bluuebunny/crossref_metadata_2025 huggingface dataset:
Nasometric Values for Normal Nasal Resonance in the Speech of Young Flemish Adults
Authors: Van Lierde K. M. , Wuyts F. L. , De Bodt M. , Van Cauwenberge P.
Published: October 2018

Objective

The purpose of this study was to obtain normative nasalance scores for adult subjects speaking the Flemish language. Additional objectives of the study were to determine if speaker sex played a role in differences in nasalance scores and if significantly different nasalance scores existed for Flemish compared with other languages or dialects.

Design

Nasalance scores were obtained while young Flemish adults read three standard nasalance passages. These passages were an oronasal passage (a text that contained the same approximate percentage of nasal consonants as found in the standard Dutch speech), an oral passage (a text that excluded nasal consonants), and a nasal passage (a reading text loaded with nasal consonants).

Participants

Subjects included 58 healthy young Flemish adults with normal oral and velopharyngeal structure and function, normal hearing levels, normal voice characteristics, and normal resonance and articulation skills.

Methods

The Nasometer (model 6200) was used to obtain nasalance scores for the three reading passages. These three reading passages were designed specifically for use with the nasometer. The nasalance data were analyzed for sex dependence, using Student's t test for each reading passage. This same test was used for comparison of our data with data of other languages.

Results

Normative nasalance data were obtained for the oronasal text (33.8%), the oral text (10.9%), and the nasal text (55.8%). Female speakers exhibited significantly higher nasalance scores than male speakers on the passages containing nasal consonants (normal text, p = .001; nasal text, p = .042). Furthermore, statistically significant cross-linguistic nasality differences were observed. The English and Spanish languages were found to have more nasalance than the Flemish language. For the North Dutch and Flemish languages, this cross-linguistic phenomenon was absent.

Conclusion

These normative nasalance scores for normal young adults speaking the Flemish language provide important reference information for Flemish cleft palate teams. Sex-related differences and cross-linguistic differences were shown.
Clinical Experience with Infants with Robin Sequence: A Prospective Study
Authors: Marques Ilza Lazarini , de Sousa Telma Vidotto , Carneiro Araken Fernando , Barbieri Marco Antonio , Bettiol Heloisa , Pereira Gutierrez Manoel Romeu
Published: October 2018

Objective

To study the clinical course of patients with Robin sequence (RS) during the first 6 months of life.

Design

A longitudinal prospective study of children with RS.

Setting

Hospital de Reabilitação de Anomalias Craniofaciais, Universidade de São Paulo, Bauru-SP, Brazil, 1997 and 1998.

Patients

Sixty-two children were studied from hospital admission to 6 months of age. Thirty-three (53.2%) presented with probable isolated RS (PIRS), 25 (40.3%) presented with syndromes or other malformations associated with RS, and 4 (6.5%) presented with RS with neurological involvement.

Interventions

The type of respiratory tract obstruction was defined by nasopharyngoscopy. The patients with type 1 and type 2 obstruction underwent nasopharyngeal intubation (NPI), and glossopexy was indicated in patients with type 1 obstruction who did not show clinical improvement with this procedure. Tracheostomy was indicated in patients with type 2 obstruction who did not show a good course after NPI, in patients with type 1 obstruction who did not show good course after glossopexy, and in patients with type 3 and type 4 obstruction.

Results

Prone position treatment (PPT) or NPI was the definitive treatment in 25 cases (75.8%) of PIRS and in 13 cases (52%) of syndromes or other malformations. Among the children with type 1 obstruction, 24 (51.1%) were submitted exclusively to PPT and 12 (25.5%) to NPI. With the type 2 groups, only one (12.5%) received PPT, and three (37.5%) were treated exclusively with NPI. All 15 infants treated exclusively with NPI (24.4%) presented with good weight, length, and neuromotor development.

Conclusions

Most patients with PIRS and type 1 obstruction improved without surgical intervention. NPI should be the initial treatment in all patients with RS with type 1 and type 2 obstruction who present with important respiratory and feeding difficulties.
Normative Values of Craniofacial Measurements in Idiopathic Benign Macrocephalic Children
Authors: Evereklioglu Cem , Yakinci Cengiz , Er Hamdi , Doganay Selim , Durmaz Yasar
Published: October 2018

Objective:

To determine normal standard values at each age for head circumference; inner canthal distance; outer canthal distance; near interpupillary distance; distant [far] interpupillary distance; canthal index; and circumference-interorbital index specific for the age, sex, and race in 7- to 16-year-old idiopathic benign macrocephalic male and female children.

Methods:

The measurements of head circumference were obtained in 8736 children (4591 boys and 4145 girls) in the city center of Malatya. From these, macrocephalic children (boys = 103, girls = 93) were chosen and invited to our clinic for detailed examination. Mean values for each parameter were obtained at each age from 7 to 16 years.

Results:

The mean age for male and female children were 11.87 ± 2.87 and 11.72 ± 2.75 years, respectively. The overall mean values for head circumference, inner canthal distance, outer canthal distance, near interpupillary distance, distant interpupillary distance, canthal index and circumference-interorbital index in boys were found to be 57.43 ± 1.46 cm, 31.90 ± 2.41 mm, 89.29 ± 4.34 mm, 58.79 ± 2.97 mm, 62.10 ± 3.23 mm, 35.73 ± 2.24, and 5.56 ± 0.44, respectively. In girls, these values were 56.57 ± 1.24 cm, 31.45 ± 2.65 mm, 87.71 ± 4.11 mm, 58.16 ± 3.46 mm, 61.26 ± 3.66 mm, 35.84 ± 2.31, and 5.56 ± 0.43, respectively.

Conclusion:

Normal values of craniofacial measurements for idiopathic benign macrocephalic healthy children are useful in early identification of some craniofacial syndromes, congenital or posttraumatic telecanthus, epicanthus, and hypo-hypertelorism and of planning surgical intervention. We suggest that the comparison of craniofacial dimensions of macrocephalic healthy children must be performed with normal standards specific for age as well as sex and race.
La Maladie de Grisel: Spontaneous Atlantoaxial Subluxation
Authors: Meek M. F. , Hermens R. A. E. C. , Robinson P. H.
Published: October 2018

Objective:

“La maladie de Grisel” (Grisel's syndrome) is a spontaneously occurring atlantoaxial subluxation with torticollis. We present a case of atlantoaxial subluxation occurring in a 20-year period of pharyngoplasty surgery. The occurrence of a “spontaneous” atlantoaxial subluxation after oral cavity or pharynx operations is rare. Because some neck pain and stiffness are commonly seen after these kinds of operations, we would like to draw attention to this unusual complication. Symptoms associated with a torticollis after an operation in the oral cavity or pharynx requires additional investigation to exclude this rare complication. A review of the available literature concerning etiology and treatment of la maladie de Grisel is presented.
Intracranial Migration of Halo Fixation Pins: A Complication of Using an Extraoral Distraction Device
Authors: Le Bach T. , Eyre James M. , Wehby Monica C. , Wheatley Michael J.
Published: October 2018

Objective:

Distraction osteogenesis is a well-accepted technique in the treatment of patients with hypoplastic craniofacial components. Complications of distraction osteogenesis are well described in the literature. We describe a complication of using an external distraction device in a 9-year-old girl with Pfeiffer.

Intervention and Results:

A modified Lefort III osteotomy was performed for maxillary hypoplasia with application of an external distraction halo device by a pediatric neurosurgeon. A postoperative computed tomography (CT) scan showed 0.5-cm skull penetration of the cranial pins. The pins were repositioned and the patient was followed up on a regular basis until discharge from the hospital. At 3-week follow-up, a CT scan of the head showed migration of the pins 1.5 cm intracranially. The halo was removed and repositioned at a different site. No detectable neurological sequelae from the pin penetration were noted. The patient developed cellulitis at the site of the penetration and was admitted to the hospital for a course of intravenous antibiotics. There were no other complications, and the rest of her treatment course proceeded as planned. A review of the literature on complications of halo usage as well as suggestions for their management in association with distraction osteogenesis is described.
Morphological Characterization of the Levator Veli Palatini Muscle in Children Born with Cleft Palates
Authors: Lindman Rolf , Paulin Gunnar , Stål Per S.
Published: October 2018

Objective:

The aim of this study was to analyze, morphologically and biochemically, one of the soft palate muscles, the levator veli palatini (LVP), in children born with cleft palate.

Subjects and Methods:

Biopsies were obtained from nine male and three female infants in connection with the early surgical repair of the hard and soft palate. Samples from five adult normal LVP muscles were used for comparison. The muscle morphology, fiber type and myosin heavy chain (MyHC) compositions, capillary supply, and content of muscle spindles were analyzed with different enzyme-histochemical, immunohistochemical, and biochemical techniques.

Results:

Compared with the normal adult subjects, the LVP muscle from the infantile subjects with cleft had a smaller mean fiber diameter, a larger variability in fiber size and form, a higher proportion of type II fibers, a higher amount of fast MyHCs, and a lower density of capillaries. No muscle spindles were observed. Moreover, one-third of the biopsies from the infantile subjects with cleft LVP either lacked muscle tissue or contained only a small amount.

Conclusions:

The LVP muscle from children with cleft palate has a different morphology, compared with the normal adult muscle. The differences might be related to different stages in maturation of the muscles, changes in functional demands with growth and age, or a consequence of the cleft. The lack of contractile tissue in some of the cleft biopsies offers one possible explanation to a persistent postsurgical velopharyngeal insufficiency in some patients, despite a successful surgical repair.
Change in Levator Veli Palatini Muscle Activity of Normal Speakers in Association with Elevation of the Velum Using an Experimental Palatal Lift Prosthesis
Authors: Tachimura Takashi , Nohara Kanji , Fujita Yoshinori , Hara Hisanaga , Wada Takeshi
Published: October 2018

Objective:

The purpose of this study was to examine whether mechanical elevation of the velum can directly affect the levator veli palatini muscle (LVP) activity using normal speakers as subjects.

Design:

Each subject was instructed to produce /mu/, /u/, /pu/, /su/, and /tsu/ in a speech task and to blow with maximum possible effort. Smoothed electromyographic activity of the LVP was recorded with an experimental palatal lift prosthesis (PLP) both in place and removed.

Participants:

Four normal speakers were used as subjects.

Results:

LVP activity for all tasks was significantly smaller (p < .001; Student's t test) with the PLP in place than without the PLP for all subjects. An analysis of variance (p < .001) clarified that activity ranges of the LVP were significantly different between the removal and placement conditions for all the subjects.

Conclusion:

The result was similar to that previously obtained for patients with velopharyngeal incompetence wearing a PLP. It is possible that the decrease in the LVP activity in association with placement of a PLP is caused by the direct effect of mechanical elevation, which decreases the distance the velopharyngeal mechanisms must travel for complete closure of the velopharynx.
Nasal Morphology and Shape Parameters as Predictors of Nasal Esthetics in Individuals with Complete Unilateral Cleft Lip and Palate
Authors: Russell K. A. , Waldman S. D. , Tompson B. , Lee J. M.
Published: October 2018

Objective:

The purpose of this study was to assess the ability of shape parameters of nasal morphology to predict esthetics in individuals with complete unilateral cleft lip and palate (CUCLP).

Methods:

This retrospective study involved 28 patients with repaired CUCLP. Nostril morphology was analyzed using nose casts and a video-imaging technique. Calculated shape parameters included area, perimeter, centroid, angle of the principal axis, major and minor moments of area, anisometry, bulkiness, lateral offset, and three-dimensional internostril angles. Esthetics was assessed using a panel of six orthodontists who rated nasal esthetics from frontal, lateral, basal, and three-quarters view slides and from nose casts. Correlations between esthetics and the shape parameters were completed using the entire group as well as using two statistically determined subsets: those with the best and those with the worst esthetics.

Results:

Nasal esthetics was related to only the perimeter and bulkiness parameter ratios. Symmetry of the perimeters between the right and left nostrils positively correlated with better esthetics using the entire sample group while symmetry of bulkiness between the right and left nostrils positively correlated with better esthetics using both the entire sample group and the best and worst subsets.

Conclusions:

Only perimeter and bulkiness showed positive correlations with nasal esthetics. The group of parameters used to assess nostril morphology had neither significant correlation with—nor predictive power for—esthetics. Thus, an assessment of the entire nasal surface topography in three dimensions needs to be completed and assessed with respect to predictability of nasal esthetics.
The Oral Health of Children with Clefts of the Lip, Palate, or Both
Authors: Chapple J. R. , Nunn J. H.
Published: October 2018

Objective:

The purpose of this study was to assess the prevalence of dental caries, developmental defects of enamel, and related factors in children with clefts.

Design:

This cross-sectional prevalence study used standard dental indices for assessment.

Setting:

Children underwent a dental examination under standard conditions of seating and lighting in the outpatient department of a dental hospital as part of an ongoing audit to monitor clinical outcomes.

Participants:

Ninety-one children aged 4, 8, and 12 years were included in the study.

Outcome Measurements

Dental caries were assessed by use of the decayed, missing, and filled index for primary teeth (dmft); Decayed, Missing, and Filled index for permanent teeth (DMFT) according to the criteria as used in the national survey of children's dental health in the United Kingdom (O'Brien, 1994). Developmental defects were assessed using the modified Developmental Defects of Enamel Index (Clarkson and O'Mullane, 1989). Dental erosion was assessed using the criteria derived for the national survey of children's dental health (O'Brien, 1994).

Results:

Caries prevalence increased with age; 63% of patients at 4 years and 34% at 12 years were caries free. The mean dmft for the 4-year-olds was 1.3 with a mean DMFT for the 12-year-olds of 1.8. All the 4-year-olds had evidence of erosion of enamel in the primary teeth (incisors and first molars) and 56% of the 12-year-olds had erosion of permanent teeth (incisors and first permanent molars). Developmental defects of enamel became more prevalent with age, with at least one opacity in 56% of 4-year-olds and 100% of 12-year-olds. Hypoplasia was not found in the primary dentition but affected permanent teeth in 38% of 8-year-olds and 23% of the 12-year-olds.

Conclusion:

This study has shown that dental disease is prevalent in these patients. These assessments not only provide a baseline on oral health parameters in young people with clefts but underline the need for a more aggressive approach to prevention of oral disease to optimize clinical outcome.
Associations between Severity of Clefting and Maxillary Growth in Patients with Unilateral Cleft Lip and Palate Treated with Infant Orthopedics
Authors: Peltomäki Timo , Vendittelli Bruno L. , Grayson Barry H. , Cutting Court B. , Brecht Lawrence E.
Published: October 2018

Objective:

The purpose of this study was to examine possible associations between severity of clefting in infants and maxillary growth in children with complete unilateral cleft lip and palate.

Design:

This was a retrospective study of measurements made on infant maxillary study casts and maxillary cephalometric variables obtained at 5 to 6 years of follow-up.

Setting:

The study was performed at the Institute of Reconstructive Plastic Surgery of New York University Medical Center, New York, New York.

Patients:

Twenty-four consecutive nonsyndromic unilateral complete cleft lip and palate patients treated during the years 1987 to 1994.

Interventions:

All the patients received uniform treatment (i.e., presurgical orthopedics followed by gingivoperiosteoplasty to close the alveolar cleft combined with repair of the lip and nose in a single stage at the age of 3 to 4 months). Closure of the palate was performed at the age of 12 to 14 months.

Results:

Infant maxillary study cast measurements correlated in a statistically significant manner with maxillary cephalometric measurements at age 5 to 6 years.

Conclusions:

The results demonstrate the large variation in the severity of unilateral cleft lip and palate deformity at birth. Patients with large clefts and small arch circumference, arch length, or both demonstrated less favorable maxillary growth than those with small clefts and large arch circumference or arch length at birth.
Craniofacial Morphology, Dentition, and Skeletal Maturity in Four Siblings with Seckel Syndrome
Authors: Kjær Inger , Hansen N. , Becktor K. B. , Birkebæk N. , Balslev T.
Published: October 2018

Objective:

To describe the craniofacial morphology, dentition, and hand maturity in four siblings with Seckel syndrome.

Patients:

Two boys and two girls, with Seckel syndrome. The children studied showed extreme growth retardation, severe microcephaly, bird-headed profile with receding chin, prominent nose, mental retardation, and extremely delayed skeletal maturation. The growth hormone axis and pituitary thyroid function was normal.

Methods:

Skeletal and dental development were investigated from radiographic material, and a cephalometric analysis was performed from profile radiographs.

Results:

The craniums were remarkably small with an extremely short anterior cranial base (−4.3 to −5.5 standard units) and maxillary length (−3.8 to −4.7 SU). Differences in the morphology of the sella turcica were observed in girls and boys. Tooth maturity progressed normally. Tooth agenesis and tooth malformations were observed. Taurodontic root morphology was observed only in the girls. The approximate skeletal maturity showed retardation from 4 years 3 months to 4 years 11 months. Malformations of the hand-wrist skeleton occurred in the epiphyseal ossification centers of the middle phalangeal bone in the second, third, and fourth finger and in the distal phalangeal bone in the fifth finger. The epiphyseal ossification centers were lacking in the middle and distal phalangeal bones of the fifth finger.

Conclusion:

The underlying gene defect in the affected children seemingly affects bone development and growth but not dental maturation and eruption.
Beyond Easy Answers: The Plastic Surgeon and Prenatal Diagnosis
Authors: Matthews Martha S.
Published: October 2018

Objective

To explore how plastic surgeons handle the issue of prenatal diagnosis of cleft lip and palate.

Design

Mail survey.

Participants

The plastic surgeon listed as team contact in the team directory of the American Cleft Palate–Craniofacial Association was identified. In cases in which a plastic surgeon was not listed as a team contact, the first plastic surgeon listed as a team member was identified. A total of 211 surgeons were contacted.

Intervention

Each surgeon was mailed a survey asking for his/her experience in prenatal diagnosis for cleft conditions. Ethical issues concerning the surgeon's personal beliefs about abortion and the potential influence of the surgeon's presentation of information were explored.

Results

A 50% response rate was obtained. Eighty-five percent of respondents were experienced in prenatal counseling; 92% offer consults on a routine basis. Ninety-six percent indicated they did so because they felt it was helpful to families. Treatment (98%), appearance of clefts (96%), cleft etiology (94%), and associated problems (40%) were discussed most commonly. Termination was rarely discussed (5%). Of those who had not met with a family, 82% said they had never been asked to do so. Most respondents felt families sought consultation for information and rarely sought consultation to decide on abortion. The majority felt their personal beliefs about abortion did not influence their willingness to meet with parents. The majority of surgeons in both groups felt it was ethical to give an opinion on pregnancy termination if asked (55% of experienced surgeons, 44% of inexperienced surgeons); however, a significantly larger number of the surgeons who had not met with a family were unsure of their opinion.

Conclusions

Prenatal counseling of parents of a fetus with cleft lip/palate is common among plastic surgeons. There is uniformity in the information discussed. There is no agreement among surgeons about the ethical issues of pregnancy termination and the surgeon's role in that discussion.
Dealing with the Prenatal Diagnosis of Clefting: A Parent's Perspective
Authors: Aspinall Cassandra L.
Published: October 2018

Objectives

Prenatal testing and information is available on an increasing basis for a variety of reasons. There are conflicting standards of care in the delivery of this type of evaluation and even more confusion around access to such services. As physicians and patients evaluate which tests will generate useful information with acceptable levels of risk, the rules of the game constantly shift. Presenting the situation from the viewpoints of both a professional and a parent, issues revolving around uncertainty and reassurance are evaluated. A description of the personal experience of the impact of the discovery of a cleft prenatally is provided.

Conclusions

Finally, efforts are made to raise questions about our motivations behind conducting prenatal testing and also developing some suggestions about how to support families better in our regular clinical work. A challenge is made to providers and patients to do more work prior to examination to explore how the unexpected will be handled. The dilemma about how to facilitate informed consent through full disclosure without overwhelming the patient is also discussed.
Use of U.S. Birth Certificate Data to Estimate the Risk of Maternal Cigarette Smoking for Oral Clefting
Authors: Wyszynski Diego F. , Wu Tianxia
Published: October 2018

Objective

The purpose of this study was to evaluate the relationship between maternal cigarette smoking and the risk of having an offspring with an oral cleft.

Design

This was a large population-based, matched case-control study derived from the United States Natality database for 1997.

Subjects

The sample consisted of 2029 cases with nonsyndromic oral clefts and 4050 nonmalformed controls. Controls were matched to cases on mother's and father's race and child's sex, county of birth, and month of birth. This sample was selected from a total of 3,093,821 births in the United States, which represents 80% of all births in this country during 1997.

Results

The association between maternal cigarette smoking and oral clefts in the offspring was close to the null (odds ratio 1.16; 95% confidence interval (CI) 1.01 to 1.33; one-sided Fisher exact testp = .0207). The comparison and pooling of results to those of a similar study that used the U.S. Natality database for 1996 resulted in a common Mantel-Haenszel odds ratio of 1.33 (95% CI 1.21 to 1.46). The dose-response analysis was slightly significant for all levels of maternal smoking.

Conclusion

This large study confirms that smoking during pregnancy is only a minor risk factor for oral clefting in the offspring.
Craniofacial Anthropometry in a Turkish Population
Authors: Evereklioglu Cem , Doganay Selim , Er Hamdi , Gunduz Abuzer , Tercan Mustafa , Balat Ayse , Cumurcu Tongabay
Published: October 2018

Objective

To present norms and demonstrate the anthropometric variations in fronto-occipital circumference, inner and outer canthal distances, near and distant [far] anatomical interpupillary distance, canthal index, and circumference-interorbital index across age and sex in urban Turkish subjects.

Participants

Three thousand four hundred forty-eight subjects (1852 male, 1596 female) aged 7 to 40 years were included in this study.

Methods

Three age groups were studied: children aged 7 to 15 years, young adults aged 16 to 25 years, and adults aged 26 to 40 years. Mean values for each measured parameter were determined at each age between 7 and 25 years. Subjects were also divided into nine age subgroups to observe the change of each parameter with advancing age.

Results

The fronto-occipital circumference and outer canthal distance of males was significantly (p < .001) wider than females in all age groups. The near and distant interpupillary distances of male subjects were, on average, wider than the female subjects with greater differences with advancing age. Across all subjects aged 7 to 40 years, the mean of all measured parameters and calculated indexes of men and boys was significantly different from girls and women (p < .001). The mean for interpupillary distances in our study in both sexes were found to be similar to Arabian, Hong Kong, and British children; larger than those of Chinese, Black, Indian, and Caucasians; and smaller than those of Mexican children and a mixed European population.

Conclusion

This study clearly shows the anthropometric variation for fronto-occipital circumference, inner canthal distance, outer canthal distance, near and distant interpupillary distance, canthal index, and circumference-interorbital index with age. These developmental data and the normal values of these measurements in healthy subjects are useful for dysmorphologists in the early identification of some craniofacial syndromes, hyper- and hypotelorism, and congenital or posttraumatic telecanthus and of planning surgical intervention. We suggest that the comparison of craniofacial dimensions of a patient must be performed with normal standards specific for age as well as sex and race.
Craniofacial and Dental Manifestations of Proteus Syndrome: A Case Report
Authors: Becktor K.B. , Becktor J.P. , Karnes P.S. , Keller E.E.
Published: October 2018

The Proteus syndrome is a rare congenital hamartomatous condition that is characterized by a wide range of malformations, sometimes involving the face. Common manifestations include partial gigantism, congenital lipomas, and plantar hyperplasia. In this report we describe the craniofacial clinicopathological development in a girl with Proteus syndrome from age 6 to 20 years. The patient had pronounced hemifacial hypertrophy, exostoses in the left parietal region, and enlargement of the inferior alveolar nerve and mandibular canal in the affected region. The dental development of the affected left mandible and maxilla was characterized by extremely premature development and eruption of the primary and permanent teeth and by pronounced idiopathic root resorptions. The multidisciplinary management of the patient and the treatment outcome is reported. A review of the Proteus patients in the literature who exhibited manifestation in the craniofacial region is presented.
A New Scale to Assess Radiographic Success of Secondary Alveolar Bone Grafts
Authors: Witherow H. , Cox S. , Jones E. , Carr R. , Waterhouse N.
Published: October 2018

Objectives:

To propose a new scale for evaluating the position of the bone graft within the cleft and assess its inter- and intraobserver reliability.

Design:

Sixty-six patients (70 cleft sites) over a 14-year period were assessed, 90% of patients retrospectively and 10% prospectively. The radiographs were reviewed by two clinicians in controlled conditions twice, with 1 week between assessments. Both clinicians were blind to patient identity.

Outcome measures:

A new scale subdividing the position of the bone into one of six categories was used. The radiographs were also assessed using the Bergland scale.

Results:

Using the Bergland scale, 62.9% of the cleft sites were type I, 21.4% type II, 4.3% type III, and 5.7% type IV. It was not possible to assess 5.7% of the clefts with this scale because the canine was unerupted. Using the Chelsea alveolar bone graft scale, 58% were category A, 20% B, 7% C, 3% D, 3% E, and 9% F.

Conclusions:

The above scale can be used to determine accurately the position of bone within the cleft site. It can be used in the mixed dentition prior to eruption of the canine. It demonstrated moderate to substantial inter- and intraobserver reliability and offers several advantages, compared with other scales.
Upper Lip Fistulas: Three New Cases
Authors: Sancho M. A. , Albert A. , Cusi V. , Grande C. , Aguilar C. , Morales L.
Published: October 2018

Objective

We present three new cases of congenital upper lip fistula. Two of them were located in the philtrum midline, one of which was associated to a double maxillary frenulum, a medial lip cleft, and a medial cleft of the primitive palate. The other was located in the left side of the vermilion. All three patients had clear fluid discharge through the fistulous orifice without pain. Two of them had a history of recurrent swelling of the philtrum area.

Conclusions

A simple surgical excision is the treatment of choice in these cases, in which the anatomy is preserved; this fact is more consistent with a completed but aberrant development than with focal dysgenesis.
Fibroblast Growth Factor 2 Induces Increased Calvarial Osteoblast Proliferation and Cranial Suture Fusion
Authors: Moursi Amr M. , Winnard Phillip L. , Winnard Alissa V. , Rubenstrunk John M. , Mooney Mark P.
Published: October 2018

Objective:

Craniosynostosis has been associated with fibroblast growth factors (FGFs) and their receptors. The purpose of this study was to quantitatively determine the effect of FGF2 on rat calvarial osteoblasts and a rat cranial suture formation model.

Design:

Fetal rat calvarial osteoblasts were cultured with and without FGF2. Cell attachment and proliferation was determined by alamarBlue dye assay and cell morphology by toluidine-blue staining. In rat calvarial organ culture, postnatal day 15 rat calvariae with dura mater were placed in serum-free media with and without FGF2. A unique quantitative analysis of suture fusion was developed by obtaining measurements of suture bridging in histological serial sections at progressive stages of fusion.

Results:

Attachment for cells treated with FGF2 was similar to control. In contrast, proliferation was higher for cells treated with FGF2 while maintaining an osteoblastic morphology. After 5 days in organ culture, FGF2-treated posterior frontal sutures showed a dramatic increase in fusion, compared with untreated controls. This increased fusion was maintained throughout days 7 and 10 in culture. Also, fusion was enhanced on the dural side of the suture, as is normally observed in vivo, and the normal tissue architecture was maintained.

Conclusions:

These results indicate that FGF2 can promote rat osteoblast attachment and normal cell morphology as well as induce cell proliferation. In calvarial organ culture, FGF2 treatment produced an enhanced suture fusion. These results provide further support for a critical role for FGF2 in cranial suture development. These studies also present a new quantitative approach to evaluating the effect of suture-perturbing growth factors on cranial suture fusion.
Seventeen-Year Follow-Up of a Patient with Median Cleft of the Lower Lip, Mandible, and Tongue with Flexion Contracture: A Case Report
Authors: Ishii Masatoshi , Ishii Yoshimasa , Moriyama Takashi , Enomoto Shoji , Ono Toaki , Ohyama Kimie , Kuroda Takayuki
Published: October 2018

Purpose:

We present a 17-year follow-up of an unusual patient with median cleft of the lower lip, mandible, and tongue with a flexion contracture.

Conclusion:

Timing of the mandibular reconstruction needs to take into account growth of the mandible and masticatory function. In this case, fixation of the mandible performed at the age of 6 years 6 months and reconstruction carried out at 7 years 8 months of age resulted in satisfactory occlusion and masticatory function.
Microsurgical Tissue Transfer for Rehabilitation of the Patient with Cleft Lip and Palate
Authors: Eufinger Harald , Machtens Egbert
Published: October 2018

Objective:

Three case reports of microsurgically revascularized tissue transfer for secondary closure of complex oronasal fistulae in cleft lip and palate patients are reported. One scapular and two radial forearm flaps were used in that respect; the scapular flap was transferred without a skin paddle and was left for secondary epithelialization whereas iliac crest bone was transplanted in the two patients with the forearm flaps in a further surgical step.

Conclusions:

These microsurgical flaps represent solutions in selected cases of oronasal fistulae in patients with cleft lip and palate with extensive scarring, large defects, or both. Alternative free flaps of the vast spectrum available today, however, also deserve consideration.
The Treatment of Hemimandibular Hyperplasia Preserving Enlarged Condylar Head
Authors: Sugawara Yasushi , Hirabayashi Shin-Ichi , Susami Takafumi , Hiyama Shigetoshi
Published: October 2018

Objective

To present a case of hemimandibular hyperplasia (HH) treated with orthognathic surgery that preserves the condyle without disturbing mandibular function.

Methods

A 27-year-old woman with HH was treated with orthognathic surgery preserving the enlarged condylar head. Radiographic examination showed typical enlargement of the right condyle, elongation of the right ascending ramus and mandibular body, and tilted occlusal plane. A mandibular sagittal split osteotomy on the unaffected side and subcondylar ramus osteotomy on the affected side, Le Fort I wedge osteotomy to relevel the tilted occlusal plane, and contouring of the lower mandibular margin were performed.

Results

Excellent results in the full-face appearance and occlusion were obtained. There was no change in the size of the reserved condylar head 4 years postoperatively. In a series of examinations of jaw function with electromyography, mandibular kinesiography, and computer-aided diagnostic axiography, more favorable findings were obtained postoperatively.

Conclusions

In a case of HH without abnormally high growth activity, orthognathic surgery preserving hypertrophic condyle produced functional improvement in addition to good occlusal and aesthetic outcomes.
Speech Development of Children with Cleft Palate before and after Palatal Surgery
Authors: Jones Courtney E. , Chapman Kathy L. , Hardin-Jones Mary A.
Published: October 2018

Objective

This study examined the speech production abilities of children with cleft palate before and after palate repair.

Participants

Twenty-eight children participated in the study, 14 with cleft palate and 14 without clefts matched for age, gender, and mothers’ educational level.

Main Outcome Measures

Comparisons were made between the children with cleft palate before and after surgery for canonical babbling ratios, size of consonant inventories, place and manner characteristics, and early developing sounds [p, b, t, d, k, g, m, n, η]. Also, comparisons were made between the children with cleft palate and their peers without clefts at postsurgery/17 months for the measures described above.

Results

Paired ttests revealed differences in pre- and postsurgery performance of the children with cleft palate for production of canonical syllables and size of consonant inventories. Although no significant differences were noted for place and manner features, production of oral stops doubled from the time before surgery to that after surgery. Specifically, production of the bilabial stop [b] showed significant change over time. Results of independent t tests indicated no difference between groups for production of canonical syllables and size of consonant inventories at 17 months. However, significant group differences were noted for production of stops, oral stops, nasals, glides, and alveolars. The children without clefts produced more stops, oral stops, and alveolars. In contrast, more nasals and glides were seen in the vocalizations of the children with cleft palate.

Conclusions

Children with cleft palate made gains in production of canonical syllables and size of consonant inventories postsurgery; however, they continued to show deficits in production of stops and alveolar place features.
Comparison of Nasalance Scores Obtained from the Nasometer and the NasalView
Authors: Lewis Kerry E. , Watterson Thomas
Published: October 2018

Objective

Nasalance scores obtained from the Nasometer and the Nasal-View were compared for five different sentences.

Design

Vowel content was controlled in the design of the five stimulus sentences. One sentence was loaded with high-front vowels, one with high-back vowels, one with low-front vowels, one with low-back vowels, and one contained a mixture of vowel types.

Subjects

The subjects were 50 elementary school children ranging from kindergarten to sixth grade. Each subject was a native speaker of English, had no history of adenoidectomy, and was not currently enrolled in speech therapy services.

Main Outcome Measures

The main outcome measures were the nasalance scores obtained from the Nasometer and the NasalView for each of the five sentences.

Results

There was a significant difference in the nasalance scores between the Nasometer and the NasalView for four of the five stimuli, but not all differences were in the same direction. For two stimuli, the Nasometer scores were significantly higher, and for two stimuli the NasalView scores were higher. Bivariate correlations between nasalance scores for individual stimuli were in the good range for the Nasometer but poor for the NasalView.

Conclusion

Speech stimuli weighted with different vowel types are differentially affected by the different acoustical filtering used in the Nasometer versus the NasalView. Nasalance scores obtained with the NasalView were qualitatively and quantitatively different from those obtained with the Nasometer. This suggests that the two machines provide different information, and the scores are not interchangeable.
Importance of Soft Tissue for Skeletal Stability in Maxillary Advancement in Patients with Cleft Lip and Palate
Authors: Baumann Arnulf , Sinko Klaus
Published: October 2018

Objective

Assessment of stability of the advanced maxilla after two-jaw surgery and Le Fort I osteotomy in patients with cleft palate based on soft tissue planning.

Subjects

Between 1995 and 1998, 15 patients with cleft lip and palate deformities underwent advancement of a retruded maxilla, without insertion of additional bone grafts. Eleven patients had bimaxillary osteotomies and four patients only a Le Fort I osteotomy. Relapse of the maxilla in horizontal and vertical dimensions was evaluated by cephalometric analysis after a clinical follow-up of at least 2 years.

Results

In the bimaxillary osteotomies, horizontal advancement was an average 4 mm at point A. After 2 years, there was an additional advancement of point A of an average of 0.7 mm. In the mandible, a relapse of 0.8 mm was seen after an average setback of 3.9 mm. In the four patients with Le Fort I osteotomy, point A was advanced by 3.8 mm and the relapse after 2 years was 0.9 mm. Vertical elongation at point A resulted in relapse in both groups. Impaction of the maxilla led to further impaction as well.

Conclusion

Cephalometric soft tissue analysis demonstrates the need for a two-jaw surgery, not only in severe maxillary hypoplasia. Alteration of soft tissue to functional harmony and three-dimensional correction of the maxillomandibular complex are easier to perform in a two-jaw procedure. It results in a more stable horizontal skeletal position of the maxilla.
Chromosome 17: Gene Mapping Studies of Cleft Lip with or without Cleft Palate in Chinese Families
Authors: Peanchitlertkajorn Supakit , Cooper Margaret E. , Liu You-E , Field L. Leigh , Marazita Mary L.
Published: October 2018

Objective

Involvement of loci on chromosome 17, including retinoic acid receptor alpha (RARA) in nonsyndromic oral clefts has been reported in Caucasian populations, although never investigated in Asian populations. The purpose of the present study was to investigate several loci on chromosome 17, including RARA, in Chinese families.

Participants

Thirty-six multiplex families (310 individuals), ascertained through nonsyndromic cleft lip with or without cleft palate surgical probands from hospitals in Shanghai, China, participated in the present study. There were 23 families whose probands had cleft lip and cleft palate (CLP) and 13 with cleft lip alone (CL).

Results

Seventeen markers, spanning chromosome 17 and about 10 cM apart were assessed. Logarithm of odds ratio (LOD) scores (two point and multipoint), model-free linkage analyses, and allelic association tests (transmission/disequilibrium, Fisher's exact tests, and chi-square) were performed on the total family sample, families with CLP probands (CLP subgroup), and families with CL probands (CL subgroup). LOD scores from the two-point analyses were inconclusive. Multipoint analyses rejected linkage except for a few regions in the CL subgroup. However, positive results were found using the model-free linkage and association methods (p < .05). The markers with positive results varied across the CL and CLP subgroups. However, the RARA region and loci nearby yielded consistently positive results.

Conclusion

Genetic variation within the RARA locus or nearby appears to be involved in the pathogenesis of nonsyndromic oral clefts in this population. Furthermore, based on the differing pattern of results in the CL versus CLP subgroups, it appears that the formation of CL and CLP is because of either differing alleles at the same genetic locus or different but related (and/or linked) genes that modify the severity and expression of oral clefting.
A Survey of Cleft Lip and Palate Management Taught in Training Programs in Korea
Authors: Lee Taik Jong , Kim Sang Tae
Published: October 2018

Objective

A national survey was conducted to obtain an overall view of the status, assess changes in trends of cleft management, and study new categories of interest.

Method

Questionnaires were sent to 90 chief residents of all Korean institutes with plastic surgery training programs.

Results

Forty-eight respondents returned completed questionnaires. The results were: (1) the most common unilateral cleft lip repair method used is the rotation-advancement technique (100%); (2) 73% of respondents correct cleft lip nasal deformity at preschool age; (3) the Millard technique is the most common management in bilateral cleft lip repair (65%); (4) in the management of protruding premaxilla, 44% of the respondents choose presurgical orthopedics; (5) the usual cleft palate repairs are the pushback (64%) and double-opposing Z-plasty (43%) in complete cleft palates and the double-opposing Z-plasty (75%) and pushback (36%) comprised the common techniques in incomplete cases; (6) 86% of responding programs perform alveolar cleft repairs during the period of mixed dentition; the most common treatment of velopharyngeal incompetence is pharyngeal flap (71%); and (8) the interdisciplinary team approach is practiced by 64% of respondents.

Conclusions

Many changes were noted since a previous 1995 survey. We hope that this study provides the necessary information needed for the eventual establishment of standard cleft management in Korea.
Case Report: Cleft Palate Closure in 18-Month-Old Female with Epidermolysis Bullosa
Authors: Cooper Jason , Seung-Jun O , Thaller Seth R.
Published: October 2018

Objective

In a patient with epidermolysis bullosa and cleft palate, perioperative guidelines and surgical management can be unclear. This case report illustrates the occurrence of a skin anomaly often associated with potentially significant morbidity and a cleft palate. Perioperative guidelines to successfully manage the cleft palate closure in these patients are presented.
Perceived Social Support of Mothers of Children with Clefts
Authors: Sank Jessica R. , Berk Nancy W. , Cooper Margaret E. , Marazita Mary L.
Published: October 2018

Objective

To identify factors that may predict perceived social support in mothers of children with clefts.

Design

A comparison was conducted from a sample of mothers of children with oral clefts identified from a single institution (multidisciplinary craniofacial clinic). The Interpersonal Support Evaluation List and Beck Depression Inventory were completed by all participants.

Results

Mothers of children with visible clefts were more likely to report higher perceived social support than mothers of children with clefts that were not visible (p = .005). Mothers of female children reported higher levels of perceived social support than mothers of male children (p = .02). Mothers who had completed more years of education reported significantly higher levels of perceived social support than mothers who had completed fewer years of school (p = .03). Mothers with more children were more likely to report lower levels of perceived social support (p = .009). Maternal depression was highly correlated with perceived social support (p < .0001).

Conclusions

Cleft visibility, family size, child sex, and maternal education level were important factors in predicting perceived social support in mothers of children with clefts. Child age, syndrome status, and cleft family history were not significant predictors.
Report of a Patient with Hypoglossia-Hypodactylia Syndrome and a Review of the Literature
Authors: Yasuda Yoshitaka , Kitai Noriyuki , Fujii Yuka , Murakami Shumei , Takada Kenji
Published: October 2018

Objective

To present the morphology of the extremities, craniofacial structures, and the oral cavity based on roentgencephalometry and three-dimensional computed tomography and magnetic resonance imaging scanning in a patient with hypoglossia-hypodactylia syndrome, discuss the orthodontic treatment method, and review the literature for the syndrome.

Patient

The patient was a 6-year-old boy diagnosed with hypoglossia-hypodactylia syndrome at birth. He had hypodactylia as well as micrognathia with steep inclination of the anterior surface of the mandible in relation to the lower mandibular plane. He had missing mandibular incisors with concomitant bone defect limited to the associated alveolar ridge and an absence of any malformations in the mandibular ramus and condylar head. The patient had a bilateral scissors bite with an extremely constricted mandibular dental arch, skeletal Class II jaw relationship with an average mandibular plane angle and maxillary incisors inclined palatally. He had extremely reduced tongue size and hypertrophy of the floor of the mouth. Anomalies of the central nervous system were not observed. There was no evidence of hearing loss.