bluuebunny/crossref_metadata_2025 huggingface dataset:
The Early Prenatal Diagnosis of Cleft Lip and the Decision-Making Process
Authors: Blumenfeld Zeev , Blumenfeld Israel , Bronshtein Moshe
Published: October 2018

In 1996, Bronshtein et al. published an article entitled “Early prenatal diagnosis of cleft lip and its potential impact on the number of babies with cleft lip” in the British Journal of Oral and Maxillofacial Surgery. In this article, Bronshtein et al. revealed that, in their experience, a cleft of the lip could be identified accurately using transvaginal sonography at 13 to 16 weeks' gestation. Of interest was the observation that, in 15 cases where cleft lip was detected, the pregnancy was terminated in all but one case. The only couple that did not choose to terminate subsequently stated that they would terminate a future pregnancy if the fetus was observed prenatally to have a cleft lip.

The notion of pregnancy termination for a relatively minor malformation raises a number of issues. In order to address those issues and to relate experiences in the U.S. to those reported in Israel, the following two papers were invited. In the first, Zeev Blumenfeld and colleagues update their experiences and discuss some practical, moral, and ethical issues. In the second paper, Marilyn Jones presents her perspective on the issue based on her experiences in Southern California.

Readers are invited to react and respond to these two papers by submitting a letter to the editor. Readers are also encouraged to submit other issues or controversies related to the care and/or study of cleft lip and palate and craniofacial anomalies to the editor for discussion.
Nasal Deformity and Microform Cleft Lip in Parents of Patients with Cleft Lip
Authors: Sigler Alicia , Ontiveros Dolores Saavedra
Published: October 2018

Objective

To describe subtle nasal deformities and microform cleft lips in parents whose children have complete cleft lip deformities.

Design

Clinical analyses of three parents whose children had complete cleft lips. Subtle nasal deformities and microform cleft lips were identified.

Setting

An institutional general hospital: Manuel Gea González Cleft Lip and Palate Clinic in Mexico City, Mexico.

Patients

The study first examined the parents of all complete and incomplete cleft lip-palate patients who were seen from March 1994 to February 1997 by the authors (n = 1000). We identified three patients, each of whom had one parent who showed signs of subtle nasal deformity and microform cleft lip.

Interventions

None for the parents. Interventions in the children with cleft lips and palates were known.

Main Outcome Measures

Qualitative photographic analyses were performed. Nostril symmetry, septal deviation, nasal floor position, and orbicularis oris malinsertions were carefully examined.

Results

Three of the evaluated parents had one alar cartilage caudally displaced and a deviated septum. One parent's nasal floor was depressed. Two parents also had evidence of a minimal orbicularis oris muscle fissure located in the upper lip.

Conclusion

Genetic evaluations of children with complete and incomplete cleft lips might also include thorough evaluation of their parents. Although small in size, this study of three case histories identified nasal and lip deformities in the patients' parents.
Secondary Alveolar Bone Grafting Using Milled Cranial Bone Graft: A Retrospective Study of a Consecutive Series of 100 Patients
Authors: Denny Arlen D. , Talisman Ran , Bonawitz Steven C.
Published: October 2018

Objective

The purpose of this study was to evaluate the outcome of secondary bone grafting of alveolar clefts using milled cranial bone graft.

Patients

The study included a consecutive series of 100 patients who were operated on between 1986 and 1995 by a single senior surgeon.

Measurements

The patients were divided into four groups; (1) unilateral alveolar cleft or (2) bilateral alveolar cleft, (3) before eruption of the canine teeth or (4) after eruption. Follow-up ranged from a minimum of 12 months to 10 years, and evaluation included a physical examination, medical photography, orthodontic reports, and a panorex X-ray and/or a three-dimensional computed tomography scan of the maxilla produced using Denta-scan software. Patient outcomes were judged to be good, acceptable, or poor. Patients who developed any fistula or required regrafting were defined as poor outcomes.

Results

The combined good and acceptable outcomes represented 83% of the entire consecutive series. These two groups were defined as successful outcomes. In patients with unilateral and bilateral clefts who were under 12 years old, the success rate was 90% and 88%, respectively. In patients grafted following full canine eruption (>12 years old), success rate decreased to 83% in patients with unilateral and 66% in bilateral clefts, respectively.

Conclusions

Our results support the use of milled cranial bone graft, which produces a stable closure of the alveolar cleft with good contour and support for adjacent tooth eruption. Our data further support the conclusion by others that outcomes of early secondary grafting are superior to delayed grafting. The experience presented here, including the success rate, ease of harvesting, and minimal morbidity, makes the cranium our preferred donor site for alveolar cleft grafting.
Adaptation of Pharyngeal Wall Adduction after Pharyngeal Flap Surgery
Authors: Karling Jonas , Henningsson Gunilla , Larson Ola , Isberg Annika
Published: October 2018

Objective

The purpose of this investigation was to study lateral pharyngeal wall adduction relative to pharyngeal flaps of different widths. The hypothesis to be tested was that pharyngeal wall adduction does not increase postoperatively but may decrease due to the mechanical hindrance of a wide flap.

Design

In this prospective study, adaptation of lateral pharyngeal wall adduction during speech was studied relative to pharyngeal flaps of different widths utilizing videoradiography. Flap width was determined nasopharyngoscopically.

Setting

All patients were treated by the Stockholm Cleft Palate Team, Sweden.

Patients

Fifty-three patients were strictly selected by discarding conditions known to exert uncontrolled influence on velopharyngeal sphincter function.

Results

The results revealed a potential for adaptation of pharyngeal wall adduction to different flap widths. The magnitude and character (increase or decrease) of change in adduction was significantly correlated with the degree of preoperative adduction and with the width of the flap. In patients with limited preoperative adduction, pharyngeal wall activity increased, more in the presence of a narrow flap while less if the flap was wide. When preoperative adduction was pronounced, the postoperative activity decreased because of mechanical hindrance by the flap, and the degree of impediment was correlated to the width of the flap.

Conclusion

A potential for increased lateral pharyngeal wall adduction after pharyngeal flap surgery was verified, but the result cannot be interpreted as generally applicable because of the strict selection of patients.
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.
Bone Substitutes
Authors: Burstein Fernando D.
Published: October 2018

The search for the ideal bone substitute began hundreds of years ago, and continues today. While numerous choices have been proposed and tested, with varying degrees of success, there remain many challenges related to the use of bone substitutes in craniofacial reconstruction. This paper presents a review of the history of bone substitute research, a discussion of currently popular materials, and elucidation of the challenges to be faced as we approach the new millennium.
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.
Euclidean Distance Matrix Analysis of Surgical Changes in Prepubertal Craniofacial Microsomia Patients Treated with an Inverted L Osteotomy
Authors: Hay A. D. , Ayoub A. F. , Moos K. F. , Singh G. D.
Published: October 2018

Objectives

Correction of craniofacial microsomia (CFM) presents several challenges concerning the modality of surgical intervention. The aim of this study was to assess early and late surgical outcome, by undertaking Euclidean distance matrix analysis (EDMA) of CFM patients exhibiting an unilateral mandibular deformity that was surgically corrected by an inverted L osteotomy and autogenous bone graft.

Design

Longitudinal study. Preoperative, ≈1-year postoperative and ≈3-year postoperative assessments of 14 consecutive children (mean age 9 years) with CFM. Posteroanterior cephalographs were scanned and five homologous mandibular landmarks were digitized in triplicate (< 1% digitization error). Average mandibular geometries, scaled to an equivalent size, were generated using a generalized rotational fit program (Procrustes superimposition) and subjected to EDMA.

Results

The mean pre- and both postoperative mandibular configurations differed statistically (p < .01). Early postoperative improvements in mandibular form were noted; increases in length arising in the treated mandibular body (≈ 19%) and ramus (≈ 13%). Comparing early and late postoperative configurations, a decrease of ≈ 22% in the late postoperative mandibular body length was evident, but the ramus maintained steady vertical growth (≈ 7%). Comparing the preoperative and late postoperative configurations, the decrease observed in the mandibular body on the treated side was reduced to ≈ 8% while the ramus maintained good growth (≈ 20%) on that side.

Conclusion

Mandibular morphology is improved significantly in CFM patients surgically treated by an inverted L osteotomy, but relapse in the mandibular body is evident after ≈ 3 years. Nevertheless, ramus growth proceeds well after the surgical reconstruction.
Orthodontic Treatment of the Patient with Complete Clefts of Lip, Alveolus, and Palate: Lessons of the past 60 Years
Authors: Long Ross E. , Semb Gunvor , Shaw William C.
Published: October 2018

This review paper summarizes 60 years of progress in the orthodontic care of patients with complete clefts of the lip, alveolus, and palate. The progress and evolution of orthodontic treatment for patients with complete clefts has taken a slow and sometimes circuitous route to present-day standards. Nonetheless, in spite of this history of slow and inefficient scientific and clinical progress, review of the literature of the past 60 years does, in fact, reveal that progress has been made. This progress has not only been in the area of improved surgical and orthodontic techniques but also relative to the scientific weaknesses of past decades. The investigations of more recent years seem to be moving in a direction of better documentation, stricter methodologies, longer-term follow-up, larger sample sizes, etc. However, there remains much work to be done. Only by adopting a more critical approach will protocols be defined that can achieve optimal outcomes, while minimizing the burden of the orthodontic treatment through elimination of superfluous intervention.
The Effect of Vowels on Nasalance Scores
Authors: Lewis Kerry E. , Watterson Thomas , Quint Teresa
Published: October 2018

Objective

Nasalance scores were compared for nine different speech stimuli with vowel content controlled.

Design

The nine speech stimuli included four vowels spoken in isolation and five sentences. The four vowels were /i/, /u/, /æ/, and /a/. Four of the five sentences were loaded with High Front, High Back, Low Front, or Low Back vowels, and the fifth sentence contained a mixture of vowel types.

Setting

Academic and clinical craniofacial center.

Subjects

The subjects were 19 children with velopharyngeal dysfunction (VPD) and 19 children without history of communication disorder.

Main outcome Measures

The main outcome measures were the nasalance scores associated with the nine different speech stimuli for two groups of subjects.

Results

For the VPD group, analysis of variance procedures revealed that nasalance scores for high-vowel sentences and the mixed-vowel sentence were significantly higher than the nasalance scores for the two low-vowel sentences. This pattern was the same for the non-VPD group except for the High Back/Low Back contrast, which was not significant. In both groups, nasalance scores for sustained vowels were significantly higher for the High Front vowel /i/ than for any other vowel, and nasalance was significantly higher for the High Back vowel /u/ than for either of the Low vowels /æ/ or /a/. There was no significant difference between Low vowels.

Conclusion

Nasalance scores may be affected by the vowel content of the speech stimulus. This should be taken into consideration on a clinical basis and for research purposes.
What Information Do Parents of Newborns with Cleft Lip, Palate, or Both Want to Know?
Authors: Young Jennifer L. , O'Riordan Maryann , Goldstein Jeffrey A. , Robin Nathaniel H.
Published: October 2018

Background

The unexpected birth of a baby with a cleft lip and palate (CL/P) is a shocking and traumatic experience, generating anxiety for parents as well as the attendant health care team. Parents frequently leave the hospital with many unanswered questions because health care professionals do not educate them adequately.

Objective

To determine what information these parents felt was “critical” for them during the immediate newborn period and to determine how the “informer” was perceived during these encounters.

Design

Retrospective, self-administered questionnaire.

Subjects and Methods

Biologic parents of children with isolated CL/P aged 6 years and younger were surveyed. The questionnaire asked parents whether they remembered discussing diagnosis, prognosis, management, home care, and psychosocial issues. Parents were also asked to rank how “critical” it would have been for the “informer” to have discussed certain issues with them during this first day.

Results

Parents gave the highest priority to feeding and learning to identify illness in their baby; 95% wanted to be shown all normal aspects of their baby's exam, and 87% wanted to be told that the CL/P was not their fault. Usage of proper terminology to describe abnormal findings and receiving assurance that their child was not in pain were also important. Unfortunately, many parents reported that the informers did not address these issues.

Conclusions

Parents of newborns with CL/P want basic information in the immediate newborn period, especially regarding feeding and recognizing illness. These data suggest that informers are not adequately discussing these issues with parents.
Oral Clefts and Vitamin Supplementation
Authors: Loffredo L. C. M. , Souza J. M. P. , Freitas J. A. S. , Mossey P. A.
Published: October 2018

Objectives

The evidence linking low levels of folic acid and orofacial clefting (OFC) is presently equivocal. There is stronger evidence for the role of folic acid supplementation in protection against the occurrence and recurrence of neural tube defects. The present investigation tested the hypotheses that cleft lip, cleft palate, or both are inversely associated with maternal intake of dietary and supplemental vitamins during the periconceptional period and first 4 months of pregnancy in a Brazilian population.

Design

A population-based, case-control study of cleft lip with or without cleft palate (CL(P)) and isolated cleft palate (CP) in a Brazilian population. In structured interviews, case histories were taken from the mothers of a consecutive sample of 450 infants born with nonsyndromic OFC.

Results

Mothers who had children with CL(P) were less likely to have been supplemented during the periconceptional period. The statistical significance of the difference in prevalence of the use of supplements between mothers of patients and of controls was greater for the CL(P) group: p < .05 for CP and p < .001 for CL(P). Multivariate analysis confirmed this finding of a protective effect for both types of orofacial cleft.

Conclusions

The use of vitamin supplements in the first 4 months of pregnancy was suggestive of a protective effect against the occurrence of CP and CL(P) in this population. The significance of an association between multivitamin supplementation and OFC and the possible role of gene/environment interaction are discussed.
Nasalance Scores in Noncleft Individuals: Why Not Zero?
Authors: Gildersleeve-Neumann Christina E. , Dalston Rodger M.
Published: October 2018

Objective

To determine whether oral or nasal acoustic energy is primarily responsible for nonzero nasalance scores observed during the production of nonnasal sentences by individuals with normal speech.

Method

Sixty adults with normal speech were asked to read the Zoo passage and produce three sustained vowels, (/i/, /a/ and /u/), with and without nares occlusion.

Results

There was a significant decrease in nasalance scores between the unoccluded and occluded conditions for all four stimulus pairs. The mean decrease across conditions ranged from 8 (/u/) to 25 (/i/). In the unoccluded condition, the nasalance score was significantly greater for /i/ than for the other stimuli.

Conclusions

The findings suggest that the majority of acoustic energy detected by the nasometer's nasal microphone during the production of nonnasal utterances is the result of sound transmission through the nose. The data obtained during this investigation, coupled with information available from other studies, suggest that this may be due to transpalatal transmission. If correct, such a conclusion would have clinical implications for patients with palatal clefts, since residual structural abnormalities and scar tissue in a repaired cleft palate may increase, dampen, or in some way alter transpalatal acoustic transmission. Thus, surgical normalization of velopharyngeal port control may not be sufficient to eliminate hypernasality in all patients.
Characteristic Forms of the Upper Part of the Oral Cavity in Newborns with Isolated Cleft Palate
Authors: Ihan-Hren Nataša , Oblak Peter , Koželj Vesna
Published: October 2018

Objective

To determine the functional and morphological compensation of the lack of integrity in the upper part of the oral cavity in newborns with isolated cleft palate. Integrity of the upper part of the oral cavity is required for the effective pumping of amniotic fluid, the essential mode of nutrition in intrauterine life. The adaptation could be seen immediately after birth.

Materials

Plaster casts of the upper part of the oral cavity in 60 newborns with isolated cleft palate of various extent and plaster casts of the upper part of the oral cavity in 27 newborns without cleft were used. A number of surface points were identified and used for a trigonometric morphological analysis.

Results

The parameters of the upper part of the oral cavity in four groups of newborns with various extent of isolated cleft palate were compared with newborns without cleft palate. The results indicate a shift of the functional oral cavity into the nasal cavity and the pharynx, depending on the extent of the cleft.

Conclusion

The pumping activity of the tongue forms the upper part of the oral cavity and consequently the tongue moves into the nasal cavity and pharynx, depending on the extent of the cleft. In this way, effective pumping of amniotic fluid is possible despite the cleft. This is of vital importance for the fetus during intrauterine life. A poorly passable or even unpassable respiratory way is only of secondary importance during that time.
Changes in Speech following Unilateral Mandibular Distraction Osteogenesis in Patients with Hemifacial Microsomia
Authors: Guyette Thomas W. , Polley John W. , Figueroa Alvaro , Botts Jennifer , Smith Bonnie E.
Published: October 2018

Objective

The purpose of this study was to describe changes in articulation, resonance, and velopharyngeal function following mandibular distraction osteogenesis.

Design

This is a descriptive, post hoc study comparing the performance of patients on measures of articulation, resonance, and velopharyngeal function before and after mandibular distraction.

Setting

The data were collected at a tertiary health care center located in Chicago.

Patients

The clinical data from preoperative and postoperative evaluations of seven mandibular distraction patients were used.

Outcome Measures

The outcome measures were number of articulation errors, severity of hypernasality and audible nasal emission, and velopharyngeal orifice size as estimated using the pressure-flow technique.

Results

Immediately after distraction, 28% (2/7) experienced a temporary deterioration in articulation and 42% (3/7) experienced a deterioration in nasal resonance. But by the long-term follow-up evaluation, all had returned to their preoperative levels. Pressure-flow test results generally support the perceptual findings.

Conclusions

Patients being considered for mandibular distraction surgery should receive preoperative and postoperative speech evaluations and be counseled about risks for changes in their speech following surgery.
“Only Skin Deep”: Health, Resilience, and Craniofacial Care
Authors: Strauss Ronald P.
Published: October 2018

The literature on psychological and social responses to cleft palate and craniofacial conditions focuses on the challenges and limitations posed by biological factors, cognitive status, and social stigma. In spite of the various challenges experienced by persons with these conditions, many individuals have rich and satisfying lives. This paper develops a theoretical perspective to elucidate factors that account for health, life success, and resiliency in persons with craniofacial conditions. Three steps are proposed to change how craniofacial conditions are envisioned by clinicians and researchers: (1) creating optimism and positivity around the time of birth and diagnosis, (2) asking children and parents new and different questions, and (3) launching a new craniofacial social science model for research built around understanding resilience and health.
Resiliency and Success in Adults with Moebius Syndrome
Authors: Meyerson Marion D.
Published: October 2018

Objective:

This study asked selected mature individuals with Moebius syndrome to discuss the sources of strength and resiliency that allowed them to achieve professional and personal success.

Design:

Adults with Moebius syndrome were selected in a nonrandom manner based primarily on their affiliation with the Moebius Syndrome Foundation. Following a letter from the author describing the objective, the subjects were interviewed by telephone and were encouraged to respond at length.

Participants:

Eighteen adults (aged 29 to 70 years) responded. All were or had been gainfully employed in a variety of professional or vocational positions.

Results:

The respondents reported the following as major sources of resiliency and success: family support, faith, humor, sense of self, special skills, determination, and networking.

Conclusions:

The recognition and reinforcement of strengths and resiliences in younger patients may help maximize their professional and personal success as adults.
Deficits versus Strengths: Ethics and Implications for Clinical Practice and Research
Authors: Mouradian Wendy E.
Published: October 2018

A shift in emphasis from deficits to strengths to promote health and well-being in patients with congenital and acquired craniofacial conditions (CFC) is appropriate given the chronic, “incurable” nature of CFC. Personal narratives are a valuable starting point for discovering sources of resilience. This paper explores such a shift by considering two areas of ethical concern suggested by patient narratives: informed consent and the physician-patient relationship. Both areas contain pitfalls and opportunities.

The powerful emotions these patients bring with them argue for caution in medical decision making. Attention to the individual's psychosocial adjustment should always supplement surgical evaluations. Because of the inequality of the physician-patient relationship, care should be taken to use this power in positive ways. The relationship between patient and surgeon is particularly charged and may be an important source of support, information, hope, and advice for patients. The changing health care system threatens the physician-patient relationship, but the rise of alternative medicine suggests patients continue to value relationships. Relationships are critical for individuals with CFC, who experience social rejection because of the fundamental importance of face in human interactions.

Future research directions should include long-term outcome studies on patients receiving modern craniofacial team care, qualitative research on resilience in patients with positive life adjustment, and development of a conceptual framework and research methodology for understanding quality of life of individuals with CFC. An emphasis upon strengths rather than defects will have implications for the structure of craniofacial teams, the care that is provided, and allocation of resources.