bluuebunny/crossref_metadata_2025_split huggingface dataset:
Neonatal Feeding of Infants Born with Cleft Lip and/or Palate: Parental Perceptions of their Experience in South Wales
Authors: Oliver Richard G. , Jones Gail 
Published: October 2018

Objective: This paper describes the responses of 100 parents of children born with cleft lip and/or palate in South East Wales to a postal questionnaire survey of their experiences of the services available to help with the feeding of their children in the immediate postnatal period. Results and Conclusions: Prior to birth, the majority of mothers intended to breast feed their child; after birth, the majority of infants were bottle fed. The majority of mothers felt that they had been a pan of the decision to change methods. Advice on feeding was obtained from a variety of sources, both in hospital and on discharge. Mothers of cleft children who were first born in the family were less critical of the support offered in hospital than were mothers whose cleft child was a second or subsequent child. Isolated clefts of the palate were associated with a high proportion of feeding via nasogastric tube both on discharge from hospital and at home. The major criticism of the service offered to parents and their children was the poor quality of advice on feeding their children, both in hospital and on discharge. A variety of mechanisms for service improvement are suggested.
Management of Congenital Buccopharyngeal Membrane
Authors: Bent John P. , Klippert Frederick N. , Smith Richard J. H. 
Published: October 2018

Objective: To describe our experience in treating congenital buccopharyngeal membranes and reflect on lessons learned managing this rare anomaly. Design: Case series of two patients followed 3 and 6 years, respectively. Setting: Two different academic tertiary-care medical centers. Patients: Two patients with congenital buccopharyngeal membranes. Intervention: Multiple pharyngoplasties, including the use of lasers, rotated mucosal flaps, and oropharyngeal stents. Results: The first patient has had her tracheotomy removed, but she still uses a gastrostomy tube and has an expressive speech delay. The second patient can eat well but has persistent speech and airway problems, largely due to his micrognathia. Conclusions: Centrally dehiscent buccopharyngeal membranes are rare and require carefully planned treatment. Associated anomalies worsen the prognosis. We propose a set of recommendations to assist in the management of this difficult problem.
Resonance Imaging of the Eustachian Tube Cartilage in Microtia
Authors: Imai Yoshinor , Matsuo Kiyoshi , Imai Naomi 
Published: October 2018

Objective The purpose of this study was to determine whether external and internal defects in microtia are related. Method Magnetic resonance images of the eustachian tube cartilage were evaluated for 20 patients who had unilateral microtia. Nineteen patients were classified as Grade 2, and one was classified as Grade 3. The Grade 3 patient also had unilateral facial palsy. Results On T1-, T2-, and proton-density-weighted images, the eustachian tube cartilage was clearly identified as a pair of straight lines with low signal intensity. There was no evidence of hypoplasia of the eustachian tube cartilage on the microtic side in any Grade 2 patient, but hypoplasia was evident on the microtic side of the patient classified as Grade 3. Conclusion These findings are consistent with the view that impairment of embryonic development before 6 weeks results in injury to the immature pri-mordium and malformation of both the external and middle ear. In contrast, injuries that occur at a later fetal age (i.e., after 3 months) do not appear to cause middle ear malformations.
Objective, Noninvasive Evaluation of Velopharyngeal Function in Cleft and Noncleft Patients
Authors: Kunkel Martin , Wahlmann Ulrich , Wagner Wilfried 
Published: October 2018

Objective The purpose of this study was to investigate a new diagnostic method that provides an approach to noninvasive, objective measurement of velopharyngeal movement by acoustic determination of epipharyngeal volume changes with velopharyngeal muscle function. Design This was a case control study, using consecutive samples. Setting This study took place at the Cleft Palate Rehabilitation Center of the University of Mainz, Germany. Patients Subjects were 29 consecutive cleft lip and palate (CLP) patients and 31 controls (21 patients with dysgnathia and 10 healthy volunteers). Intervention A series of transnasal acoustic measurements (pressure wave: 55 dB for 2 milliseconds) of epipharyngeal volume were performed with the pharyngeal muscles relaxed in end-expiration and while the velopharyngeal orifice was closed, with the difference in volume representing maximal pharyngeal movement. Results Cleft palate patients yielded significantly lower values of velopharyngeal movement (6.5 cm3) than did the control group (8.0 cm3)(p < .05; Mann-Whitney U test). Overlapping ranges of values were measured for the C(L)P and control groups. The least mobility (4.75 cm3) was measured in patients who had undergone pharyngeal flap surgery. Different patterns of restriction were observed in patients with and without a pharyngeal flap. Conclusion Acoustic pharyngometry may provide access to noninvasive quantitative measurement of velopharyngeal movement and a better understanding of the pattern of movement in C(L)P-patients. We expect it to be a helpful tool in objectively monitoring the progress of logopedic therapy.
Cleft Lip with or without Cleft Palate: Effect of Family History on Reproductive Planning, Surgical Timing, and Parental Stress
Authors: Andrews-Casal Melanie , Johnston Dennis , Fletcher Jack , Mulliken John B. , Stal Samuel , Hecht Jacqueline T. 
Published: October 2018

Objective This study tested whether the presence of a family history of non-syndromic cleft lip with or without cleft palate (NSCLP) lessens the negative impact on reproductive planning, decreases treatment anxiety, and relieves parental stress when there is a recurrent cleft. Design Sixty-one participating families, retrospectively ascertained through their children with NSCLP, were divided into two groups: those with a family history of clefting and those in which only the proband was affected. Setting Each family completed a questionnaire either at their home or during a clinic visit. Participants Questionnaires were distributed to 117 families with an NSCLP child. Of these 117, 49 (42%) had another family member with NSCLP and 68 (58%) had no prior history of clefting. Sixty-one (52%) families returned the questionnaire and were categorized by family history. Main outcome Measures The Parenting Stess Index and the Hollingshead Index of Social Position were included with family and medical history questions in the 137-item questionnaire. Results The reproductive plans of the two groups were quite similar and were not dependent on the families' actual or perceived recurrence risks of NSCLP. The majority (85%) of the children with NSCLP underwent surgical closure at the recommended age. The two groups did not significantly differ on any score on the Parenting Stress Index, indicating a similar parental stress level between the groups. Conclusions These findings suggest that family history does not significantly influence reproductive plans, timing of cleft repair, or stress of parents of a child with NSCLP. Further, these results indicate a need for additional psychosocial study of this population.
Atrial Septal Defect in Cyanotic CL/Fr Mice
Authors: Kadowaki Shingo , Sakamoto Michiko , Kamiishi Hiroshi , Tanimura Takashi 
Published: October 2018

Objective It is widely known that some newborn CL/Fr mice with cleft lip and palate (CLP) also have cyanotic symptoms, which have been thought to depend on an atrial septal defect (ASD). In a previous study, we found that cyanotic mice tended to have more severe types of CLP. We hypothesize that the mechanical airway obstruction due to a poorly developed palatal shelf and unmoved tongue in CLP(+) mice might be related to the occurrence of cyanosis. The purpose of this study was to examine the relationships between ASD and cyanosis in CLP(+) newborns. Method The newborn hearts from CLP(-), noncyanotic CLP(+), cyanotic CLP(+), CL/Fr mice and ICR mice were examined histologically, and the incidence and size of ASD was determined on neonatal day (ND) 0. In CLP(-) newborns, similar procedures were performed from ND 1 to ND 4. Furthermore, in CLP(+) newborns, development of the palatal shelf was examined. Results While all the ICR mice had a well-developed atrial septum, and the incidence of ASD was 0%, about 80% of CL/Fr mice had ASD, irrespective of the presence or absence of CLP and cyanosis. On ND 0, the septum primum was significantly shorter in cyanotic CLP(+) mice than in CLP(-) mice. It also tended to be shorter in CLP(+) mice than in CLP(-) mice. Between the cyanotics and noncyanotics, there were no significant differences in the incidences of ASD and the rate of septal development. In CLP(-) mice, the septum primum developed well later and no ASD was observed on ND 4. Cyanotic newborns had significantly less developed palatal shelves than did noncyanotics. Conclusions Cyanosis may not be related to ASD and the rate of septal development, but may be related to the occurrence of CLP in this strain. Furthermore, we confirmed that some relationship exists between the development of the palatal shelf and cyanosis. The present study supports our hypothesis concerning the cause of cyanosis in CL/Fr mice.
Cleft Palate Formation in Fetal Br Mice with Midfacial Retrusion: Tenascin, Fibronectin, Laminin, and Type IV Collagen Immunolocalization
Authors: Singh G.D. , Johnston J. , Ma W. , Lozanoff S. 
Published: October 2018

Objective This study tested the hypothesis that altered craniofacial morphology does not affect the expression of extracellular matrix (ECM) molecules such as fibronectin (FN), laminin (LN), type IV collagen, and tenascin-C (TN) but is associated with failure of palatal shelf elevation and fusion concomitant with cleft palate formation. Design To test this hypothesis, a comparative immunohistological analysis of FN, LN, type IV collagen, and TN was undertaken on brachyrrhine (Br/Br) mice and normal (+/+) fetuses during secondary palate formation. Normal and Br/Br fetuses were collected at gestational days E13 and E14 (representing prefusion stages) and E15 and E18 (representing postfusion stages). Cryostat palatal sections (8 μm) were postfixed in methanol, washed, and stained with primary antibody. All sections were washed and coated with secondary antibody (swine-anti-rabbit IgG) and mounted with citifluor. Results Immunohistological analysis showed that LN and type IV collagen were located near the presumptive medial epithelial seam (MES) or edge (MEE) in +/+ or Br/Br fetuses, respectively. Fibronectin showed a homogeneous distribution at all stages in both groups of mice. In contrast, TN became localized below the presumptive MES or MEE in both groups of mice at E14. In +/+ animals at E15, TN dissipated and became confined to the oral basement membrane by E18. At E15 and E18 in cleft Br/Br mutants, TN stained beneath the MEE. Conclusion Although the distributions of ECM molecules are similar during normal and cleft palatogenesis, differences in TN expression are associated with cleft palate formation.
Reduced Need for Alveolar Bone Grafting by Presurgical Orthopedics and Primary Gingivoperiosteoplasty
Authors: Santiago Pedro E. , Grayson Barry H. , Cutting Court B. , Gianoutsos Mark P. , Brecht Lawrence E. , Kwon Soon Man 
Published: October 2018

Objective The purpose of this study was to evaluate if narrowing and approximation of the alveolar cleft through presurgical alveolar molding followed by gingivoperiosteoplasty (GPP) at the time of lip repair reduces the need for a bone-grafting procedure. Design This was a retrospective blind study of patients with unilateral or bilateral alveolar clefts who underwent presurgical infant alveolar molding and GPP by a single surgeon. Alveolar bone formation was assessed prior to the eruption of the maxillary lateral incisor or canine by clinical examination, panoramic and periapical radiographs, and/or a dental CT scan. The criterion for bone grafting was inadequate bone stock to permit the eruption and maintenance of the permanent dentition. Setting This study was performed at the Institute of Reconstructive Plastic Surgery by the members of the Cleft Palate Team. Patients All patients with unilateral (n = 16) or bilateral (n = 2) alveolar clefts who underwent presurgical infant alveolar molding and GPP by a single surgeon from 1985 to 1988 were studied. The control population consisted of all alveolar cleft patients (n = 14) who did not undergo alveolar modeling or GPP during the same time period. Interventions Presurgical alveolar modeling was performed with an intraoral acrylic molding plate. This plate was modified on a weekly basis to align the alveolar segments and close the alveolar gap. The surgical intervention consisted of a modified Millard GPP. Main outcome Measures The primary study outcome measure was the elimination of the need for a secondary bone graft in patients who underwent presurgical alveolar molding and GPP. Results Of the 20 sites in the 18 patients who underwent GPP, 12 sites did not require an alveolar bone graft. Of the 8 sites requiring a bone graft, 4 presented minimal bony defects. All 14 patients in the control group required bone grafts. Conclusions In this series of 20 alveolar cleft sites treated with presurgical orthopedics and GPP, 60% did not need a secondary alveolar bone graft in the mixed dentition.
Articulation and Nasality Changes Resulting from Sustained Palatal Fistula Obturation
Authors: Pinborough-Zimmerman Judith , Canady Claudia , Yamashiro Duane K. , Morales Louis 
Published: October 2018

Objective The purpose of the study was to determine differences in articulation and nasality with obturation over time in children with a palatal fistula. Design Articulation and nasality were measured with the fistula open, immediately after obturation, and 4 to 7 weeks postobturation. Setting, Patients, Participants Subjects were 15 patients with a palatal fistula secondary to a repaired cleft palate who were seen through the Orofacial Program, Utah Department of Health, ranging in age from 4 years 6 months to 13 years 1 month. Interventions Acrylic palatal obturators were designed to provide coverage specific to the unique shape and location of each child's fistula. Obturators were cemented to molar teeth using wire clasps for control of usage. Main outcome Measures Measurements consisted of listener judgments of hypernasality, hyponasality, and nasal emissions; instrumental ratings of nasalance using the Nasometer 6200-2; and performance on a standardized articulation test. Results Significant improvement occurred only on nasal emission measures from the preobturation condition to immediate postobturation. However, significant improvement was found in articulation, listener judgments of hypernasality, nasal emissions, and Nasometric Nasal Sentence mean scores from the preobturation condition to 4 to 7 weeks postobturation and from the immediate postobturation condition to 4 to 7 weeks postobturation. No significant differences were found between conditions for listener judgments of hyponasality and Nasometric Zoo and Rainbow Passage scores. Obturation of the palatal fistula over a 4- to 7-week period resulted in no adverse effect on articulation ability, perceptual ratings of nasality, or instrumental ratings of nasalance. Conclusions Clinical management of patients with a palatal fistula can be enhanced with treatment using obturation over time. For subjects who continue to exhibit hypernasality immediately postobturation, sustained obturation is advocated prior to consideration of surgical intervention for treatment of a palatal fistula and/or velopharyngeal dysfunction.
Craniofacial Morphology in Adult Patients with Unoperated Complete Bilateral Cleft Lip and Palate
Authors: Da Silva Filho Omar Gabriel , De Cássia Moura Carvalho Lauris Rita , Filho Leopoldino Capelozza , Semb Gunvor 
Published: October 2018

Objective This report is a retrospective study that compares the craniofacial morphology of adult subjects with unoperated bilateral complete cleft lip and palate (BCLP) with that of a noncleft group. Methods The study was performed on standardized lateral cephalograms obtained at the Hospital for Research and Rehabilitation of Cleft Lip and Palate, University of São Paulo, Brazil. The research group consisted of 28 subjects (20 males, 8 females) with unoperated BCLP, ranging in age from 15 to 41 years. The control group was matched to the cleft group with regard to gender and age. The findings were analyzed on the basis of the two-way analysis of variance (ANOVA) for cleft and gender. Results The most striking difference between the groups was the extremely prominent premaxilla in the cleft group that gave the BCLP face a very convex profile. The mandible exhibited a vertical growth pattern that resulted in a steep mandibular plane, an obtuse gonial angle and a long lower face height. The posterior face height was reduced. The cranial base dimensions were smaller, but there was no difference in cranial base angulation. Conclusions These findings confirm that in subjects with unoperated BCLP, the initial characteristics of the cleft malformation persist during growth.
Sensitivity of a Method for the Analysis of Facial Mobility. I. Vector of Displacement
Authors: Trotman Carroll-Ann , Faraway Julian J. , Silvester Kirsten T. , Greenlee Geoffrey M. , Johnston Lysle E. 
Published: October 2018

Objective (1) To determine which facial landmarks show the greatest movement during specific facial animations and (2) to determine the sensitivity of our instrument in using these landmarks to detect putatively abnormal facial movements. Design Movements of an array of skin-based landmarks on five healthy human subjects (2 men and 3 women; mean age, 27.6 years; range, 26 to 29 years) were observed during the execution of specific facial animations. To investigate the instrument sensitivity, we analyzed facial movements during maximal smile animations in six patients with different types of functional problems. In parallel, a panel was asked to view video recordings of the patients and to rate the degree of motor impairment. Comparisons were made between the panel scores and those of the measurement instrument. Results Specific regions of the face display movement that is representative of specific animations. During the smile animation, landmarks on the mid- and lower facial regions demonstrated the greatest movement. A similar pattern of movement was seen during the cheek puff animation, except that the infraorbital and chin regions demonstrated minimal movement. For the grimace and eye closure animations, the upper, mid-facial, and upper-lip regions exhibited the greatest movement. During eye opening, the upper and mid-facial regions, excluding the upper lip and cheek, moved the most, and during lip purse, markers on the mid- and lower face demonstrated the most movement. We used the smile-sensitive landmarks to evaluate individuals with functional impairment and found good agreement between instrument rankings based on the data from these landmarks and the panel rankings. Conclusion The present method of three-dimensional tracking has the potential to detect and characterize a range of clinically significant functional deficits.
Sensitivity of a Method for the Analysis of Facial Mobility. II. Interlandmark Separation
Authors: Trotman Carroll-Ann , Faraway Julian J. 
Published: October 2018

Objective This study demonstrates a method of quantifying facial movements based on distortions of the skin surface. Design Landmarks were identified on the faces of five healthy human subjects (2 men and 3 women; mean age, 27.6 years; range, 26 to 29 years), and the distortions were characterized by changes in the separation between 20 pairs of landmark distances during specific maximal facial animations: smile, lip purse, cheek puff, grimace, eye closure, and eye opening. Data were recorded with a video-based tracking system for a period of 3 seconds at a sampling rate of 60 Hz or frames per second. For each subject, we analyzed the change in the separation of 20 pairs of landmarks, of which the majority were bilaterally symmetrical and functionally active. Results Characteristic patterns of movement emerged for each animation. We found that smiling involved movements of the lateral orbital, circumoral, and chin regions; grimacing involved the inner orbital, lateral orbital, lateral nasal, and upper-lip regions; eye closure involved the inner orbital, lateral orbital, and, to a lesser degree, lateral nasal regions; eye opening involved the inner and lateral orbital regions; cheek puffing involved the cheek and lower-lip regions; and the lip purse animation involved the nasolabial, cheek, commissure, and lip regions. Conclusion This measurement of distortion provided a quantitative estimate of facial movement, and this approach is especially applicable to patients with unilateral problems in which the patient can serve as his or her own control.
Multiple Suture Synostosis and Increased Intracranial Pressure following Repair of Single Suture, Nonsyndromal Craniosynostosis
Authors: Hudgins Roger J. , Cohen Steven R. , Burstein Fernando D. , Boydston William R. 
Published: October 2018

Objective Increased intracranial pressure, frequently associated with closure of multiple cranial sutures, has been reported to occur in 36% of cases following correction of syndromal craniosynostosis. Although much less common, multiple suture closure may occur following repair of single suture, non-syndromal craniosynostosis and we present cases that concern two such children. Results Two children with nonsyndromal craniosynostosis, one metopic and one left-coronal, underwent fronto-orbital advancement at age 3 months. At age 19 months and at age 5 years, respectively, both patients re-presented with headaches, decrease in head circumference percentile, and acceptable cosmetic outcome. Both had computerized tomographic evidence of multiple closed cranial sutures and increased intracranial pressure (ICP) (determined by monitoring). Both patients improved following a cranial expansion procedure. Conclusion Delayed closure of multiple sutures and resultant increased ICP may occur following correction of nonsyndromal, single suture craniosynos-tosis. This may be more likely when the initial suture is contiguous with the facial sutures. Children should be followed for many years following cranio-synostosis repair with cranial, neurologic, and possibly funduscopic examinations as well as head circumference measurements to detect delayed closure of cranial sutures.