Facial transplantation: history and update.

BACKGROUND: Composite tissue allografting (CTA) represents the essence of reconstructive surgery, combining principles of solid organ transplantation (SOT) and modern plastic surgery techniques. The purpose of this article is to give a review of the history of facial CTA and an update of the cases that have been operated so far worldwide. METHODS: A systematic review of the medical literature was performed. Ten relevant publications were selected and analyzed for clinical data of the patients, surgical aspects of transplantation, complications and outcome. RESULTS: The past 9 years, 31 face transplants have been performed worldwide. The main indication was posttraumatic deformity. In all cases standard triple drug immunosuppression as used in SOT was successfully used and at least 1 period of acute rejection was seen in all patients, controllable with conventional immunosuppressive regimens. Overall functional outcomes are good and satisfaction rate is high, surpassing initial expectations. The main complications are opportunistic infections; 4 deaths occurred. CONCLUSIONS: Facial CTA is a life changing procedure and has led to new treatment options for patients with complex, devastating and otherwise unreconstructable facial deformities to restore appearance and overall wellbeing in a single operation. The key to success lies in the selection of the appropriate patient, who is stable, well-motivated and therapy compliant. Thorough screening and follow-up by a multidisciplinary team, well prepared surgical approach and intensive, early rehabilitation are all crucial factors for minimizing complications and a safe and rapid recovery.

Articulation and oromyofunctional behavior in children seeking orthodontic treatment.

OBJECTIVES: The purpose of this controlled study is to document articulation and oromyofunctional behavior in children seeking orthodontic treatment. In addition, relations between malocclusions, articulation, and oromyofunctional behavior are studied. MATERIALS AND METHODS: The study included 56 children seeking orthodontic treatment. The control group, consisting of 54 subjects matched for age and gender, did not undergo orthodontic intervention. To determine the impact of the occlusion on speech, the Oral Health Impact Profile was used. Speech characteristics, intelligibility and several lip and tongue functions were analyzed using consensus evaluations. RESULTS: A significant impact of the occlusion on speech and more articulation disorders for/s,n,l,t/were found in the subjects seeking orthodontic treatment. Several other phenomena were seen more often in this group, namely more impaired lip positioning during swallowing, impaired tongue function at rest, mouth breathing, open mouth posture, lip sucking/biting, anterior tongue position at rest, and tongue thrust. Moreover, all children with a tongue thrust showed an anterior tongue position at rest. CONCLUSIONS: Children seeking orthodontics have articulatory and oromyofunctional disorders. To what extent a combined orthodontic and logopaedic treatment can result in optimal oral health (i.e. perfect dentofacial unit with perfect articulation) is subject for further multidisciplinary research.

Speech characteristics in a Ugandan child with a rare paramedian craniofacial cleft: a case report.

The purpose of this study is to describe the speech characteristics in an English-speaking Ugandan boy of 4.5 years who has a rare paramedian craniofacial cleft (unilateral lip, alveolar, palatal, nasal and maxillary cleft, and associated hypertelorism). Closure of the lip together with the closure of the hard and soft palate (one-stage palatal closure) was performed at the age of 5 months. Objective as well as subjective speech assessment techniques were used. The speech samples were perceptually judged for articulation, intelligibility and nasality. The Nasometer was used for the objective measurement of the nasalance values. The most striking communication problems in this child with the rare craniofacial cleft are an incomplete phonetic inventory, a severely impaired speech intelligibility with the presence of very severe hypernasality, mild nasal emission, phonetic disorders (omission of several consonants, decreased intraoral pressure in explosives, insufficient frication of fricatives and the use of a middorsum palatal stop) and phonological disorders (deletion of initial and final consonants and consonant clusters). The increased objective nasalance values are in agreement with the presence of the audible nasality disorders. The results revealed that several phonetic and phonological articulation disorders together with a decreased speech intelligibility and resonance disorders are present in the child with a rare craniofacial cleft. To what extent a secondary surgery for velopharyngeal insufficiency, combined with speech therapy, will improve speech intelligibility, articulation and resonance characteristics is a subject for further research. The results of such analyses may ultimately serve as a starting point for specific surgical and logopedic treatment that addresses the specific needs of children with rare facial clefts.

Impact of fixed implant prosthetics using the 'all-on-four' treatment concept on speech intelligibility, articulation and oromyofunctional behaviour.

The purpose of this case control study is to determine the impact of screw-retained fixed cross-arch prostheses, supported by four osseointegrated implants, on articulation and oromyofunctional behaviour. Objective (acoustic analysis) and subjective assessment techniques were used to determine the overall intelligibility, phonetic characteristics and oromyofunctional behaviour at an average period of 7.3 months after placement of the fixed implant prosthesis in 15 patients and 9 age-matched controls with intact dentition and without prosthetic appliances. Overall satisfaction with the prosthesis was 87%, but 53% of the subjects mentioned an impact on speech. 87% of the subjects presented with one or more distortions of the consonants. The most common distortions were distortions of the sound /s/ (sigmatismus simplex, 40% and sigmatismus stridens, 33%), simplex /z/ (27%), insufficient frication of /f/ (20%), /[symbol in text]/ (20%), addental production of /d/ (20%), /t/ (20%) or /s/ sound (13%) and devoiced /d/ (7%). In the control group, no articulation disorders were noted. Oromyofunctional behaviour in both groups was normal. To what extent motor-oriented speech therapy (with focus on tongue function) immediately after periodontal treatment (after wound healing) would decrease the persistent phonetic distortions is a subject for further research.

Parent and child ratings of satisfaction with speech and facial appearance in Flemish pre-pubescent boys and girls with unilateral cleft lip and palate.

The purpose of this controlled study is to determine satisfaction with speech and facial appearance in Flemish pre-pubescent children with unilateral cleft lip and palate. Forty-three subjects with unilateral cleft lip and palate and 43 age and gender matched controls participated in this study. The Cleft Evaluation Profile was used to assess the perceived satisfaction for individual features related to cleft care. Both the cleft palate subjects and their parents were satisfied with the speech and facial appearance. The Pearson χ(2) test revealed significant difference between the cleft palate and the control group regarding hearing, nasal aesthetics and function, and the appearance of the lip. An in depth analysis of well specified speech characteristics revealed that children with clefts and their parents significantly more often reported the presence of an articulation, voice and resonance disorder and experienced /s/ /r/ /t/ and /d/ as the most difficult consonants. To what extent the incorporation of specific motor oriented oral speech techniques regarding the realisation of specific consonants, attention to vocal and ear care, and the recommendation of secondary velopharyngeal surgery, with the incorporation of primary correction of the cleft nose deformity simultaneously with primary lip closure, will aid these patients are future research subjects.

Impact of anterior single-tooth implants on quality of life, articulation and oromyofunctional behaviour: a pilot study.

The main purpose of this study is to determine the impact (one and a half year after implantation) of a single-tooth implant on articulation and oromyofunctional behaviour. This information is important for dentists, orthodontists or stomatologists who treat professional speakers. Objective (acoustic analysis) as well as subjective (questionnaire, phonetic inventory, phonetic analysis, oromyofunctional assessment of lip and tongue function, blowing, sucking and swallowing) assessment techniques were used to determine the quality of life, articulation and oromyofunctional behaviour. Fourteen subjects who received a single-tooth implant and a control group of nine subjects participated in this study. The mean overall satisfaction of single-implant users was 95%. The subjects who received a single-tooth implant were capable of producing all Dutch vowels and consonants. The phonetic analysis revealed a sigmatism in 57% of the cases. Sigmatisms (stridens sigmatism followed by simplex sigmatism) were the most frequently observed phonetic disorders. There was interobserver agreement about normal lip and tongue functions. Moreover, blowing, sucking and swallowing patterns were normal (concordance value 96%). Detailed analyses 1 or 2 years after implantation of an anterior single-tooth implant revealed normal speech intelligibility and oromyofunctional behaviour. Articulation was characterised by the presence of a persistent phonetic disorder. Further research involving more subjects with an anterior single-tooth implant is needed.

Speech intelligibility of children with unilateral cleft lip and palate (Dutch cleft) following a one-stage Wardill-Kilner palatoplasty, as judged by their parents.

This study determined the intelligibility (words, sentences and story telling) of 43 children (mean age 4.9 years) with unilateral cleft lip and palate who received a Wardill-Kilner palatoplasty, as judged by their parents and determined the influence of age and gender. A comparison with normative data for intelligibility of 163 Dutch speaking children was made. Each child and the parents completed The Dutch intelligibility test. Measures reported include group mean intelligibility percentages for words, sentences and story telling for children aged 2.5-5.0 and 5.0-7.6 years. A significant correlation between age of the children and intelligibility was measured. No significant gender-related differences were found. An interesting finding is the absence of significant difference in intelligibility percentage between the children with cleft palate and the normative data for story telling. The reported intelligibility percentages provide important prognostic reference information for surgeons who perform palatoplasty and speech pathologists who assess the speech production of children with cleft palate.

Long-term impact of tongue reduction on speech intelligibility, articulation and oromyofunctional behaviour in a child with Beckwith-Wiedemann syndrome.

The purpose of the present case study was to determine the long-term impact of partial glossectomy (using the keyhole technique) on overall speech intelligibility and articulation in a Dutch-speaking child with Beckwith-Wiedemann syndrome (BWS). Furthermore the present study is meant as a contribution to the further delineation of the phonation, resonance, articulation and language characteristics and oral behaviour in a child with BWS. Detailed information on the speech and language characteristics of children with BWS may lead to better guidance of pediatric management programs. The child's speech was assessed 9 years after partial glossectomy with regard to ENT characteristics, overall intelligibility (perceptual consensus evaluation), articulation (phonetic and phonological errors), voice (videostroboscopy, vocal quality), resonance (perceptual, nasometric assessment), language (expressive and receptive) and oral behaviour. A class III malocclusion, an anterior open bite, diastema, overangulation of lower incisors and an enlarged but normal symmetric shaped tongue were present. The overall speech intelligibility improved from severely impaired (presurgical) to slightly impaired (5 months post-glossectomy) to normal (9 years postoperative). Comparative phonetic inventory showed a remarkable improvement of articulation. Nine years post-glossectomy three types of distortions seemed to predominate: a rhotacism and sigmatism and the substitution of the alveolar /z/. Oral behaviour, vocal characteristics and resonance were normal, but problems with expressive syntactic abilities were present. The long-term impact of partial glossectomy, using the keyhole technique (preserving the vascularity and the nervous input of the remaining intrinsic tongue muscles), on speech intelligibility, articulation, and oral behaviour in this Dutch-speaking child with congenital macroglossia can be regarded as successful. It is not clear how these expressive syntactical problems demonstrated in this child can be explained. Certainly they are not part of a more general developmental delay, hearing problems or cognitive malfunctioning. To what extent the presence of expressive syntactical problems is a possible aspect of the phenotypic spectrum of children with BWS is subject for further research. Multiple variables, both known and unknown can affect the long-term outcome after partial glossectomy in a child with BWS. The timing and type of the surgical technique, hearing and cognitive functioning are known variables in this study. But variables such as children's motivation, the contribution of the motor-oriented speech therapy, the parental articulation input and stimulation and other family, school and community factors are unknown and are all factors which can influence speech outcome after partial glossectomy. Detailed analyses in a greater number of subjects with BWS may help further illustrate the long-term impact of partial glossectomy.

The vocal quality in female student teachers during the 3 years of study.

The purpose of the present cross-sectional study was to determine the objective vocal quality and the vocal characteristics (vocal risk factors, vocal and corporal complaints) in 143 female student teachers during the 3 years of study. The objective vocal quality was measured by means of the Dysphonia Severity Index (DSI). Perceptual voice assessment, the Voice Handicap Index, questionnaires addressing vocal risks, and vocal and corporal complaints during and/or after voice usage were performed. Student teachers have a normal perceptual and objective vocal quality corresponding with a DSI% of 76. The analysis of variance revealed a significant improvement of the vocal quality between the first and the third year of study. No psychosocial handicapping effect of the voice was observed, though there are some vocal complaints and almost all student teachers reported the presence of corporal pain during and/or after speaking. Especially sore throat and headache were mentioned as the most present corporal pain symptoms. Due to the decreased awareness and the multifactorial genesis of the potential vocal risk factors, the student teachers are at risk for developing an occupational dysphonia during their teaching career. Because teaching is a high-risk profession for the development of voice problems, the incorporation of a direct vocal training technique to increase vocal endurance during teaching together with a vocal hygiene program, dietetics, and a stress management training program during the 3 years of study is needed to prevent occupational dysphonia.

Speech intelligibility of Flemish children as judged by their parents.

OBJECTIVE: Reduced intelligibility is a central concern in speech-impaired children, especially for parents. The main purpose of this study was to develop normative data for the intelligibility of Flemish-speaking boys and girls (age 2;06-5;0 years) as judged by their parents. Additionally, the influence of familiarity, gender and age of the child was determined. PATIENTS AND METHODS: Word, sentence and story productions of 163 healthy children (83 boys and 80 girls) between 2;06 and 5;0 years were transcribed by their own parents. Speech intelligibility was determined according to the intelligibility index described by Shriberg. Additionally, a panel of 12 unfamiliar judges listened to the recordings of a subgroup of 24 randomly selected children. RESULTS: Speech intelligibility improved with increasing age and was around 90% for children aged 4;6-5;0 years. Boys and girls performed equally on the task. For the intelligibility of words, mothers scored significantly better than unfamiliar listeners did. CONCLUSION: The reported normative speech intelligibility data provide important reference information for speech pathologists who assess child speech. Moreover, it enables them to measure the effect of a specific therapeutic approach and the spontaneous transfer to daily speech.

Vocal quality in university teachers: a pilot study.

OBJECTIVE: The main purpose of this study was to determine the vocal quality of 20 male and 9 female university teachers using a multi-parameter approach. Secondly, the effect of an academic lecture on the voice profiles of the university teachers was measured. METHODOLOGY: All groups underwent subjective voice evaluations (perceptual evaluation, Voice Handicap Index, anamnesis of vocal complaints and vocal abuse) and objective voice evaluations (aerodynamic and acoustic parameters, vocal performance, and the Dysphonia Severity Index). The same voice assessment was performed after an academic lecture with a mean length of one and a half hours. RESULTS: The mean DSI score was + 2.2 for the male teachers and + 4.0 for the female teachers. The mean VHI score was 13. Perceptually, all voice parameters were rated as normal. The questionnaire revealed a relatively high amount of vocal abuse. No changes in the objective vocal parameters were found after the lecture. Perceptually, however, the voices of the university teachers were significantly less instable after the lecture. Although no negative changes in objective vocal quality were observed, 48% of the university teachers experienced subjective vocal changes. CONCLUSION: The authors concluded that university teachers are professional voice users with good vocal quality who suffer no handicapping effect from possible voice disorders. No important changes in the vocal profile after a teaching activity of one and a half hours were found, despite the high prevalence of voice complaints.

Long-term outcome of hyperfunctional voice disorders based on a multiparameter approach.

SUMMARY: The purpose of this study is to determine the long-term voice outcome (6.1 years after a well-defined voice treatment program) of hyperfunctional voice disorders in 27 subjects. All patients showed a muscle tension pattern type I (MTP I). Perceptual ratings, aerodynamic and acoustical analyses, Voice Handicap Index (VHI) value, and a determination of the Dysphonia Severity Index (DSI) were performed. The laryngovideostroboscopic images indicated that 51% of the subjects still show pathological laryngological findings. The negative evolution of the DSI from -1 to -3.2 is in agreement with this finding. Analysis of the components of the DSI shows that the main responsible variable for this negative change is the lowest intensity (I-low) that increased with 8.1 dB, indicating that subjects generally speak too loud, which is a typical problem for vocal hyperfunction. The VHI-score indicates an unimportant psychosocial impact of the voice disorder. The more objective and laryngostroboscopic findings indicate a chronic situation for a substantial part of the subjects and even a worse situation for some of them. Whether the long-term voice outcome results can be changed with the insertion of several follow-up voice rehabilitation sessions over the years remains unanswered and is a subject for further research.

The impact of mandibular advancement on articulation, resonance and voice characteristics in Flemish speaking adults: a pilot study.

Bilateral saggital split osteotomy (BSSO) of the mandible is a frequently performed mandibular orthognathic procedure, used to resolve mandibular disharmonies. Literature review showed contradictory findings regarding the effect of the orthognathic surgery on speech characteristics. The purpose of the present study was to determine a detailed analysis of the articulation, resonance and voice characteristics after BSSO with mandibular advancement for the treatment of Class II malocclusions using objective and subjective assessment techniques (perceptual evaluations, Dysphonia Severity Index, nasalance scores) in eight subjects. The findings of the present study indicate that before and after BSSO with mandibular advancement three types of articulation disorders may predominate in the Flemish language: the incorrect production of the trill sound /r/ and the /s/ sound and devoicing of the /z/. After orthognathic surgery most patients showed an identical articulation pattern (normal or disturbed pattern) as in the presurgical condition. In this study the BSSO with mandibular advancement had no significant impact on the nasality characteristics and the nasalance values probably due to the competent velopharyngeal valving in the presurgical condition. And, as expected the vocal quality revealed no significant difference. The maxillofacial surgeon and the speech language pathologist must be aware of the persistency of these preoperative articulation errors in the postsurgical condition.

Vocal quality characteristics in children with cleft palate: a multiparameter approach.

The main purpose of the present study was to examine the vocal quality and to investigate the effects of gender on vocal quality in 28 children with a unilateral or bilateral cleft palate. In this study, the vocal quality was determined using videolaryngostroboscopic and perceptual evaluations, aerodynamic, voice range, acoustic, and dysphonia severity index (DSI) measurements. The DSI is based on the weighted combination of four voice measurements and ranges from +5 to -5 for, respectively, normal and severely dysphonic voices. Additional objectives were to compare the vocal quality characteristics of children with cleft palate with the available normative data and to investigate the impact of the cleft type on vocal quality. Gender-related vocal quality differences were found. The male cleft palate children showed an overall vocal quality of +0.62 with the presence of a perceptual slight grade of hoarseness and roughness. The female vocal quality had a DSI value of +2.4 reflecting a perceptually normal voice. Irrespective of the type of cleft, all subjects demonstrated a significantly lower DSI-value in comparison with the available normative data. The results of the present study have provided valuable insights into the vocal quality characteristics of cleft palate children.

Effect of cleft type on overall speech intelligibility and resonance.

The main purpose of this study was to measure and compare the effect of an unilateral cleft lip and palate (UCLP) and a bilateral cleft lip and palate (BCLP) on overall speech intelligibility, nasalance and nasality. An additional objective was to compare the nasalance and nasality patterns of cleft palate children with the available normative data for normal children. The subjects were 37 children with an UCLP (19/37) or a BCLP (18/37) with normal cognitive and motor functions and normal hearing levels. All subjects had a non-syndromic cleft, followed the same surgical protocol and did not undergo secondary pharyngeal surgery. To measure and compare the effect of cleft type, objective and subjective assessment techniques were used. For the objective assessment of nasal resonance the nasometer and the mirror-fogging test were used. Nasalance scores were obtained, while patients produced sounds, read three standard passages or repeated sentences focusing on specific consonants. A perceptual evaluation of speech, the Gutzmann (1913) test and the tests of Bzoch (1989) were used as subjective assessment techniques. The perceptual assessment of the overall speech intelligibility was based on the spontaneous speech and reading or repeating of the nasometric sentences. These samples were judged by three speech pathologists using a 4-category nominal scale. A subjective test (Morley, 1945) was used to evaluate the velopharyngeal mechanism. The findings of the present study show no statistically significant differences for nasalance values, nasality and overall speech intelligibility between the UCLP and BCLP children. Significant differences were found between the data of the normal group and the cleft palate children. These differences included nasalance values as well as nasality data and overall speech intelligibility.

Speech patterns in children with velo-cardio-facial syndrome. Two case studies.

The purpose of this study was to document the articulation patterns in children with velo-cardio-facial syndrome. The subjects were 2 children with this syndrome, aged 6 years 6 months and 3 years 11 months. Phonetically transcribed speech samples collected by means of a picture naming test formed the basis for this study. The speech samples were subjected to two types of analyses: independent analyses describing the children's sound productions regardless of their relation to the adult targets and relational analyses comparing the children's productions with the adult standard form. It was found that the articulatory problems in velo-cardio-facial syndrome are not simply due to a delay in speech sound acquisition. Articulation errors found in this syndrome are uncharacteristic of normal speech development. Whether errors are syndrome-specific awaits further investigation.

Nasometric values for normal nasal resonance in the speech of young Flemish adults.

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.

Speech patterns in Kabuki make-up syndrome: a case report.

The case of a girl aged 3 years and 8 months with Kabuki make-up syndrome is reported. At presentation, she had normal cognitive functioning, and she also had a history of otitis media, a submucous cleft palate, and some hypotonia. Language testing showed normal receptive skills and good expressive vocabulary but poor morphosyntactic abilities. Speech analysis showed that she was capable of producing most of the sounds of her native language but demonstrated high variability in production of the sounds. In addition, she inconsistently simplified words by application of several phonologic processes. Possible explanations for the communication problems demonstrated are discussed.