillary malposition is a difficult problem in cleft lip and palate repair. Orthopedic palatal devices are excellent in positioning the premaxilla, though they are somewhat cumbersome and require complex techniques in adjusting precisely the position of the premaxilla prior to repair. A new technique has been developed for premaxillary repositioning in conjunction with palatal shelf expansion and obturation. The procedure implements microplate fixation anterior to the premaxillary segment and linked to a palatal splint by adjustable elastics. The microplate is Inserted through a nasal floor incision and secured by a tight submucosal tunnel through minimal dissection between the prolabium and premaxllla. The last hole of each mlcroplate protrudes through the mucosa and is attached to a pin-retained palatal splint by an elastic chain. Differential tension is applied to the chains to allow gradual repositioning of the protruding maxilla while the splint expands and maintains positioning of the lateral palatal segments. These elastic retractors can be adjusted by staff in the outpatient office. During the past 2 years, this technique has been used successfully in 21 consecutive patients with unilateral or bilateral cleft lip and palate. Its technical ease and design allows simple adjustments to control premaxillary positioning and growth before definitive surgical closure.
Augmentation of Oral Tissues in Rabbit Using Autogenous Fat
Authors: Canady John W. , Thompson Sue Ann , Moon Jerald B. , Glowacki Richard L.
Published: October 2018

Patients with mild velopharyngeal incompetence (VPI) may have speech disorders, which are not sufficiently severe to warrant extensive surgical intervention, yet may not be amenable to correction by speech therapy alone. Augmentation of the posterior pharyngeal wall to aid in closure of the velopharyngeal sphincter may be beneficial in establishing better speech patterns, especially when combined with speech therapy. A variety of materials and techniques have been used in the past for this purpose. In this setting, autogenous fat may be transplanted without the risks incurred by augmentation with synthetic materials and involves very little donor site morbidity. The literature is somewhat contradictory, however, regarding the stability of the augmentation achieved using autogenous fat and there are no histologic studies describing the fate of fat injected into tissues of the oral cavity. Prior to introduction of this technique into clinical practice, this study was designed to investigate the fate of autogenous fat injected submucosally in the oropharyngeal region. Autogenous fat was injected into the anterior soft palate using the rabbit as a model. Histologic and gross inspections were performed at 2 days, 1, 2, and 4 weeks after injections. At the end of 4 weeks, at least 50% of the injection sites had visible evidence of augmentation, and 90% had histologic evidence of submucosal fat. In some instances most of the fat was resorbed; however, there were no instances of clinical infection or necrosis of the injection site. We conclude that submucosal injection of autogenous fat is a feasible alternative to using synthetic or other biologic materials for augmentation in the oral cavity.
Treatment of Cleft Palate Associated with Robin Sequence: Appraisal of Risk Factors
Authors: Lehman James A. , Fishman Jefrey R.A. , Neiman Gary S.
Published: October 2018

Mandibular hypoplasia, airway obstruction, and a typical wide U-shaped cleft palate comprise the Robin sequence. Although much has been written regarding the treatment of these patients in the neonatal period, the literature reveals little information regarding later care of the cleft palate in these patients. The purpose of this study is to examine patients with the Robin sequence and evaluate the risk of postsurgical problems and outcome related to the neonatal period. Thirty-six patients with the Robin sequence presenting from 1972 through 1990 were reviewed. A majority of patients had feeding and respiratory difficulties, to varying degrees, following birth. These problems were treated successfully by maneuvers ranging from positioning to two infants who eventually required tracheostomy. Thirty-four patients had palate repair. Age at repair averaged 16.2 months, and one third of patients had associated anomalies. Infants who experienced problems following palatoplasty were those who had histories of severe difficulties and complications in the early months of life. In addition, patients with associated congenital anomalies had significantly more problems at the time of palate repair than those without anomalies. Those patients with the Robin sequence, who historically had minimal difficulty following birth, experienced few complications at the time of palate repair. Of the 34 patients with repaired palates, 23 demonstrated sufficient follow-up to allow for evaluation of speech outcome. Satisfactory or normal speech production was noted in 65.4%. This is not significantly different from that observed in all patients undergoing cleft palate repair during this same time period (74.9%). Secondary pharyngoplasty procedures were required in 17.4%. An overall complication rate of 29.4% was noted with palatal fistula occurring in 11.8%. Examination of an infant's immediate postnatal period, as well as for the presence of associated anomalies, will provide important predictive information on the potential difficulties following cleft palate repair. In addition, palatoplasty, as part of the overall team approach to the cleft patient, results in a satisfactory speech outcome in approximately two thirds of patients with the Robin sequence.
Intrauterine Repair of Surgically Created Defects in Mice (Lip Incision Model) with a Microclip: Preamble to Endoscopic Intrauterine Surgery
Authors: Oberg Kerby C. , Evans Michele L. , Nguyen Thanh , Peckham Norman H. , Kirsch Wolff M. , Hardesty Robert A.
Published: October 2018

We compared the difference between a nonpenetrating silver microclip and suture on wound healing, inflammatory response, and application time in the repair of surgically created standardized unilateral cleft lip type defects in fetal mice. Excellent lip continuity and dermal reconstitution were achieved by both methods of repair. Furthermore, collagen accumulation did not occur. Occasional mononuclear cells were seen around sutured repairs in contrast to microclipped repairs. The most significant difference, however, was in application time with the microclip requiring an average of 7 seconds (±2) compared to 90 seconds (±15) for suture. We conclude that the microclip offers distinct advantages for Intrauterine cleft lip repair: (1) nonpenetrating tissue approximation; (2) less inflammatory response than suture; (3) technically more rapid and less difficult to apply than suture; and (4) can be utilized more readily than suture for endoscopic approaches limiting the risks of fetal surgery for both the mother and the fetus.
Laryngeal Airway Resistance in Children with Cleft Palate and Adequate Velopharyngeal Function
Authors: Zajac David J.
Published: October 2018

Laryngeal airway resistance during vowel production was determined for 10 children without cleft palate and 14 children with cleft palate and adequate velopharyngeal function. The children with cleft palate were further grouped according to either complete or incomplete velopharyngeal closure. All children performed a syllable repetition task with nostrils occluded at self-determined effort and a targeted effort typical of adult speech. Results indicated that all children exhibited significantly greater laryngeal resistance at self-determined effort. No significant differences occurred among the groups of children at either effort level. Some children with incomplete velopharyngeal closure, however, tended to exhibit relatively increased laryngeal resistance at the targeted effort level when their nostrils were unoccluded. Implications for a regulation-control model of speech production and clinical assessment are discussed.
Incomplete Midline Cleft of Lower Lip
Authors: Arshad Ahmad Ridzwan
Published: October 2018

Midline cleft of the lower lip is defined as a midline vertical cleft of the soft tissue of the lower lip. It may present with a midline cleft of the mandible. It may also be accompanied by other congenital anomalies such as a cleft tongue, ankyloglossia, a heart lesion, and absence of the hyoid bone. The etiologic cause is thought to be a failure of mesodermal penetration into the midline structures of the first branchial arch. This case report is on a female child who presented with an incomplete midline cleft of the soft tissue of the lower lip. It was surgically corrected with a vertical wedge excision and primary closure.
Periosteum in Regeneration of Palatal Defects
Authors: Dixit Uma B. , Kelly Kevin M. , Squier Christopher A. , Bardach Janusz
Published: October 2018

The influence of the periosteum on the regeneration of palatal bone was investigated in this study. Eighty, 8-week-old, purebred beagle dogs were assigned randomly to four groups: (1) unoperated dogs as a group control; dogs in which the mid-third of the palate was surgically removed and (2) left unrepaired (unrepaired controls); (3) repaired with mucosal flaps; (4) repaired with mucoperiosteal flaps. Five animals from each group were killed at 4, 6, 8, and 12 weeks after surgery and coronal sections examined under light microscopy. Among the animals with complete soft tissue healing, 8 of 12 dogs from Group 2, 11 of 20 from Group 3, and 10 of 19 from Group 4 showed complete bone regeneration. No significant differences were found overall in bone thickness and bone density measurements between Group 3 and Group 4. Histologically, a well-differentiated periosteum was present on the maxilla at 4 weeks, even in animals in which the periosteum had not been preserved in the original flap. These results suggest that maintaining the periosteum at surgical closure does not influence bone regeneration in beagles up to 12 weeks of age. We suggest that osteoprogenitor cells, migrating from the undisturbed local periosteum adjacent to the defect, were responsible for the new bone growth in our study.
Microform Cleft Lip Associated with a Complete Cleft Palate
Authors: Thaller Seth R. , Jong Lee Taik
Published: October 2018

Clefting of the lip with or without an associated cleft palate may be present in varying degrees of severity. The so-called microform cleft lip or forme fruste has been characteristically described as having the appearance of a repaired cleft lip. The following case describes a patient with microform cleft lip and a complete cleft of the hard and soft palates. To the best of our knowledge, this is the first report of such an association.
Spatial Dysmorphology of the Foot in Apert Syndrome: Three-dimensional Computed Tomography
Authors: Dale Collins E. , Marsh Jeffrey L. , Vannier Michael W. , Gilula Louis A.
Published: October 2018

Computer assisted medical imaging was used to define the spatial dysmorphology of the foot in three patients with Apert syndrome and to correlate that dysmorphology with ambulation and footwear. Thin slice (2 mm), abutting, high resolution axial computed tomography (CT) foot scans were obtained. The CT data were post processed, using Analyze, to generate three-dimensional surface shaded and volumetric reformations. The reformatted images were evaluated by a bone and joint radiologist to identify abnormalities of bone shape, size, and orientation, of joint morphology, and of the foot as a whole. Five consistent findings were observed among the three pairs of feet: (1) anomalous great toes with phalangeal and metatarsal pathology; (2) simple syndactyly of toes 2-5; (3) fusions between metatarsals; (4) tarsal coalitions; and (5) limitations in commercial footwear. One patient underwent bilateral fifth metatarsal wedge osteotomies to facilitate the wearing of shoes. The dysmorphology of the Apert foot is a combination of congenital malformations and postnatal deformations, secondary to progressive synostosis. Prophylactic foot surgery may be indicated in Apert patients to facilitate shoe fitting.
Guided Bone Regeneration in Calvarial Bone Defects Using Polytetrafluoroethylene Membranes
Authors: Bosch Carles , Melsen Birte , Vargervik Karin
Published: October 2018

Guided bone regeneration is defined as controlled stimulation of new bone formation in a bony defect, either by osteogenesis, osteoinduction, or osteoconduction, re-establishing both structural and functional characteristics. Bony defects may be found as a result of congenital anomalies, trauma, neoplasms, or infectious conditions. Such conditions are often associated with severe functional and esthetic problems. Corrective treatment is often complicated by limitations in tissue adaptations. The aim of the investigation was to compare histologically the amount of bone formed in an experimentally created parietal bone defect protected with one or two polytetrafluoroethylene membranes with a contralateral control defect. A bony defect was created bilaterally in the parietal bone lateral to the sagittal suture in 29 6-month-old male Wistar rats. The animals were divided into two groups: (1) In the double membrane group (n=9), the left experimental bone defect was protected by an outer polytetrafluoroethylene membrane under the periosteum and parietal muscles and an inner membrane between the dura mater and the parietal bone. (2) In the single membrane group (n=20), only the outer membrane was placed. The right defect was not covered with any membrane and served as control. The animals were killed after 30 days. None of the control defects demonstrated complete or partial bone regeneration. In the single membrane group, the experimental site did not regenerate in 15 animals, partially in four, and completely in one. In the double membrane group, six of the experimental defects had complete closure with bone, two had partial closure, and one no closure. The use of two membranes protecting the bone edges of the parietal defect from the overlying tissues and underlying brain enhanced bone regeneration in experimental calvarial bone defects. The biologic role of the dura mater may not be of critical importance in new bone regeneration in these calvarial bone defects.
Effects of Gravity on Velopharyngeal Muscle Activity during Speech
Authors: Moon Jerald B. , Canady John W.
Published: October 2018

Assessment of the role of gravitational forces in the motor control of the velopharyngeal mechanism was the focus of this study. Specifically, the effect of gravity on activation levels of the levator veli palatini and palatoglossus muscles was assessed. Nineteen volunteers repeated a CV syllable in upright and supine body positions. Overall, lower peak activation levels of levator veli palatini were observed in the supine body position. The results suggest that less muscle activity was seen in the levator veli palatini in the supine body posture, where gravitational effects worked in the same direction (i.e., toward closure). No statistically significant group effects were seen in muscle activation levels of palatoglossus across the two body postures, although clear gravity effects were observed in some subjects. The implications of these findings from a speech motor control perspective are discussed in relation to normal and disordered velopharyngeal function.
Oral Air Pressure and Nasal Air Flow Rate on Levator Veli Palatini Muscle Activity in Patients Wearing a Speech Appliance
Authors: Tachimura Takashi , Hara Hisanaga , Wada Takeshi
Published: October 2018

This study was designed to determine if levator veli palatini muscle activity can be elicited by simultaneous changes in oral air pressure and nasal air flow when a speech appliance is in place. The speech appliances routinely worn by 15 subjects were each modified experimentally by drilling a hole in the vertical center of the pharyngeal bulb. The air flow rate into the nasal cavity through the opening in the bulb was altered by changing the circular area of the opening in the bulb from the occluded condition (Condition I), to circular area of 12.6 mm2 (4 mm in diameter; Condition II), and then to 38.5 mm2 (7 mm in diameter; Condition III). Electromyographic activity was measured from the levator veli palatini muscle with changes in nasal air flow rate and oral air pressure. Levator veli palatini muscle activity was correlated with changes in nasal air flow and oral air pressure. Increases in levator veli palatini muscle activity were associated with increases in nasal air flow rate compared to oral air pressure changes. The results indicated that aerodynamic variables of nasal air flow and oral air pressure might be involved in the neural control of speech production in individuals wearing a speech appliance, even if the subjects exhibit velopharyngeal incompetence without using a speech appliance. Also, the stimulating effect of bulb reduction therapy on velopharyngeal function might be achieved through the change in aerodynamic variables in association with the bulb reduction.
Do Palatal Lift Prostheses Stimulate Velopharyngeal Neuromuscular Activity?
Authors: Witt Peter D. , Rozelle Arlene A. , Marsh Jeffrey L. , Marty-Grames Lynn , Muntz Harlan R. , Gay W. Donald , Pilgram Thomas K.
Published: October 2018

The purpose of this investigation was to evaluate the ability of palatal lift protheses to stimulate the neuromuscular activity of the velopharynx. Nasendoscopic evaluations were audio-videotaped preprosthetic and postprosthetic management for 25 patients who underwent placement of a palatal lift prosthesis for velopharyngeal dysfunction (VPD). These audio-videotapes were presented in blinded fashion and random order to three speech pathologists experienced in assessment of patients with VPD. They rated the tapes on the following parameters: VP gap size, closure pattern, orifice estimate, direction and magnitude of change, and qualitative descriptions of the adequacy of VP closure during speech. VP closure for speech was unchanged in 69% of patients and the number of patients rated as improved or deteriorated was nearly identical at about 15%. Postintervention gap shape remained unchanged in 70% of patients. The extent of VP orifice closure during speech remained unchanged in 57% of patients. Articulations that could impair VP function improved in 30% of patients, deteriorating in only 4%. Results of this study neither support the concept that palatal lift prostheses alter the neuromuscular patterning of the velopharynx, nor provide objective documentation of the feasibility of prosthetic reduction for weaning.
Who Decides? Patients, Parents, or Gatekeepers: Pediatric Decisions in the Craniofacial Setting
Authors: Mouradian Wendy E.
Published: October 2018

Special ethical issues arise for the craniofacial team dealing with pediatric patients, which include competency, surrogacy, and the “best interests” standard. Medical decisions for children are made by surrogates, usually parents, who must use the “best interests” standard. The team's primary responsibility is to the child, not the parents. Children should participate as abilites allow, especially for elective procedures. Increasingly, cost considerations also influence medical decisions. The craniofacial team is often a de facto gatekeeper. Ethically responsible team behavior includes: weighing risks and benefits of proposed interventions; promoting discussion with families and patients to identify “best interests;” monitoring outcomes; and advocacy for craniofacial patients individually and at a policy level. Care guidelines and definitions of basic levels of care should be developed to assist teams with decision-making and advocacy efforts. Ethical analysis is part of both good patient care and good policy formation, and should be a part of regular team deliberations.
Comparison of Facial Form in Primary Alveolar Bone-Grafted and Nongrafted Unilateral Cleft Lip and Palate Patients: Intercenter Retrospective Study
Authors: Trotman Carroll-Ann , Long Ross E. , Rosenstein Sheldon W. , Murphy Carole , Johnston Lysle E.
Published: October 2018

The purpose of this study was to describe and compare posttreatment craniofacial morphology in samples of complete unilateral cleft lip and palate (CUCLP) patients treated at two leading clinics: The Children's Memorial Hospital Cleft Palate Clinic, Chicago, Illinois, and the Lancaster Cleft Palate Clinic, Lancaster, Pennsylvania. These centers have well-defined treatment protocols that allow the long-term effects on craniofacial form of the following treatment regimes to be contrasted: (1) Chicago—primary alveolar bone grafting, with definitive lip repair at age 4 to 6 months and hard and soft palate repair at 6 to 12 months; and (2) Lancaster—definitive triangular-flap lip repair at 3 months of age, followed by staged surgeries of the hard and soft palates, both completed by 18 months of age, but without primary alveolar bone grafting. Although the Lancaster center now performs secondary alveolar bone grafting, the majority of the patients studied here were treated before this procedure became part of their protocol. Patients were eligible for inclusion if they had no other congenital anomalies and no previous orthodontic treatment. A sample of 43 (24 male, 19 female) CUCLP patients was obtained from the Chicago Center, each of which was then matched to a non-grafted Lancaster CUCLP patient. The matching criteria were age, sex, and sella-nasion distance (to control, at least in part, for size differences). Lateral cephalometric radiographs of these 86 CUCLP patients were traced, digitized, and analyzed. Additionally, all linear data were adjusted to a standard magnification of 8% because the cephalograms from each center featured different enlargements. The Chicago and Lancaster samples had mean posttreatment ages of 10.32 years (SD = 1.96) and 10.40 years (SD = 2.18), respectively. The grafted Chicago group had faces that were on average less maxillary protrusive compared with the nongrafted Lancaster sample; it appeared, however, that the mandible compensated for the maxillary position by downward and backward rotation. As a result, a similar maxillomandibular relationship was noted in both groups, although, in the Chicago group, the lower anterior facial height increased.
Craniofacial Morphology in Twins with Cleft Lip and Palate
Authors: Laatikainen Tuula , Ranta Reijo , Nordström Rolf
Published: October 2018

The craniofacial morphology of 11 pairs of monozygotic (MZ) and 28 pairs of dizygotic (DZ) Finnish twins, discordant or concordant for cleft of the lip (CL), unilateral cleft lip and palate (UCLP), or cleft palate only (CP) were investigated by means of lateral cephalometric radiographs. The results were compared to those of age-, sex-, and cleft-type matched single-birth cleft subjects, and also with normative data from the Nordic population. The co-twins with no cleft lip or palate (NONC) showed only slightly more obtuse gonial and steeper mandibular angles compared to normative data. Twins with CL, UCLP or CP had a more retrusive mandible, a wider cranial base and mandibular angle, and a wider angle between the maxilla and mandible than did the single-birth cleft subjects. Comparison of the noncleft twin group with the CL, UCLP, and CP twin groups for the CL twins showed no significant differences. For the UCLP twins, a more retrusive and down- and backward rotation of both jaws, a wider gonial angle, and a wider cranial base angle was seen. The CP twins had their maxillae slightly retrusive, the down- and backward rotation of both jaws was apparent, and the gonial angle was more obtuse. A comparison between the noncleft MZ and noncleft CDZ twins showed no significant differences. The MZ CP twins had a more retrusive mandible and more down- and backward rotation of both jaws than did DZ CP twins. It thus can be suggested that twinning itself does not seem to have an effect on maxillofacial morphology, but the features of the mandibular structure, the cranial base angulation, and the inclination of the jaws are at least partly genetically induced.
Electropalatography Treatment in an Adult with Late Repair of Cleft Palate
Authors: Whitehill Tara L. , Stokes Stephanie F. , Yonnie Man Yuk Han
Published: October 2018

This paper describes the use of electropalatography (EPG) in the treatment of a speech disorder in a Cantonese-speaking woman who had primary repair of the palate at age 13. A multiple-baseline approach was used to document treatment efficacy using electropalatography. The client showed rapid improvement in articulatory placement, with generalization to nontarget phonemes. In addition, improvement was noted in her manner of articulation, with a reduction of nasal emission. The relationship between articulatory placement errors and nasal emission in late repair cleft is discussed. Explanations for the effectiveness of EPG with this client are offered.
Proprioceptive Representation of the Levator Veli Palatini Muscle in the Solitary Nucleus of the Rat
Authors: Furusawa Kiyofumi , Yasuda Kouichi , Okuda Daizo , Yamaoka Minoru
Published: October 2018

The levator veli palatini muscle is innervated by motoneurons of the glossopharyngeal nerve, which are located within the ambiguus nucleus; however, little is known about the afferent fibers of this muscle. A horseradish peroxidase study was conducted in rats following injection into the levator veli palatini muscle branch to reveal the location and the distribution of dendrites of the afferent fibers of the muscle. Terminal labels were densely distributed in the lateral region of the solitary nucleus, which receives afferents of the glossopharyngeal nerve, ipsilateral and contralateral to the injection site. The relationship of the levator veli palatini muscle with respiration was suggested by the localization of labeled terminals at sites where the afferents from the respiratory organs project densely, and by the demonstrated proprioceptive role of the afferent fibers passing through the muscle spindles contained in the levator veli palatini muscle.
Coronal Suture Pathology and Synostotic Progression in Rabbits with Congenital Craniosynostosis
Authors: Mooney Mark P. , Smith Timothy D. , Burrows Annie M. , Langdon Herbert L. , Stone Cynthia E. , Losken H. Wolfgang, , Caruso Kelly , Siegel Michael I.
Published: October 2018

The purpose of the present study was to describe coronal suture pathology and cross sectional synostotic progression in an inbred strain of rabbits with congenital craniosynostosis. Calvaria from 102 perinatal rabbits (39 unaffected; 63 bilateral or unilateral synostosis) were collected at fetal days 21 (n = 12), 25 (n = 20), 27 (n = 22), 30 (term) (n = 32), and 3 days post-term (n = 16) for gross morphologic and histologic examination. Synostotic foci, the extent of relative bony bridging, and suture morphology were evaluated qualitatively and quantitatively. Of the 204 coronal sutures examined, 91 sutures were synostosed, and 113 were patent. All synostosed sutures showed similar foci by day 25, which originated as bony bridges in the middle of each suture on the ectocortic surface. Bony bridging width increased significantly (p < .001) from day 25 through 3 days post-term, and was best described by a linear regression equation. Osteogenic front areas of synostosed sutures were up to 2.5 times greater than patent sutures in term fetuses. Findings demonstrate that coronal suture synostosis in the congenital rabbit model (1) begins early during suture morphogenesis (before 25 days of gestation); (2) consistently radiates from a single focus corresponding to a normal interdigitating region (i.e., a high-tension environment); (3) varies in onset and rate as evidenced by low R2 value between age and extent of bony bridging; and (4) is the result of early hyperostosis of the osteogenic fronts and sutural agenesis. A number of possible pathogenetic mechanisms are discussed.
Vomeronasal Organ Growth and Development in Normal and Cleft Lip and Palate Human Fetuses
Authors: Smith Timothy D. , Siegel Michael I. , Mooney Mark P. , Burdi Alphonse R. , Todhunter John S.
Published: October 2018

As part of an ongoing investigation of normal and cleft lip and palate (CLP) fetal midfacial development, the vomeronasal organ (VNO) was examined in 35 human fetuses (26 normal and 9 CLP) ranging in age from 8 to 30 weeks postmenstrual age. All specimens were examined histologically, and a computer reconstruction technique was used to quantify lengths and volumes of right and left VNOs and anterior paraseptal cartilages (PCs). Growth curves were generated for these data and were compared between normal and cleft samples. VNOs and PCs were present in all normal fetuses, whereas VNOs were found in only 5 of 9 CLP specimens and PCs were found in 8 CLP specimens. The side of the palatal cleft in specimens with unilateral CLP was associated with PC dysmorphologies but appeared to have no influence on VNO presence or absence. Septal dysmorphologies were observed in most CLP specimens with absent or abnormal VNOs. VNO length increased in a linear fashion across ages, and VNO volume was seen to change in a logarithmic fashion. Individual CLP specimens did not differ significantly from the mean of the normal sample for VNO length or volume. PC length was seen to change in a sigmoidal fashion, and PC volume changes were best described by a logarithmic curve. In contrast to findings on the VNO, CLP specimens exhibited significantly different growth rates (line slopes) for PC length and volume compared to the normal sample. Results on the normal sample suggested that the human fetal VNO exhibits volumetric increases between the 16th and 30th weeks of development, but this increase begins later and proceeds more slowly than that of the PCs. Examination of the CLP sample suggested that the VNOs were of “normal” size for age, but may be vulnerable to septal dysmorphologies that result in altered location or disruption.
First Year-of-Life Mortality among Infants with Oral Clefts: New York State, 1983–1990
Authors: Druschel Charlotte M. , Hughes Jeffery P. , Olsen Carolyn L.
Published: October 2018

This study examined the mortality experience of children with oral clefts using the New York State Congenital Malformations Registry. Infants born in the years 1983 to 1990 to New York residents, diagnosed with an oral cleft and matched to their birth certificate were included in the analysis. Children with oral clefts were compared to a sample of live births from the years 1983 to 1990 without malformations. Children with cleft palate without additional malformations had a statistically nonsignficant adjusted risk of 1.2 when compared to children with no malformations. Children with cleft lip with or without cleft palate had a 1.1 adjusted risk. However, 35% of children with oral clefts had associated malformations and experienced much higher mortality. Children with oral clefts should be carefully evaluated for additional malformations; if none are found, their mortality appears not to be elevated.
Subperiosteal Release of the Floor of the Mouth in Pierre Robin Sequence: Experience with 12 Cases
Authors: Caouette-Laberge Louise , Plamondon Caroline , Larocque Yvan
Published: October 2018

Between 1983 and 1995, 12 children with Pierre Robin sequence were treated with a subperiosteal release of the floor of the mouth (SRFM) to correct severe upper-airway obstruction (group 1: n = 10) and prolonged feeding difficulties (group 2: n = 2). Eight children had isolated Pierre Robin sequence, while four had associated anomalies. The age at surgery was 2 to 13 weeks (mean, 5.2 wk) in group 1, and 8.5 months to 5 years in group 2. Postoperative endotracheal intubation was maintained for 1 to 14 days, and nasopharyngeal intubation for 3 to 13 days. One child with a tracheostomy prior to SRFM was decanulated 11 weeks postoperatively. Postoperative course was uneventful and control polysomnograms (group 1) showed a marked improvement of the obstructive phenomenons and O2 saturation levels. Children were discharged 2 to 6 weeks postoperatively (mean, 4 wk) except for the child with a tracheostomy (19 wk). The only complication was a wound infection treated with systemic antibiotics. Oral feedings were initiated shortly after extubation: six children were discharged with oral feedings only, one child required supplemental gavages for 6 weeks at home, and three children had persistent inadequate oral intake. We have found SRFM to be an effective surgical procedure for the management of infants with Pierre Robin sequence.
Septoplasty for Obstructive Sleep Apnea in Infants after Cleft Lip Repair
Authors: Josephson Gary D. , Levine Jamie , Cutting Court B.
Published: October 2018

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.
Palatal Asymmetry in Cleft Palate Subjects
Authors: Kilpeläinen Pauli V.J. , Laine-Alava Maija T.
Published: October 2018

In subjects with different types of cleft palate, palatal shape and symmetry were evaluated utilizing the moiré contourography technique. The sample consisted of 95 subjects with cleft palate and 68 controls. The differences between cleft and control subjects in the transverse and anteroposterior location of the highest point of the palate, palatal axis angle, and the palatal index were assessed by analysis of variance. Effects of cleft type, gender, developmental stage of the dentition, missing teeth, and eight skeletal and pharyngeal cephalometric variables on palatal shape and symmetry were assessed using multiple-regression analyses. In comparison with the noncleft individuals, the cleft palate subjects showed parallel but clearly more remarkable asymmetry in palatal shape and position of the first maxillary molars. The anteroposterior location of the deepest point in the palate was more posterior, and the palate was relatively shallower. The severity of the cleft type affected both the anteroposterior and transverse position of the highest point in palatal morphology.
Frontonasal Malformation and the Oculoauriculovertebral Spectrum: The Oculoauriculofrontonasal Syndrome
Authors: Casey Holly D. , Braddock Stephen R. , Haskins Richard C. , Carey John C. , Morales Louis,
Published: October 2018

Frontonasal malformation (FNM) is a developmental field defect representing abnormal morphogenesis of the frontonasal eminence. The oculoauriculovertebral spectrum (OAVS) has been used to describe a broader range of first and second branchial-arch defects including hemifacial microsomia and Goldenhar's syndrome. A combination of FNM and OAVS has been described in the literature in 13 cases. This condition has been labeled as the oculoauriculofrontonasal syndrome, as well as ophthalmofrontonasal dysplasia. We have evaluated four patients with both FNM and OAVS. The pattern of malformation involves only the craniofacies: they have no vertebral defects, heart disease, or encephaloceles. The categorization of these four individuals and those in the literature raises interesting issues regarding syndrome classification. Originally, it was suggested that perhaps this disorder was a variation of Goldenhar's syndrome. However, now that it has become evident that FNM and OAVS are malformation patterns of etiologic and presumably pathogenetic heterogeneity, a more likely hypothesis is that when these two defects occur together, this represents a unique syndrome pattern. The purpose of this article is to suggest that the combination of OAVS and FNM may be a distinct entity, representing a discreet subset of patients.
Gingivoperiosteoplasty and Midfacial Growth
Authors: Wood Robert J. , Grayson Barry H. , Cutting Court B.
Published: October 2018

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.
Three-Dimensional Analysis Techniques—Part 3: Color-Coded System for Three-Dimensional Measurement of Bone and Ratio of Soft Tissue to Bone: The Analysis
Authors: Mccance Andrew M. , Moss James P. , Fright W. Rick , Linney Alf D.
Published: October 2018

A new color-coded method of illustrating three-dimensional changes in the bone and the ratio of soft tissue to bone movement is described. The technique is illustrated by superimposing preoperative and 1-year postoperative CT scans of three patients following bimaxillary surgery. The method has proved to be a very simple, effective, and readily interpreted method of quantifying both bone and the ratio of movement of the overlying soft tissues across the face following surgery.