bluuebunny/crossref_metadata_2025_split huggingface dataset:
Three-Dimensional Analysis of Craniofacial Form in a Familial Rabbit Model of Nonsyndromic Coronal Suture Synostosis Using Euclidean Distance Matrix Analysis
Authors: Burrows Annie M. , Richtsmeier Joan T. , Mooney Mark P. , Smith Tim D. , Losken H. Wolfgang , Siegel Michael I. 
Published: October 2018

Objective Simple craniosynostoses produce predictable morphologies of the cranial vault, with growth deficits in a direction parallel to the synostosed suture and compensatory growth at sutures that are perpendicular to and attached to the synostosed one. In coronal suture synostosis, anteroposterior growth is inhibited, with compensatory growth in a transverse direction. Information on growth patterns and influence on other craniofacial regions are not as clear. This study tested the hypotheses that (1), both juvenile and adult rabbits with familial, nonsyndromic coronal suture synostosis exhibit significant size and shape differences of the entire craniofacial region relative to normal rabbits as a result of altered growth patterns and that (2), shape differences of the calvaria will precede those of the basicranium. Design Fifty anatomic landmarks were located on 94 New Zealand white rabbit crania. The crania were divided into a juvenile, six-week-old age category (n = 53) and an adult, 18-week-old category (n = 41) in order to assess shape differences at different ages. Each age category was sorted into three groups based on growth at the coronal suture: normal suturai growth, delayed onset synostosis, and complete synostosis. Landmarks were digitized in three-dimensions, and statistical analyses on shape differences were carried out using Euclidean distance matrix analysis (EDMA). Results and conclusions Results showed that delayed onset synostosis did not produce craniofacial morphology that was different from normal at any age. However, complete synostosis yielded predictable and global craniofacial shape differences at both ages relative to normal skulls, producing an overall shorter, wider cranium with the most markedly compensating regions in a posterosuperior position of the skull. In addition, delayed onset synostosed crania showed no shape differences in the basicranium, relative to normal crania, suggesting primacy of the calvaria in this model of coronal synostosis. However, further investigations are necessary to verify primacy of the calvaria in this model.
Heterotopic Nasopharyngeal Brain Tissue Associated with Cleft Palate
Authors: Uemura Tetsuji , Yoshikawa Atsushige , Onizuka Takuya , Hayashi Takashi 
Published: October 2018

Objective The occurrence of extracranial brain tissue is rare. Most of the literature describes cases in which it is located around the nose and throat and has been classified as nasal glioma. Even more unusual is heterotopic brain tissue in the nasopharynx. We were able to find only 17 previously reported cases. Of these 17 cases, 6 had heterotopic brain tissue located in a cleft palate. This report comments on the identification and treatment of heterotopic brain tissue associated with cleft palate without connection to the central nervous system. Our case subject is a 10-month-old girl diagnosed with heterotopic nasophranygeal brain tissue associated with cleft palate. Results Excision and palatoplasty were performed conjunctively with excellent results. Conclusions Simultaneous excision of heterotopic nasopharyngeal brain tissue and palatoplasty of the cleft palate is an excellent option for treatment of these cases.
Advantages of Percutaneous Hollow Needle Technique for Iliac Bone Harvest in Alveolar Cleft Grafting
Authors: Hardy Stephen P. , Wilke Robert C. , Doyle John F. 
Published: October 2018

Objective To evaluate the percutaneous hollow needle technique for bone harvest to determine if morbidity, from the bone donor site can be reduced significantly. Methods A retrospective chart review was performed evaluating all patients undergoing alveolar bone grafting at our institution from January 1992 through December 1996. Patients who underwent additional major procedures were excluded. Group I consisted of 12 patients in whom the percutaneous technique was utilized. The patients had an average age of 11.1 years (range: 8 to 15 years). Six were male and six were female. One had a bilateral cleft. Group II consisted of 15 patients in whom the conventional open technique for iliac crest bone harvest was used. They had an average age of 13.1 years (range: 7 to 31 years). Six were male and nine were female. Two had bilateral clefts. Minimum follow-up was 6 months. We evaluated intraoperative blood loss, total postoperative analgesia requirement, and length of hospital stay based on a retrospective hospital chart review. Results A significant difference was found between the two groups regarding intraoperative blood loss (group I: 83.3 cm3, group II: 208 cm3; p = .0015), postoperative total analgesia requirement (group I:0.04 mg/kg, range: 0 to 0.17 mg/kg; group II: 0.34 mg/kg, range: 0.03 to 0.74 mg/kg; p = .0002), and length of hospital stay (group I: 1.0 days, group II: 2.13 days; p = .0001). There was no significant change in these results when bilateral clefts were excluded. Conclusion Iliac bone graft harvest using the percutaneous hollow needle technique results in less blood loss, decreased postoperative pain, and shorter hospital stays compared with the open technique.
Mandibular Lengthening by Distraction Osteogenesis in Children—A One-Year Follow-Up Study
Authors: Huang Chiung-Shing , Ko Wen-Ching , Lin Wen-Yuan , Liou Eric Jein-Wein , Hong Kai-Fong , Chen Yu-Ray 
Published: October 2018

Objective The present study was undertaken to document whether mandibular length could be increased by distraction osteogenesis in children and what happens to the mandibular position 1 year after the distraction. Methods Five patients with hemifacial microsomia (four unilateral, one bilateral) had distraction osteogenesis performed to lengthen the mandible. All patients had cephalometric and panoramic radiographs obtained before and 3, 6, and 12 months after the distraction osteogenesis. Results The total mandibular length (Co-Gn) on the affected side was increased by 11.2 mm after the distraction and remained stable during a 1-year observation period. However, the increased mandibular length tended to move the chin downward (8 mm) more than forward (2 mm) during the same period. Conclusions Distraction osteogensis can be used to lengthen the hypoplastic mandible. However, most of the increased mandibular length moved the chin downward more than forward.
Factors Influencing Changes in Maxillary Arch Dimensions in Unilateral Cleft Lip and Palate Patients until Six Months of Age
Authors: Stellzig Angelika , Basdra Efthimia K. , Hauser Christine , Hassfeld Stefan , Komposch Gerda 
Published: October 2018

Objective The objectives of the present study were (1) to investigate whether growth increments until 6 months of age are influenced by particular factors, (2) to analyze whether anterior cleft reduction is dependent on the extent of the cleft width at birth, and (3) to examine the correlation between maxillary measurements at birth and the anterior cleft width at 6 months of age. Design The study design was prospective and longitudinal. Setting Heidelberg University Hospital Interdisciplinary Cleft/Craniofacial Center. Patients and method The longitudinal records of 34 patients (24 male and 10 female) with complete unilateral cleft lip, alveolar ridge, and hard and soft palate were included in this study. All patients were treated with the same protocol. All participants were assessed at 0 and 6 months of age. Maxillary plaster casts of the patients were analyzed using a computer-controlled three-dimensional digitizing system. Main outcome measure Maxillary models were measured and compared to putative factors influencing growth. Results No statistically significant differences were found between maxillary growth changes and increases in weight and length. Similarly, there was no significant interaction between the extent of the alveolar cleft width at birth and its reduction prior to lip closure. In contrast, significant differences of maxillary growth increments could be found between male and female patients. Stepwise regression analysis demonstrated a correlation between maxillary measurements at birth and growth increments. Conclusion The results of the study indicate that gender plays a certain role in growth changes within the first 6 months of age.
Costochondral Grafts Replacing the Mandibular Condyle
Authors: Ross R. Bruce 
Published: October 2018

Objective The purpose of this study was to determine the success rate of costochondral bone grafts used to replace absent or nonfunctioning temporomandibular joints and the subsequent growth of these grafts when placed in young children. Design This is a retrospective study of all cases with adequate follow-up records that were treated at the Craniofacial Centre at Toronto's The Hospital for Sick Children from 1974–1986. A total of 55 patients were evaluated, of whom 13 were growing children. Results The findings suggest that there was increased success when surgery was performed at an early age. Poorer results were achieved when previous surgery had been performed or when pathology was present. Growth of the graft did not always equal the growth of the “normal” side, but in most cases a satisfactory symmetry was achieved. Several cases exhibited excessive overgrowth. Conclusions Surgery at 4 to 5 years of age will alleviate the impact of a severe facial deformity on the child during the early school years, when self-esteem is fragile and patterns of social interactions are developing. Development of the dentition is better if the jaw relationship is close to normal at an early age. It would appear that early temporomandibular joint (TMJ) construction by costochondral grafting is, at present, the method of choice for severe hemifacial microsomia.
Ellis-van Creveld Syndrome: Craniofacial Morphology and Multidisciplinary Treatment
Authors: Susami Takafumi , Kuroda Takayuki , Yoshimasu Hidemi , Suzuki Ruri 
Published: October 2018

Objective The craniofacial morphology and multidisciplinary treatment of a patient with Ellis-van Creveld Syndrome are presented. Patient The patient presented is female. Her treatment began at the age of 10 years 6 months. She exhibited small stature with acromelic shortening of the extremities, postaxial syndactyly, and dysplastic nails. In the oral region, hyperplastic frena, absent anterior teeth, and small barrel-shaped teeth were noted. Setting and intervention The patient was treated at the University Hospital, Faculty of Dentistry, of the Tokyo Medical and Dental University. The treatment was divided into three stages: growth observation during which the patient wore acrylic plates with artificial anterior teeth; surgical-orthodontic treatment with saggital split ramus osteotomy; and prosthodontic treatment with removable partial dentures. Main outcome measures Cephalometric analyses were performed to evaluate the changes in craniofacial morphology with growth during treatment. Results Characteristic craniofacial morphology included a small posterior cranial base, small maxilla, and a large mandible with an increased gonial angle. Maxillo-mandibular relation was skeletal class III, and skeletal open bite was found. The skeletal class III relationship was progressive with growth. The patient was satisfied by the results of the multidisciplinary treatment. Conclusion The craniofacial morphology of the patient demonstrated disturbed growth of the cartilage bones. An acceptable result was obtained through combined surgical-orthodontic-prosthodontic treatment.
Harvesting Iliac Bone Graft: Decreasing the Morbidity
Authors: de la Torre Jorge I. , Tenenhaus Mayer , Gallagher Pamela M. , Sachs Stephen A. 
Published: October 2018

Objective: This is a review of modifications made to the classic technique for harvesting bone graft from the iliac crest. Prior techniques for harvesting iliac bone often resulted in significant postoperative pain, disability, and a cosmetically unacceptable scar and contour deformity. Design: A retrospective review of patients who underwent bone graft harvest over a 7-year period was done. Interventions: The modifications described use a skin incision medial to the anterior superior iliac crest. The medial aspect of the iliac crest is elevated along the midsagital axis of the crest. The medial cap is reflected outward, exposing cancellous bone. Results: A review of 51 patients demonstrated only two minor self-limiting complications. Conclusions: This procedure provides an abundant supply of both cortical and cancellous bone, an aesthetically acceptable scar, and decreased patient discomfort.
Associated Malformations in Cases with Oral Clefts
Authors: Stoll C. , Alembik Y. , Dott B. , Roth M.P. 
Published: October 2018

Objective Infants with oral clefts (OCs) often have other associated congenital defects. The reported incidence and the types of associated malformations vary between different studies. The purpose of this investigation was to assess the prevalence of associated malformations in a geographically defined population. Method The prevalences at birth of associated malformations in infants with OCs were collected between 1979 and 1996 on all infants born in the area covered by the registry of congenital anomalies of Northeastern France in 238,942 consecutive births. Results Of the 460 cleft infants born during this period, 36.7% had associated malformations. Associated malformations were more frequent in infants who had cleft palate (46.7%) than in infants with cleft lip and palate (36.8%) or infants with isolated cleft lip (13.6%). Malformations in the central nervous system and in the skeletal system were the most common other anomalies, followed by malformations in the urogenital and cardiovascular systems. Weight, length, and head circumference of children with OCs and multiple associated malformations were lower than in controls, as was the weight of the placenta. Prenatal diagnosis was rarely done by fetal ultrasonographic examination in isolated clefts. However, even in multiple associated malformations, prenatal diagnosis by fetal ultrasonographic examination had a low sensitivity, 31.6%. Conclusion The overall prevalence of malformations, which was one in more than three infants, emphasizes the need for a thorough investigation of infants with clefts. A routine screening for other malformations especially skeletal, central nervous system, and cardiac defects may need to be considered in infants with clefts, and genetic counseling seems warranted in most of these complicated cases.
Crouzon Syndrome with Acanthosis Nigricans: Case Report and Mutational Analysis
Authors: Nagase Takashi , Nagase Miki , Hirose Shigehisa , Ohmori Kitaro 
Published: October 2018

Objective To describe the 22nd case of Crouzan syndrome with acanthosis nigricans, a hyperkeratotic skin disorder with hyperpigmentation. Methods DNA analysis and sequencing of the FGFR3gene were performed. Results The 13-year-old Japanese boy described here also had dyspnea, facial palsy, sensorineural hearing loss, and skeletal and mental retardation. Examination of a skin biopsy specimen revealed the typical findings of acanthosis nigricans. Genetic analysis revealed the Ala391Glu mutation in one FGFR3 gene. Conclusions Crouzon syndrome with acanthosis nigricans is a distinct clinical entity different from classic Crouzon syndrome.
Three-Dimensional Analysis of the Child Cleft Face
Authors: Duffy S. , Noar J. H. , Evans R. D. , Sanders R. 
Published: October 2018

Objective This study examined the facial surfaces of cleft children and unaffected children aged 8–11 years with the aim of identifying and assessing differences in their facial surface morphology. The investigation was carried out using an Optical Surface Scanner, an instrument that utilizes laser light to construct and archive a three-dimensional image of the face suitable for linear measurement and direct surface comparisons. Design, Setting, and Patients Thirty-nine cleft lip and palate (CLP) patients and 25 unaffected subjects were voluntarily recruited from two southeast England hospitals. A range of linear facial measurements was compared. Three-dimensional differences between the cleft subgroups and the control group were visualized by superimposition of averaged cleft scans over the averaged control group images. Results Statistically significant dimensional differences (p ≤ .05) in interocular width, nose base widths, mouth widths, and nose base/mouth width ratios were found between the cleft group and the control group. Qualitative differences over the whole of the face were readily demonstrated between the groups by superimposition. Face width and submandibular area depth differed consistently between the groups, the cleft face appearing narrower with a deeper submandibular area. Conclusion Significant differences exist between the facial surface morphology of CLP patients and control subjects.
Descriptive Epidemiology of Nonsyndromic Cleft Lip with or without Cleft Palate in Shanghai, China, from 1980 to 1989
Authors: Cooper Margaret E. , Stone Roslyn A. , Liu You-E , Hu Dan-Ning , Melnick Michael , Marazita Mary L. 
Published: October 2018

Objective The purpose of this study was to characterize nonsyndromic cleft lip with or without cleft palate (CL±CP) in an Asian population. Design Birth prevalence was assessed in a large birth series in Shanghai, China. A 1:3 sex-age-hospital matched case-control design was used to assess the effects of parental ages and pregnancy history on risk of CL±CP. Participants Records of live births from 1980 to 1989 in 22 hospitals in Shanghai, China, were reviewed, comprising 541,504 consecutive births, which is by far the largest such Chinese sample ever investigated. The case-control study included 528 (308 male, 220 female) nonsyndromic CL±CP cases and 1563 (912 male, 651 female) controls. Results From 1980 to 1989, the overall birth prevalence was 1.2 per 1000 live births with statistically significant seasonal variation (more CL±CP births in January to July). The overall male:female ratio was 1.40:1. For males, statistically significant associations were identified with maternal age for the most severe clefts (bilateral overall, and also bilateral CL+CP subgroup). For females, statistically significant association was shown for pregnancy age with birth order (overall and in most subgroups). Conclusions The birth prevalence of CL±CP in this Asian population was similar to published Caucasian rates. The observed seasonal variation would be consistent with possible environmental factors. Significant associations with maternal age, pregnancy age, and birth order warrant additional study of pregnancy history in Asian CL±CP.
Craniofacial Comparisons in 22-Month-Old Lip-Operated Children with Unilateral Complete Cleft Lip and Palate and Unilateral Incomplete Cleft Lip
Authors: Hermann N.V. , Jensen B.L. , Dahl E. , Bolund S. , Kreiborg S. 
Published: October 2018

Objective The objective of the study was to analyze the craniofacial morphology in infants with unilateral complete cleft lip and palate (UCCLP) in which the lip and the anterior part of the palate had been surgically closed at 2 months of age and to compare the morphology with that of a control group with unilateral incomplete cleft lip (UICL) in which the lip had also been surgically closed at 2 months of age. Design The sample consisted of a total of 108 cleft children all fulfilling the entry criteria, besides diagnosis, as follows: The child was of Danish origin; the age of the child was between 650 and 750 days (approximately 22 months) at the time of examination; the child was healthy except for its single cleft malformation; the surgical procedure in each group had been performed at about 2 months of age by the same surgeon. The surgical methods used were a Tennison procedure (UICL group) and a Tennison procedure supplemented by palatovomer plasty (UCCLP group). Methods The method of investigation was infant cephalometry in the lateral, frontal, and axial projections. Linear, angular, and area variables describing the craniofacial morphology were calculated and supplemented by mean plots from the cephalometric projections in the two groups. Results and Conclusions Statistical analysis based on Student's t test showed that the facial morphology in the 22-month-old UCCLP group differed significantly from that of the UICL group. The most pronounced differences were found in the maxillary complex and the mandible. The deviations observed in the UCCLP group at 22 months of age were similar to those previously observed at 2 months of age. However, several of the dysmorphic traits had become less pronounced; some had remained the same; and a few had become worse with time.
Unilateral Cleft Lip Repair—State of the Art
Authors: Schendel Stephen A. 
Published: October 2018

Objective A number of surgical techniques are utilized to correct the unilateral cleft lip, including variations of the rotation-advancement technique. This attests to the variability of the original deformity and the esthetic and functional results from any one technique, especially those based on traditional geometric rearrangement of the skin and associated tissues. Results Most recent advances in cleft lip repair have occurred in two main areas. The morphological result has been improved by functional muscular reconstruction of the lip with or without orthopedic molding. Early correction of the nasal deformity has also been readvocated based on newer principles with excellent results demonstrated. Conclusion Further work continues in these areas and improved outcomes will continue to be seen along with a clearer understanding of surgical affects on growth and development.
Nasalance Score Variation in Normal Adult Japanese Speakers of Mid-West Japanese Dialect
Authors: Tachimura Takashi , Mori Chihiro , Hirata So-Ichiro , Wada Takeshi 
Published: October 2018

Objective The aims of this study were to examine nasalance score variation for normal adult Japanese speakers of Mid-West dialect and the gender difference in average mean nasalance score. Design Nasalance scores were obtained using a nasometer model 6200. The sample stimulus “Kitsutsuki passage,” constructed of four sentences containing no Japanese nasal sounds, was used three times by each subject. Participants One hundred normal adult speakers (50 women and 50 men) of Japanese served as subjects. The subjects ranged in age from 19 to 35 years of age (24.0 ± 3.2). Main Outcome Measure A mean nasalance score as well as an overall average nasalance value across speakers was calculated for each subject. The average mean nasalance scores between men and women were compared. Results The average mean nasalance score for the normal Japanese speakers was 9.1% (± 3.9). There was no statistically significant sex difference (p < .01). Average mean scores of 9.8% (± 3.5) and 8.3% (± 4.0) were obtained for the female and male speakers, respectively. Conclusions The results provide important information concerning criteria to evaluate hypernasal speech due to velopharyngeal inadequacy of Japanese speakers with cleft palate using the nasometer.