bluuebunny/crossref_metadata_2025 huggingface dataset:
The Presurgical Status of the Alveolar Cleft and Success of Secondary Bone Grafting
Authors: Aurouze Catherine , Moller Karlind T. , Bevis Richard R. , Rehm Kelly , Rudney Joel
Published: October 2018

Objective

The primary purpose of this study was to evaluate presurgical status of the alveolar cleft site and success of secondary alveolar bone grafting.

Design

Thirty patient records were retrospectively reviewed. Patients selected for inclusion had isolated cleft of at least the primary palate. Patients with additional anomalies were not selected. The study population consisted of 15 female sites and 16 male cleft sites. There were two bilateral cleft lip and palate (CLP) patients and 28 unilateral CLP patients. The age at the time of the secondary alveolar bone grafting ranged from 7 years to 14 years, 4 months.

Setting

The study was conducted at the Cleft Palate Clinic at the University of Minnesota, School of Dentistry.

Method

Presurgical radiographs taken at least 1 month prior to the secondary bone grafting and postsurgical radiographs taken at least 6 months after bone surgery were measured. Measurements included size of the cleft defect and bone support for distal and mesial teeth adjacent to the cleft. Evaluation of success was determined on the basis of postsurgical measurements of satisfactory, intermediate, and unsatisfactory outcomes.

Results and Conclusion

The size of the cleft defect was not correlated with the success rate of the secondary alveolar bone grafting. If the amount of distal bone support for the mesial tooth was the same as those in a periodontally healthy individual, a satisfactory outcome was 5.8 times more likely. If the amount of mesial bone support for the distal tooth was the same as those in a periodontally healthy individual, the satisfactory outcome was 3.8 times more likely. Although not a primary purpose of the study, it was found that in this study population, if the patient was female, a satisfactory outcome was 3.8 times more likely.
A New Technique for the Quantitative Analysis of Cranial Suture Biology
Authors: Sherick Daniel G. , Buchman Steven R. , Goulet Robert W. , Goldstein Steven A.
Published: October 2018

Objective

Our objective was to assess the ability of the microcomputed tomography scanner to correctly image normal and synostosed cranial sutures at the ultrastructural level.

Design and Methods

Two specimens of coronal sutures were collected from operative specimens. After appropriate preparation, histological sections were obtained and stained with toluene blue for evaluation. Representative histological sections were compared to microcomputed tomography slices.

Results and Conclusions

With microcomputed tomography, we successfully imaged one normal and one synostosed human coronal suture and performed a quantitative analysis of these specimens. Microcomputed tomography scanning was found to be a highly accurate imaging device for the evaluation of cranial suture development. Microcomputed tomography offers three-dimensional imaging at the microscopic level and allows for rapid quantitative analysis of bone architecture, including several measurements unavailable through histologic analysis. We believe that microcomputed tomography can play an important role in imaging and in the quantitative analysis of the stereology of bone microarchitecture. Among its advantages, microcomputed tomography is able to image many more slices than are obtainable through histology, and the method is not prone to human error. Microcomputed tomography slices are generated without destruction of the specimen and without loss or corruption of reproducible data. Structure-oriented slices from microcomputed tomography together with cellular-oriented sections from histology are complementary in the overall quantitative analysis of cranial sutures.
The Basis for Presurgical Orthopedic Treatment of Infants with Unilateral Complete Cleft Lip and Palate
Authors: Koželj Vesna
Published: October 2018

Objective

The purpose of this study was to describe the posttreatment morphology of the upper part of the oral cavity of complete unilateral cleft lip and palate (UCLP) patients and to compare it to noncleft contemporaries. Patients were treated according to a protocol designed to keep a proper resting posture of the oral cavity.

Design

Retrospective study on dental casts.

Setting

The study was performed at a maxillofacial center serving a population of 2 million inhabitants. Data for noncleft subjects are the result of a longitudinal study at the same institution.

Patients

Twenty-one Caucasian UCLP patients (13 males, 8 females) aged 5 to 9 years with no other congenital anomalies and no postoperative orthodontic treatment.

Interventions

Each patient received 5 to 6 months of preoperative orthopedics with a passive plate and external nonelastic strapping with definitive lip repair at age 5 to 7 months, soft palate repair at 11 to 15 months, and hard palate repair with mucoperiosteal closure of the alveolus at 30 to 36 months. Each patient was compared to the mean values obtained from a longitudinal study of a group of 25 healthy noncleft children of the same ethnic group (11 males, 14 females).

Results

Analysis of dental casts indicated that 16 patients had a width, depth, and length of the alveolar arch in the range of the mean normal values minus two standard deviations. Their analyzed palates were flatter than normal. Six of 21 children had too small an alveolar arch for their ages, and they did not acquire a correct posture of the oral cavity.

Conclusion

The results indicate that the upper part of the oral cavity of UCLP patients can reach the dimensions of noncleft contemporaries despite surgery.
Genetics of Nonsyndromic Cleft Lip and Palate: A Review of International Studies and Data regarding the Italian Population
Authors: Carinci Francesco , Pezzetti Furio , Scapoli Luca , Martinelli Marcella , Carinci Paolo , Tognon Mauro
Published: October 2018

The aims of this review are (1) to illustrate current knowledge of the mode of inheritance and the loci involved in the cleft lip and palate and (2) to summarize the results of our investigations, which were carried out in Italy. It is well established that nonsyndromic cleft of the lip with or without the palate (CL±P) and cleft palate only (CPO) are separate entities. Genetic heterogeneity has been observed in CL±P, which involves different chromosome regions, mainly 6p23 (OFC1), 2q13 (OFC2), and 19q13.2 (OFC3), as well as other loci, such as 4q25-4q31.3 and 17q21. Furthermore, an interaction between different genes has been suggested in the oligogenic model. In one case at least, an OFC1 and OFC2 interaction has been demonstrated.

The mode of inheritance of CPO is compatible with a recessive single major gene model, while an association with a candidate gene, mapping on the chromosome region 2q13/TGF α, remains to be confirmed.
Histological Evaluation of Autogenous Iliac Particulate Cancellous Bone and Marrow Grafted to Alveolar Clefts—A Preliminary Report of Five Young Adult Cases
Authors: Iino Mitsuyoshi , Ishii Hiroaki , Sato Junichi , Seto Kanichi
Published: October 2018

Objective

We histologically evaluated iliac particulate cancellous bone and marrow (PCBM) grafted to alveolar clefts in five young adults with cleft lip and palate.

Patients and Methods

Five young adults with cleft lip and palate underwent secondary alveolar bone grafting. The mean age of the patients was 21.2 years (range = 17 to 27 years). Bone specimens were taken from the graft site 5 to 10 months after the surgery simultaneously with implant fixture placement (two cases), vestibuloplasty (two cases), and rebone grafting (one case). These five bone specimens were prepared and stained with hematoxylin and eosin and examined microscopically.

Result

One specimen, taken 5 months after surgery, showed immature trabecular bone partially lined by osteoblasts. The remaining four specimens showed well-mineralized trabeculae and fatty change in the marrow space.

Conclusion

In young adult cases, PCBM remodeled after grafting and became mature bone about 5 to 6 months after the surgery.
Laryngeal Airway Resistance in Cleft Palate Children with Complete and Incomplete Velopharyngeal Closure
Authors: Guyette Thomas W. , Sanchez Anita J. , Smith Bonnie E.
Published: October 2018

Objective

This study investigated the effect of velopharyngeal insufficiency on aerodynamic measures of laryngeal function in children with cleft palate.

Design

Data were analyzed using analysis of covariance. The independent variable was velopharyngeal closure, and the dependent variables were laryngeal resistance, laryngeal airflow, and transglottal pressure. Age and gender were covariates.

Setting

The data were collected at The Craniofacial Center, University of Illinois, a tertiary health care center located in Chicago.

Patients

Thirty-six children with cleft palate were recruited from among the patients at The Craniofacial Center. Ten children with velopharyngeal areas > 5 mm2 during oral speech were placed in the incomplete closure group, while 26 children with areas < 1 mm2 were placed in the complete closure group.

Outcome Measures

The three dependent variables (transglottal pressure, transglottal airflow, and laryngeal resistance) were measured.

Results

Laryngeal resistance and transglottal pressure were significantly higher, and transglottal airflow was significantly lower in the group with complete closure.

Conclusions

In summary, cleft palate patients with complete velopharyngeal closure exhibited higher laryngeal resistances than those with incomplete closure.
Facial Growth in a Cleft Palate Patient Treated with the Herbst Appliance: A Long-Term Profile Roentgenographic and Roentgen Stereometric Analysis of Profile Changes and Displacement of the Jaws
Authors: Sarnäs Karl-Victor , Rune Bodil , Åberg Magnus
Published: October 2018

Objective

To monitor facial development in a patient with cleft palate who was treated with the Herbst appliance. Monitoring was in terms of changes in the skeletal profile and growth in the circummaxillary sutures and temporomandibular joints (TMJs).

Design

Prospective profile roentgenography (between the ages of 6 and 20 years) and roentgen stereometric analysis (between the ages of 8 and 19 years).

Setting

Center for Craniofacial Anomalies and Department of Plastic and Reconstructive Surgery, Malmö University Hospital, Malmö, Sweden.

Patient

Boy with cleft of the soft and posterior part of the hard palate and marked facial convexity.

Interventions

Surgical repair of the soft palate at age 9 months, velopharyngeal flap at age 8 years, and insertion of implants under general anesthesia and treatment with the Herbst appliance at age 11 years. Roentgen examinations were performed in connection with continued clinical evaluations and treatment.

Main outcome Measures

Profile roentgenograms were traced and measured by one of the authors using conventional point-based analysis. Stereo roentgenograms were digitized by the Department of Orthopedic Surgery, Malmö University Hospital.

Results

The direction of profile changes was partly discordant with the direction of articular growth in the circummaxillary sutures and TMJs. The successful treatment result was accomplished by a temporary influence on sagittal growth direction in the circummaxillary sutures and on rotational growth direction in the TMJs, combined with a favorable natural remodeling and articular growth pattern.

Conclusions

The mode of growth by which treatment aims were reached was partly unexpected, i.e., discordant with the generally accepted principal concept that treatment with the Herbst appliance positions the mandible forward.
Epignathus Teratoma: Report of Three Cases with a Review of the Literature
Authors: Vandenhaute B. , Leteurtre E. , Lecomte-Houcke M. , Pellerin P. , Nuyts J.P. , Cuisset J.M. , Soto-Ares G.
Published: October 2018

Three cases of epignathus teratoma associated with other midline anomalies are reported. The first case involved Pierre Robin sequence and a bifid tongue. The second case was characterized by two teratomas, a meningoencephalocele, and a cleft lip and nose. The third case had Pierre Robin sequence associated with duplication of the pituitary gland and hypoplasia of the corpus callosum.
Posterior Pharyngeal Flap and Sphincter Pharyngoplasty: The State of the Art
Authors: Sloan Gerald M.
Published: October 2018

Surgical management of velopharyngeal insufficiency by attachment of posterior pharyngeal flap or construction of sphincter pharyngoplasty is reviewed. Posterior pharyngeal flap surgery is well established, with a long history dating back to the 19th century. Flaps have been based superiorly, inferiorly, or laterally. There have been reports of airway obstruction and obstructive sleep apnea associated with posterior pharyngeal flap surgery. The concept of surgical creation of a dynamic sphincter pharyngoplasty to provide velopharyngeal closure was first introduced by Hynes in 1950. Hynes and others have proposed several subsequent anatomic modifications. Airway dysfunction has also been reported following sphincter pharyngoplasty, but may not be as frequent or severe as with posterior pharyngeal flap. While several studies have compared posterior pharyngeal flap and sphincter pharyngoplasty in terms of speech outcome or complications, there is not, as yet, a consensus regarding the specific choice of one versus the other for surgical management of velopharyngeal insufficiency.
Quantitative Analysis of the Velopharyngeal Sphincter Function during Speech
Authors: Poppelreuter Sabine , Engelke Wilfried , Bruns Thomas
Published: October 2018

Objective

This study reports a new concept for quantitative analysis of velopharyngeal function using electromagnetic articulography and simultaneous transoral video endoscopy.

Participants

The velopharyngeal closure mechanism was studied in 16 healthy German subjects during production of the vowel-consonant-vowel sequences /afa/ and /apa/.

Design

Velar movements in the sagittal and vertical direction were measured by electromagnetic articulography (EMA) while video endoscopy was used for recording in the sagittal and transverse directions.

Main outcome Measure

A transverse sagittal quotient (TSQ) was defined after the measurement of sagittal and transverse diameters of the velopharyngeal port (VPP) in the video-endoscopic pictures on the basis of a frame-by-frame analysis.

Results

The sphincter morphology was objectively determined with the TSQ base value. A terminal sphincter deformation (TSD) for closure phases of articulatory movement was revealed by a qualitative shape analysis of the TSQ curves.

Conclusions

The assessment of the TSQ of velopharyngeal port diameters and the TSD in the terminal closure period lead to a new dynamic interpretation of velopharyngeal closure patterns.
Role of the Superior Pharyngeal Constrictor Muscle in Forced Breathing in Dogs
Authors: Yaman Zekai , Kogo Mikihiko , Senoo Hitomi , Iida Seiji , Ishii Shoichirou , Matsuya Tokuzo
Published: October 2018

Objective

Respiratory-related electromyographic (EMG) activity of the superior pharyngeal constrictor (SPC) muscle was analyzed during the early stage of forced breathing.

Design

Four adult dogs anesthetized with sodium pentobarbital were used. In the first part of the study, oral and nasal breathing tubes were placed into the respective cavities, and a tracheotomy tube was placed in the second part of the study. Two conditions, the presence (oral-nasal tube breathing) and absence (tracheotomy breathing) of airflow in the upper airway, were achieved in each dog. Following quiet breathing, animals were connected to a closed breathing system, first by an oral-nasal tube and then by a tracheotomy tube. We proposed to induce a forced breathing condition mechanically by using this system for 1 minute. We increased resistance to airflow during forced breathing by means of connecting tubes and a bag. Our aim was not to produce chemical drive but to produce a forced respiration by increasing the resistance to airflow. Tidal volume, breathing frequency, minute volume, chest wall movement, and EMG activity of the SPC muscle were measured and analyzed.

Results

During quiet breathing through an oral-nasal or tracheotomy tube, low-amplitude EMG activity of the SPC muscle corresponding to the expiratory cycle of the respiration was observed. In both study conditions, phasic expiratory EMG activity increased immediately after the advent of the breathing from the closed system. Tidal volumes and frequencies also increased rapidly during forced breathing.

Conclusions

An increase in the resistance to airflow increased the activity of the SPC muscle. This augmented respiratory activity probably assists the patency of the upper airway. The augmented respiratory activity was independent of the local reflex pathways. Respiratory-related activity of the SPC muscle may help dilate and stiffen the pharyngeal airway, promoting airway patency.
Gastroesophageal Reflux in Pierre Robin Sequence—Early Surgical Treatment
Authors: Dudkiewicz Zofia , Sekuła Ewa , Nielepiec-Jałosińska Anna
Published: October 2018

Objective

To assess the prevalence of gastroesophageal reflux (GER) in patients with Pierre Robin sequence and to assess whether early palatoplasty with subperiosteal release of the floor of the mouth eliminates upper respiratory tract infections and gastroesophageal reflux.

Methods

In the period from 1993 to 1997, 24 children with Pierre Robin sequence were treated. Because GER is known to occur in patients with this syndrome, we screened for GER in each child for whom an early palatoplasty was to be performed at 3 months of age. In 21 cases, the presence of GER was confirmed. Within the past 2 years, palatoplasty has been combined with subperiosteal release of the floor of the mouth (SRFM). Prompt clinical improvement and shortened hospital stays have resulted.

Results and Conclusions

Follow-up studies performed several months after the surgery showed no evidence of GER. The authors conclude that an early closure of the impaired palatopharyngeal ring and the elimination of the mechanism that causes the posterior rotation of the tongue can reduce the need for such surgical procedures as glossopexy, gastrostomy, tracheostomy, and Nissen's fundoplication.
Intrasphenoidal Migration of a Premaxillary Kirschner Wire
Authors: KayikçOǧlu Aycan , Karamüsel Sebat , Mavili Emin , Erk Yücel , Benli Kemal
Published: October 2018

Objective

The use of Kirschner wire for the fixation of premaxilla is a well-known method in bilateral cleft lip surgery. We report a case in which the Kirschner wire of the premaxillary fixation had migrated intrasphenoidally.

Results and Conclusions

The foreign body was accidentally discovered during a cephalometric analysis and was taken out surgically through an upper lip sulcus incision. Although the wire remained asymptomatic for 10 years, it constituted a potential danger for intracranial migration.
The State of the Art in Cleft Palate Surgery
Authors: Larossa Don
Published: October 2018

Overview

This update focuses on current practices and controversies in surgical repair of the hard and soft palate posterior to the alveolus. Our current understanding of the advantages and disadvantages of presurgical active and passive manipulation of the hard palate shelves including the use of periosteoplasty is reviewed. The evolution of the multiple methods of repair of the hard and soft palate is given in a historical context along with a discussion of the concerns about timing of palate repair.
Management of Alveolar Cleft Bone Grafting—State of the Art
Authors: Eppley Barry L. , Sadove A. Michael
Published: October 2018

Overview

Bone grafting of the alveolus is an essential step in the reconstruction of the orofacial cleft deformity. Secondary grafting with iliac marrow consistently produces trabecular bone to unify the maxilla and provide odontogenic support. It requires preoperative maxillary alignment, well designed mucoperiosteal flaps, and good oral hygiene to be optimally successful. Its high success rate currently makes it the preferred approach at most centers. Primary grafting with rib results in a unified maxilla, eliminates the oronasal fistula, and does not adversely affect midfacial growth. It assists in preventing maxillary segmental collapse, particularly in the bilateral cleft patient. Whether it can produce enough alveolar bone to support long-term odontogenic needs awaits further clinical documentation in the limited numbers of centers that routinely perform this procedure.
A Method of “Unilateral Operation” for Early Repair of Unilateral Complete Cleft Palate. Preliminary Report
Authors: Song Ruyao , Song Yeguang , Liu Cunming , Ma Haihuan , Zhao Yu , Zhao Ru , Fang Zhen
Published: October 2018

Objective

This article describes a method of “unilateral operation” and the preliminary results of a group of patients with unilateral complete cleft palate undergoing the operation at early age.

Design

The “unilateral operation” consists of four relaxation maneuvers. After all of the four maneuvers have been performed on the deformed side of an unilateral complete cleft palate, the deformed side can be moved posteriorly and medially to contact with the normal side. Then the cleft can be closed without tension.

Results

From 1995 to 1998, 19 cases of unilateral complete cleft palate were repaired with this method at 5–12 months of age. Postoperatively, there were no deaths nor dehiscences. Under the care and guidance of an experienced speech pathologist, 15 of 17 of these children have normal vocal quality at 1–2 years of age.

Conclusions

The “unilateral operation” is a rational, adequate, and safe method for early repair of unilateral complete cleft palate. It's design addresses four principles. First, operating only on the deformed side of a unilateral complete cleft palate leaves the normal side unperturbed. Second, complete relaxation of the deformed side is achieved before closing the cleft. Third, in comparison with conventional procedures, which operate on both sides of the palate, this method has the advantage of less surgical trauma, less blood loss, and shorter time of operation. Fourth, all of these advantages are beneficial to early cleft palate repair, which is an important factor in achieving good speech.
A Comparison of the Effects of Veau-Wardill-Kilner Palatoplasty and Furlow Double-Opposing Z-Plasty Operations on Eustachian Tube Function
Authors: Güneren Ethem , Özsoy Zafer , Ulay Mithat , Eryilmaz Erhan , Özkul Haluk , Geary Philip Michael
Published: October 2018

Objective

The purpose of this study was to compare the effects of Veau-Wardill-Kilner (VWK) two-flap palatoplasty and Furlow double-opposing Z-plasty operations on eustachian tube (ET) opening in patients with cleft palate (CP).

Design

Twenty-six patients underwent CP repair between 1993 and 1997. VWK and Furlow operations were used in 13 patients each. All patients were examined by the otorhinolaryngologist before and after the operations; mean follow-up time was 27.3 months. Secretory otitis media (SOM), as identified by tympanograms, was the comparison criterion used.

Results

The prevalence of SOM was 69% (n = 18) in all CP patients pre-operatively. Among those with preoperative SOM, we did not find any statistically significant difference in the frequency of postoperative SOM between the two techniques.

Conclusion

This study suggests that there is no difference in postoperative ET function between patients undergoing either VWK or Furlow operations, in spite of the differences in muscle positioning in these operations. We discuss the implications of this finding in relation to surgical anatomy and histology of the soft palate muscles.
Prevalence of Intranasal Ectopic Teeth in Children with Complete Unilateral and Bilateral Cleft Lip and Palate
Authors: Medeiros Alexandra Sárzyla , Gomide Marcia Ribeiro , Costa Beatriz , De Carvalho Carrara Cleide Felicio , Neves Lucimara Teixeira Das
Published: October 2018

Objective

The objective of this study was to determine the prevalence of ectopic eruption of intranasal teeth.

Design

This was a retrospective study, where records of children with repaired cleft lip and palate were analyzed.

Setting

The study was conducted at a large craniofacial center, the Hospital for Rehabilitation of Craniofacial Anomalies, USP, Bauru, São Paulo, Brazil.

Subjects

The sample consisted of 815 records from patients with bilateral cleft lip and palate (BCLP) and 1495 records from patients with unilateral cleft lip and palate (UCLP). The age of the subjects was 5 to 10 years old and the groups included both males and females.

Results

The results showed that 0.61% of the children with BCLP and 0.40% of those with UCLP had an intranasal tooth. The prevalence of an intranasal tooth for the whole group was 0.48%, and it appeared to be more common in females.
Comparative Study between Children with and without Cleft Lip and Cleft Palate, Part 1: Cephalometric Analysis
Authors: Zúñiga Claudia , Miralles Rodolfo , Carvajal Raúl , Ravera María José , Contreras Paula , Cavada Gabriel
Published: October 2018

Objective

This study was conducted to compare craniofacial relationships, position, and curvature of the cervical spine between children with cleft lip and cleft palate who had been operated on and children without clefts.

Method

This study was performed in 28 children with mixed dentition. They were divided into two groups. The study group included 14 children with unilateral operated cleft lip and cleft palate, ranging in age from 6 to 12 years, who clinically presented with a short upper lip, abnormal lip seal, and inhibition of sagittal development of the midface that was radiographically assessed. The control group included 14 children without clefts, ranging in age from 8 to 11 years. All of them had normal lip seal, nasal breathing, and a clinically normal body posture.

Design

A lateral craniocervical radiograph in a self-balanced natural head position in an erect posture, and without using a head holder, was taken for each child of both groups, with the mandible in maximum intercuspation and lips in habitual posture. The true vertical was marked on all the films. Specific angular and linear dimensions were used to assess the craniocervical relationships, as were the position of the cervical spine, its curvature, or both.

Results and Conclusions

The study group presented a significant increase in the extension of the head on the neck, forward position of the cervical spine, and a decrease in the curvature of the cervical spine in comparison with the children without clefts. These results are more relevant considering that the study group also presented higher significant values of lower facial height than children without clefts.
Orthodontic Treatment for a Patient with Pierre-Robin Sequence Complicated by Juvenile Periodontitis
Authors: Okada Kinya , Yamashiro Takashi , Tenshin Satoru , Takano-Yamamoto Teruko
Published: October 2018

Objective

To arrest the advancement of periodontitis.

Patient

A 17-year-old boy diagnosed with Pierre-Robin sequence at birth exhibited localized juvenile periodontitis. Severe bone loss and mobile teeth were localized in the incisors and molars, which were irregularly positioned, possibly associated with a residual scar from palatoplasty for a soft palate cleft at an early age.

Conclusion

Combined orthodontic-periodontic-prosthodontic treatment arrested the advancement of the periodontitis and established a stable occlusion.
Speech and Language Issues in the Cleft Palate Population: The State of the Art
Authors: Kuehn David P. , Moller Karlind T.
Published: October 2018

Objective

State-of-the-art activity demands a look back, a look around, and, importantly, a look into the new millennium. The area of speech and language has been an integral part of cleft palate care from the very beginning. This article reviews the development and progression of our knowledge base over the last several decades in the areas of speech; language; anatomy and physiology of the velopharynx; assessment of velopharyngeal function; and treatment, both behavioral and physical, for velopharyngeal problems.

Method

The clear focus is on the cleft palate condition. However, much of what is reviewed applies to persons with other craniofacial disorders and with other underlying causes of velopharyngeal impairment. A major challenge in the next several years is to sort through speech disorders that have a clear anatomic underpinning, and thus are more amenable to physical management, versus those that may be treated successfully using behavioral approaches. Speech professionals must do a better job of finding and applying ways of treating individuals with less severe velopharyngeal impairment, thus avoiding the need for physical management in these persons or ignoring the speech problem altogether.

Conclusion

Early and aggressive management for speech and language disorders should be conducted. For most individuals born with cleft conditions, a realistic goal should be normal speech and language usage by the time the child reaches the school-age years.
Standardization of 3-D CT Measurements for Length and Angles by Matrix Transformation in the 3-D Coordinate System
Authors: Kitaura Hideki , Yonetsu Koichi , Kitamori Hideki , Kobayashi Kazuhide , Nakamura Takashi
Published: October 2018

Objective

We attempted to establish a new 3-D cephalometry with helical computed tomography (CT) by introducing the matrix transformation of the 3D coordinate system.

Subjects and Methods

Three-dimensional cephalometric landmarks on the craniofacial bones were expressed as 3-D vectors originating from the center of the sella. These vectors were standardized by the matrix transformation so that the midsagittal plane and cranial base line coincided with the XZ plane and X axis of the 3-D coordinate system, respectively. We also applied this new method to trace the normal growth of the craniofacial bones in 44 patients with head and neck cancer (age range, 5 to 26 years; 19 women and 25 men).

Results

The accuracy for length measurements was less than 3% of standard error of means with a slice thickness of 1 or 3 mm for 28 out of the 29 length measurements recorded. The precision errors by repeated measurements were 0.8% to 1.0% coefficients of variation for intra- and interobserver variability. Standardization of 3-D vectors representing the cephalometric landmarks allowed us to assess successfully the age-related transition of these landmarks of the patients' craniofacial bones.

Conclusion

A new assessment method for 3-D CT cephalometry has been developed by standardizing cephalometric landmarks using a matrix transformation of the 3-D coordinate system. This new assessment method may offer potential in planning plastic and reconstructive surgery.
Linkage Analysis of Candidate Regions in Swedish Nonsyndromic Cleft Lip with or without Cleft Palate Families
Authors: Wong Fung Ki , Hagberg Catharina , Karsten Agneta , Larson Ola , Gustavsson Maria , Huggare Jan , Larsson Catharina , Teh Bin Tean , Linder-Aronson Sten
Published: October 2018

Objective

To analyze linkage of five candidate regions for nonsyndromic cleft lip with or without palate (CLP) on chromosome 2p13, 4q, 6p23, and 19q13; in addition chromosome 1q32, the locus for van der Woude syndrome, on Swedish CLP families.

Design

Three to five linked microsatellite markers were selected from each candidate region. Polymerase chain reaction (PCR) with fluorescent-labeled microsatellite markers was performed on DNA samples from the participating families. Electrophoresis of the PCR products was performed on a laser-fluorescent DNA sequencer. The genotype data were analyzed with multipoint linkage analysis. Modes of inheritance tested included two autosomal dominant, an autosomal recessive, and a nonparametric model. Multipoint logarithm of odds (LOD) scores were also calculated by assuming genetic heterogeneity.

Participants

Nineteen Swedish multigenerational families with at least two first-degree relatives affected with CLP. Greater than 50% of the families studied show vertical transmission of the clefting phenotype and both inter- and intrafamilial variability were noted.

Results

Cumulative multipoint LOD scores for the whole group of families calculated under autosomal dominant modes of inheritance were negative in all regions and less than −2 except chromosome 6p23. LOD scores calculated under recessive inheritance and the nonparametric model were inconclusive. There was no significant evidence of genetic heterogeneity among the sample group.

Conclusions

The group of Swedish CLP families did not demonstrate significant linkage to any of the five candidate regions examined. This might suggest a new but yet unknown CLP locus or loci in this family group. However, because linkage could not be excluded in some individual families, they should still be tested with candidate genes from these regions.
The Relationship between Temporal Aspects of Oral-Nasal Balance and Classification of Velopharyngeal Status in Speakers with Cleft Palate
Authors: Jones David L.
Published: October 2018

Objective

The purpose of this investigation was to determine whether temporal patterns of oral-nasal balance differentiate speakers with cleft palate who exhibit adequate and inadequate velopharyngeal function.

Design

The Nasal Accelerometric Vibrational Index (NAVI) was used to measure the time course and amplitude of oral-nasal balance during the productions of syllables, words, and sentences. The measures obtained include mean amplitude, time integral (area under the curve), absolute duration, relative duration, rise time, and fall time.

Participants

The subjects for this study were 20 children with repaired cleft palates with or without cleft lip. Ten children (aged 5 through 18 years) were assigned to the velopharyngeal competence (VPC) group (normal oral-nasal resonance, no nasal emission of air). Ten children (aged 3 through 19) were assigned to the velopharyngeal insufficiency (VPI) group (hypernasal speech, velopharyngeal gap observed via nasal endoscopy).

Results

Both mean amplitude and time integral of NAVI were greater in the VPI group than in the VPC group (p < .01). Absolute duration and relative duration of the NAVI signal were greater in the VPI group (p < .01). NAVI rise time was shorter in the VPI group (p < .01), and NAVI fall time was longer in the VPI group (p < .01). The amplitude measures contributed the most to discrimination of speaker group, but the durational measures become increasingly influential as a function of a speech task.

Conclusions

Although amplitude of nasalization may have the strongest perceptual salience in classification of velopharyngeal status, the temporal component may exert greater influence during more complex speech tasks.
Facial Growth in Adulthood after Primary Periosteoplasty or Primary Bone Grafting in UCLP
Authors: Sameshima Glenn T. , Šmahel Zbyněk
Published: October 2018

Objective

The primary research aim of this study was to compare long-term differences in craniofacial morphology at adulthood between two groups of unilateral cleft lip and palate (UCLP) patients: one operated by primary periosteoplasty and the other by primary bone grafting.

Design

The two groups were followed longitudinally at 5-year intervals; this study examined the 15- to 20-year age range.

Setting

University hospital/center located in Prague, Bohemia, Czech Republic.

Patients

17 men with primary bone grafting, 29 men with primary periosteoplasty.

Interventions

Primary bone grafting or periosteoplasty.

Methods

Serial cephalometric radiographs.

Main outcome Measures

Conventional cephalometric and finite element measures.

Results

The mandibular symphysis was larger in the bone-grafted group. The periosteoplasty group displayed a more horizontal growth of the mandible with less rotation than the bone-grafted group. There were no other significant differences.

Conclusions

We conclude that the original outcomes differences observed in childhood and adolescence were masked by skeletal and dental compensations in adulthood. These observations can be attributed to both natural growth and clinical intervention.
Vascular Endothelial Growth Pattern of Endochondral Bone Graft in the Presence of Demineralized Intramembranous Bone Matrix—Quantitative Analysis
Authors: Chow K.M.C. , Rabie A.B.M.
Published: October 2018

Objective

To determine the timely ingrowth of new blood vessels of composite endochondral (EC) bone and demineralized bone matrix (DBM) prepared from intramembranous (IM) origin (EC-DBMIM) and to compare it with EC bone graft alone.

Design

Thirty-two rabbits with 32 critical-size (10 × 5 mm), full-thickness bony defects in rabbit parietal bone were divided into two groups: composite EC-DBMIM group—mplanted with compositeautogenousEC bone and DBMIM; EC bone group—mplanted whEC bone alone. Two rabbitsfrom each group were sacrificed 1, 2, 3, 4, 5, 6, 7, and 14 days after grafting. Neovascularization was assessed by immunohistochemical staining with antihuman angiogenesisrelated endothelial cell antibodies (EN 7/44). Quantitative analysis of neovascularization, represented by percentage area of positive immunohistochemical staining, was performed on 320 sections of the experimental groups by a computer-assisted image analyzer.

Results

Positive immunohistochemical staining was first identified on day 2 post grafting for the composite EC-DBMIM group in comparison with day 4 in the EC bone graft group. The composite EC-DBMIM bone graft group showed earlier and almost 100% more neovascularization when compared with the EC bone graft group.

Conclusion

DBMIM enhances healing and integration of EC bone graft by enhancing vascularization as well as increasing the amount of new blood vessels formed. In clinical cases in which EC autogenous bone is used to graft a large defect such as in cleft palate and craniofacial surgery, DBMIM should allow better integration and healing of the EC bone graft to the host bone.
An Investigation of the Dentition of Parents of Children with Cleft Lip and Palate
Authors: Haria S. , Noar J.H. , Sanders R.
Published: October 2018

Objective

The aim of this investigation was to identify whether any dental features in parents of children with cleft lip and palate can be useful as predictors of clefting.

Methods

The dentition of 28 pairs of parents of children with complete unilateral (17) and bilateral (11) cleft lip and palate and 21 pairs of parents of noncleft children were evaluated. Clinical and radiographic examinations were carried out to identify abnormalities of tooth number and morphology. Study cast assessment was undertaken to evaluate incisor relationship, overjet, over-bite, intercanine widths, and mesiodistal widths of individual teeth, and these data were subjected to statistical analysis.

Results

The prevalence of abnormalities of tooth number in parents of cleft children was similar to those reported elsewhere for general populations. In parents of children with unilateral clefts, there was no tooth-size asymmetry between teeth on the side corresponding to the child's cleft and noncleft sides, respectively. No differences were found in tooth widths and intercanine widths between parents of children with unilateral clefts and parents of children with bilateral clefts. The incisor relationship, overjet, overbite, and intercanine widths were found not to differ statistically between the parents of cleft children and parents of noncleft children.

Conclusions

No predictors of clefting could be identified in the dentition of parents of children with cleft lip and palate.
Evaluation of Primary Dentition in Cleft Lip and Palate Children with and without Natal/Neonatal Teeth
Authors: Cabete Haline Farha , Gomide Marcia Ribeiro , Costa Beatriz
Published: October 2018

Objective

Natal/neonatal teeth are very common in children with complete unilateral and bilateral cleft lip and palate. The extraction of these teeth is the usual treatment in the Hospital for Rehabilitation of Craniofacial Anomalies. The objective of this study was to verify whether these teeth could be of the normal complement or whether they were supernumerary.

Design

The primary dentition of children with cleft lip and palate with and without natal/neonatal teeth was compared at the Hospital for Rehabilitation of Craniofacial Anomalies.

Subjects

The sample consisted of 55 children with complete unilateral and bilateral cleft lip and palate with natal/neonatal teeth and 54 without.

Results

No positive association between these groups and missing lateral incisor or supernumerary or complete dentition was found.

Conclusion

The extraction of the natal/neonatal teeth did not alter the final complement of primary teeth, and these teeth could be the lateral incisor or supernumerary.
Double Tongue, Intraoral Anomalies, and Cleft Palate—Case Reports and a Discussion of Developmental Pathology
Authors: Britto J.A. , Ragoowansi R.H. , Sommerlad B.C.
Published: October 2018

Objective

Isolated cleft palate is the most common presentation of the non-syndromic cleft lip/palate combinations and is multifactorial in etiology. We report two cases of children with clefts of the secondary palate coexistent with double tongue and in either case mandibular epulis or superiorly displaced salivary gland.

Results and Discussion

In each case, the palatal cleft correlated anatomically with the intraoral space–occupying lesion. The ratio of tongue volume to intraoral volume during palatogenesis is discussed with reference to the pathogenesis of cleft palate. These clinical cases propose the model of a unifying sequence of developmental events whereby deformation of palatal shelf elevation results in secondary palatal clefting.
A Possible Otological Complication Due to Maxillary Expansion in a Cleft Lip and Palate Patient
Authors: Aizenbud Dror , Hefer Tsila , Rachmiel Adi , Figueroa Alvaro A. , Joachims Henry Z. , Laufer Dov
Published: October 2018

Objective

We present a possible negative side effect of a sudden onset of secretory otitis media in a 12-year-old boy with unilateral cleft lip and palate who underwent maxillary expansion. The secretory otitis media caused a temporary hearing loss developed during the activation of the expander appliance. The possible causes for this complication are discussed.

Conclusions

Clinicians should be aware of the possible association between maxillary expansion and secretory otitis media.
Treacher Collins Syndrome: Current Evaluation, Treatment, and Future Directions
Authors: Posnick Jeffrey C. , Ruiz Ramon L.
Published: October 2018

Objective

Treacher Collins syndrome (TCS) is an inherited disorder in which there are bilateral symmetric anomalies of the structures within the first and second branchial arches. In general, there is complete penetrance and variable expressivity of the trait. The craniofacial rehabilitation of a child with TCS is tailored to the extent of the deformities involved: the orbitozygomatic region, the maxillomandibular region, the nose, facial soft tissues, and external and middle ear structures.

Conclusion

This article reviews the range of clinical features and specific dysmorphology observed in TCS. Functional and aesthetic objectives are discussed, and a comprehensive staged reconstructive approach is outlined, which may be used as a roadmap for treatment planning.
Standardized Facial Photography of Cleft Patients: Just Fit the Grid?
Authors: Vegter Florine , Hage J. Joris
Published: October 2018

Objective

The objective results of craniofacial surgery are usually evaluated by comparing pre- and postoperative photographs. To allow for optimal comparison, these photographs should be of constant and reproducible lay-out and quality. For this, various techniques to standardize photography have been suggested.

Design, Setting, and Patients

A review of the literature on standardization of medical photography is presented. The advantages and disadvantages of standardizing techniques are evaluated. The difficulty to achieve standardization in cleft patients is illustrated using clinical photographs. Our personal protocol is presented as it tries to optimize comparability of photographs.

Results

Use of a grid or cephalostat will not lead to comparable photographs in cleft patients. Personal protocols also have shortcomings, but they offer reproducible and comparable results. A personal effort of the photographer is essential to adjust any allegedly standardizing protocol to the individual patient.

Conclusion

Photographs are only as good as the photographer. True standardization of facial photographs is a utopia in cleft patients.
Abnormal Brain Morphology in Patients with Isolated Cleft Lip, Cleft Palate, or Both: A Preliminary Analysis
Authors: Nopoulos Peg , Berg Stephanie , Canady John , Richman Lynn , Van Demark Duane , Andreasen Nancy C.
Published: October 2018

Objective

The aim of this study was to determine whether adult men with cleft lip and palate (CLP) have aberrant cerebral morphology.

Design

Brain morphology of 14 adult men with isolated CLP were analyzed and compared with 14 healthy controls matched for sex, age, and parental socioeconomic status.

Setting

The research took place at a large, tertiary care hospital, with participation on an outpatient basis.

Participants

The 14 males with CLP were recruited from a large cleft lip and palate registry, while their 14 matched controls were selected from a registry of healthy volunteers collected via the Mental Health Clinical Research Center.

Results

The males with CLP have significantly smaller cerebellar size (p = .04), significantly larger frontal lobes (p = .02), and significantly smaller temporal and occipital lobes (p = .02; p = .009, respectively). No significant difference in gray/white matter ratios or laterality were found.

Conclusions

Adult males with CLP have a significantly different pattern of brain morphology, compared with healthy controls, which is most likely due to aberrant cerebral development. This study highlights the complex interaction and interdependence of craniofacial and cerebral development.
Dental Health Indices and Caries Associated Microflora in Children with Unilateral Cleft Lip and Palate
Authors: Lucas Victoria S. , Gupta Ragini , Ololade Olubenga , Gelbier Marilyn , Roberts Graham J.
Published: October 2018

Objective

To investigate the dental health and caries related microflora of children with unilateral cleft lip and palate.

Study Group

Sixty children with unilateral cleft lip and palate and matched controls.

Outcome Measures

The decayed, missing, and filled teeth and surfaces in both the deciduous and permanent dentitions. The presence of developmental defects and plaque and gingivitis scores were also recorded. Plaque was collected from 25 of the children and their matched controls from three different sites, which were (1) the first approximal site distal to the cleft, (2) a contralateral anterior site, and (3) a remote site. It was cultured for Streptococcus mutansand lactobacilli. Plaque was collected from two sites in the matched controls.

Results

There was no significant difference in the caries, plaque, and gingivitis scores between the children with cleft palate and the controls. A greater number of enamel opacities were recorded in the control group, and there was a higher prevalence of enamel discoloration in the children with cleft lip and palate. There was no significant difference in the proportion of S. mutanso r lactobacilli at the cleft site, compared with the unaffected site in the study group, although there was an anterior-posterior gradient in the proportion of S. mutans. There was no significant association between the stagnation area at the cleft site and the bacteria associated with dental caries.