Language skills of young children with unilateral cleft lip and palate following infant orthopedics: a randomized clinical trial.

OBJECTIVE: To investigate the effects of infant orthopedics (IO) on the language skills of children with complete unilateral cleft lip and palate (UCLP). DESIGN: In a prospective randomized clinical trial (Dutchcleft), two groups of children with complete UCLP were followed up longitudinally: one group was treated with IO based on a modified Zurich approach in the first year of life (IO group); the other group did not receive this treatment (non-IO group). At the ages of 2, 2(1/2), 3, and 6 years, language development was evaluated in 12 children (six IO and six non-IO). Receptive language skills were assessed using the Reynell test. Expressive language skills of the toddlers were evaluated by calculating mean length of utterance (MLU) and mean length of longest utterances (MLLU); in the 6-year-olds, the expressive language skills were measured using standardized Dutch language tests. PATIENTS: The participants had complete UCLP without soft tissue bands or other malformations. RESULTS: IO did not affect the receptive language skills. However, the expressive language measures MLU and MLLU were influenced by IO. At age 2(1/2) and 3 years, the IO group produced longer utterances than the non-IO group. In the follow-up, the difference in expressive language between the two groups was no longer significant. CONCLUSIONS: Children treated with IO during their first year of life produced longer sentences than non-IO children at the ages of 2(1/2) and 3 years. At 6 years of age, both groups presented similar expressive language skills. Hence, IO treatment did not have long-lasting effects on language development.     

Use of a perceptual evaluation instrument to assess the effects of infant orthopedics on the speech of toddlers with cleft lip and palate.

OBJECTIVE: To investigate the effects of infant orthopedics (IO) administered in the first year of life on the speech characteristics of 2.5-year-old children with complete unilateral cleft lip and palate (UCLP) using a perceptual evaluation instrument with equal-appearing interval (EAI) scales. DESIGN: In a prospective randomized clinical trial (Dutchcleft), two groups of children with complete UCLP were followed longitudinally. One group received IO based on a modified Zurich approach (IO group), and the other group did not (non-IO group). The appliance was used until soft palate closure at age 12 months. Hard palate closure is delayed until 9 years of age. PARTICIPANTS: Three groups of 2.5-year-old toddlers participated in this investigation: 10 IO, 10 non-IO, and 8 noncleft controls matched for age and socioeconomic status. METHOD: Five trained listeners assessed the children's speech in a blinded perceptual rating procedure. They judged 13 specific speech characteristics and indicated their total impression of speech on EAI scales. RESULTS: The reliability and consistency of 11 of the rating scales was good. The intelligibility rating scale was the single speech characteristic that distinguished the IO group from the non-IO group; the IO group was judged to be superior. The cleft groups differed from the noncleft group on 9 of the 11 scales. CONCLUSIONS: Evaluation of speech by means of the present newly developed perceptual rating instrument showed that the IO group obtained significantly higher ratings for intelligibility than the non-IO group. The groups did not differ regarding any of the other speech aspects.     

Cost-effectiveness of infant orthopedic treatment regarding speech in patients with complete unilateral cleft lip and palate: a randomized three-center trial in the Netherlands (Dutchcleft).

OBJECTIVE: To investigate the cost-effectiveness of infant orthopedic treatment (IO), compared with no such treatment in children with complete unilateral cleft lip and palate (UCLP) focusing on the effects on speech development at age 2.5 years. DESIGN: In a three-center prospective, randomized clinical trial (Dutchcleft), two groups of children with complete UCLP were followed longitudinally: one group was treated with IO based on a modified Zurich approach in the first year of life (IO group) and the other group did not receive this treatment (non-IO group). PATIENTS: The participants had complete UCLP without soft tissue bands or other malformations. They were born at term and their parents were native Dutch speakers. OUTCOME MEASURES: The effect of IO on speech development at age 2.5 years was measured blindly by five expert listeners judging the "total impression of speech quality" on a 10-point equal-appearing interval scale. Costs were measured from a societal viewpoint in Euro. RESULTS AND CONCLUSION: The IO group (n = 10) obtained a statistically significant higher rating, compared with the non-IO group (n = 10). The effect size was large, indicating that the improvement may be considered a clinically important change. The cost for treatment by the orthodontist was higher in the IO group. For both groups, the mean cost was related to the mean rating for "total impression of speech quality." The resulting cost-effectiveness for IO, compared to non-IO was 1041 for 1.34 point speech quality improvement. The financial investment that is necessary to obtain this improvement seems limited. Thus, from the perspective of speech development, the cost-effectiveness of IO over non-IO seems acceptable at this point in time.     

Effect of infant orthopedics on facial appearance of toddlers with complete unilateral cleft lip and palate (Dutchcleft)

OBJECTIVE: To evaluate the effect of infant orthopedics (IO) on facial appearance of 54 patients with unilateral cleft lip and palate (UCLP), aged 4 and 6 years. DESIGN: Prospective two-arm randomized controlled clinical trial in three Cleft Palate Centers in the Netherlands (Dutchcleft-trial). INTERVENTIONS: Patients were divided randomly into two groups. Half of the patients (IO+) had a plate until surgical closure of the soft palate at the age of +/- 52 weeks; the other half (IO-) received no intervention. MAIN OUTCOME MEASURES: Facial appearance at 4 and 6 years of age assessed on full face photographs and photographs showing only nose and mouth. Ratings were performed on a VAS-scale by professionals and laymen. RESULTS: At 4 years of age the full face pictures of IO+ children were scored to be more attractive than those of IO- children. However, this difference had disappeared at 6 years of age. At the age of 6, only professionals saw a significant difference on nasolabial photographs between IO+ and IO-. Regression analysis showed a minor effect of occlusion, lip revision, or type of nose reconstruction on the esthetic results. CONCLUSIONS: IO had a positive effect on full facial appearance of UCLP children at the age of 4 years, but at the age of 6, only professionals saw a positive effect of IO on the nasolabial photographs. This is irrelevant for UCLP patients since they deal with laymen in their daily life.     

The effect of infant orthopedics on the occlusion of the deciduous dentition in children with complete unilateral cleft lip and palate (Dutchcleft).

OBJECTIVE: Evaluation of the effect of infant orthopedics (IO) on the occlusion of the deciduous dentition in patients with unilateral cleft lip and palate (UCLP). DESIGN: Prospective, two-arm, randomized, controlled clinical trial with three participating cleft palate centers (Dutchcleft). SETTING: Cleft Palate Centers of the University Medical Center Nijmegen, Academic Center of Dentistry Amsterdam, and Dijkzigt University Hospital Rotterdam, The Netherlands. PATIENTS: Children with complete UCLP (n = 54) were included. INTERVENTIONS: In a concealed allocation procedure, half of the patients was randomized to wear a plate till surgical closure of the soft palate (IO+), and the other half (IO-) did not have a plate. MEAN OUTCOME MEASURES: Dental arch relationships were assessed at 4 and 6 years of age with the 5-year-old index; the Huddart-score; and measurements of overjet, overbite, and sagittal occlusion. RESULTS: There were no significant differences found between the IO+ and IO- groups for the 5-year-old index; the Huddart-score; and overjet, overbite, and sagittal occlusion. CONCLUSIONS: IO had no observable effect on the occlusion in the deciduous dentition at 4 and 6 years of age. Considering the occlusion only, there is no need to perform IO in children with UCLP.     

Midfacial morphology in children with unilateral cleft lip and palate treated by different surgical protocols

The purpose of this study was to compare cranio-facial, particularly midfacial morphology, in two groups of children with complete unilateral cleft lip and palate (UCLP) treated at two different cleft centres (Hannover. Germany and Brussels, Belgium) following different surgical treatment protocols. A total of 62 children (40 males; 22 females) with non-syndromic UCLP were included in this study at approximately the age of 10. The Hannover group comprised 36 children, who had repair of the lip at a mean age of 5.83 +/- 1.16 months, followed by repair of the hard and soft palate at a mean age of 29.08 +/- 4.68 and 32.25 +/- 4.29 months. respectively. The Brussels group consisted of 26 children who underwent surgical treatment according to the Malek protocol: the soft palate was closed at a mean age of 3.04 +/- 0.20 months, followed by simultaneous repair of the lip and hard palate at a mean age of 6.15 +/- 0.68 months. Midfacial morphology was evaluated by means of cephalometric analysis according to Ross. The children in the Hannover UCLP group did not differ significantly from those in the Brussels group in the anteroposterior dimension of the midface. However, the maxillary plane was significantly more open in the Brussels group due to less posterior vertical maxillary development.     

A cross-sectional study of speech in 10-year-old children with cleft palate: results and issues of rater reliability.

OBJECTIVES: To describe speech based on perceptual evaluation in a group of 10-year-old children with cleft palate. A secondary aim was to investigate the reliability of speech-language pathologists' perceptual assessment of cleft palate speech. DESIGN: Retrospective cross-sectional study in children with cleft palate. External raters made assessments from randomized speech recordings. SUBJECTS: Thirty-eight children with unilateral cleft lip and palate (UCLP) or cleft palate only (CPO) and 10 children in a comparison group. MAIN OUTCOME MEASURES: Ratings of hypernasality, hyponasality, audible nasal air leakage, weak pressure consonants, and articulation. Exact agreement and weighted kappa values were used for reliability. RESULTS: Hypernasality was found in 25% of children with a cleft of the soft palate (CSP), 33% of children with a cleft of the hard and soft palate (CHSP), and 67% of children with a UCLP. Similar results were found for audible nasal air leakage. Articulation errors were found in 6% of the CHSP group and 25% of the UCLP group, whereas no child in the CSP group had articulation errors. The reliability was moderate to good for different variables, with lowest values for hypernasality. CONCLUSIONS: Speech results in this series seem less satisfactory than those reported in other published international studies, but it is difficult to draw any certain conclusions about speech results because of large methodological differences. Further developments to ensure high reliability of perceptual ratings of speech are called for.     

Facial characterization of infants with cleft lip and palate using a three-dimensional capture technique.

OBJECTIVE: To characterize the soft tissue features of infants with unilateral cleft lip (UCL) and unilateral complete cleft lip and palate (UCLP) prior to primary surgery and compare with noncleft controls. DESIGN: Prospective controlled capture of the facial morphology of infants using a noninvasive three-dimensional stereophotogrammetry method. PARTICIPANTS: 23 children with presurgical cleft: 11 UCL (M = 6, F = 5); 12 UCLP (M = 9, F = 3), and 21 noncleft controls (M = 7, F = 14) were imaged at approximately 3 months of age (range 10 to 16 weeks). MAIN OUTCOME MEASURE: Accurate, repeatable quantification of facial soft tissues in infants with clefts prior to surgery. RESULTS: Significant differences (p <.05) were found between the UCLP group and UCL and control groups in anatomical and soft nose width, cleft-side alar wing length, and nasal tip horizontal displacement. Both cleft groups were significantly different from controls and from each other in cleft-side nostril dimensions, alar wing angulation, columella angle, and alar base to corner of mouth dimension; alar base width; and soft tissue defect in nose and the lip and philtrum length bordering the cleft. Significant differences between clefts and controls were identified in the nostril and philtrum on the noncleft side. CONCLUSIONS: The use of children with UCL as controls for UCLP studies is inappropriate. This technique overcame the limitations of direct measurement of infant faces to aid the surgeon in the planning and subsequent re-evaluation of surgical rationale.     

A longitudinal study of speech production in Swedish children with unilateral cleft lip and palate and two-stage palatal repair.

OBJECTIVE: To describe speech production longitudinally in a group of children with unilateral cleft lip and palate (UCLP). PARTICIPANTS: Twenty consecutive children with UCLP and nine age-matched children without clefts in a comparison group. INTERVENTION: A two-stage palatal repair procedure with soft palate closure at 6 months and hard palate repair at 3 to 4 years. MAIN OUTCOME MEASURES: Percent correct consonants (PCC), percent correct places (PCP), and percent correct manners (PCM) at 3, 5, and 7 years of age. Cleft speech errors at the same ages. Previously collected data on number of consonant tokens, consonant types, frequency of occurrence of places and manners of articulation at 18 months. RESULTS: PCC and PCP were significantly lower in the UCLP group than in the comparison group at all ages. Number of consonant types and frequency of occurrence of dental plosives at 18 months correlated significantly with PCC at age 3. A high frequency of velar plosives at 18 months correlated significantly with a high prevalence of retracted oral articulation (dental/alveolar to palatal or velar) at both 3 and 5 years of age. CONCLUSIONS: The UCLP group performed worse than the comparison group at all ages. A high occurrence of dental plosives as well as a high number of consonant types in babbling and first words seem to be good indicators for better consonant production in later speech. The same prevalence of retracted oral articulation as in previous studies is attributed to the surgical technique.     

婴儿单侧完全性唇腭裂唇鼻体表特点的测量研究

目的:研究单侧完全性唇腭裂的唇鼻体表特点,为适龄婴儿手术治疗及治疗后效果的评估提供理论参考和指导。方法:按纳入标准随机选取92例单侧完全性唇腭裂患儿,采用直接测量法对其唇鼻体表进行测量,与正常对照组比较。结果:获得单侧完全性唇腭裂婴儿和正常婴儿唇鼻体表的28项数据的基本均值和标准差。单侧完全性唇腭裂的数据与对照组比较:除去患侧高点上唇红厚、健侧唇高及健侧鼻孔底宽无统计学差异外,其余数据均有显著性统计学差异(P<0.01)。单侧完全性唇腭裂数据间的相关分析发现多项数据间存在相关性,有统计学意义(P<0.05)。结论:单侧完全性唇腭裂具有复杂的唇、鼻畸形,在三维空间上形成一个畸形的整体,既表现有组织的缺损又存在组织的移位,各项测量指标的差异很大但在同一个体的成对指标间却具有明显的正相关性。     

Infant orthopedics in UCLP: effect on feeding, weight, and length: a randomized clinical trial (Dutchcleft).

OBJECTIVE: To study the effects of infant orthopedics (IO) on feeding, weight, and length. DESIGN: Prospective two-arm randomized controlled trial in three academic Cleft Palate Centers. Treatment allocation was concealed and performed by means of a computerized balanced allocation method. SETTING: Cleft Palate Centers of Amsterdam, Nijmegen, and Rotterdam, the Netherlands. PATIENTS: Infants with complete unilateral cleft lip and palate (UCLP), no other malformations. INTERVENTIONS: One group (IO+) wore passive maxillary plates during the first year of life, but the other group (IO-) did not. All other interventions were the same for both groups. MAIN OUTCOME MEASURES: Bottle feeding velocity (mL/min) at intake, 3, 6, 15, and 24 weeks (T0 to T24); weight-for-age, length-for-age, and weight-for-length using z scores; reference values from the Netherlands' third nationwide survey on growth. RESULTS: Feeding velocity increased with time from 2.9 to 13.2 mL/min in the IO- group and from 2.6 to 13.8 mL/min in the IO+ group; no significant differences were found between groups. Weight-for-age, length-for-age, and weight-for-length (z scores) did not differ significantly between groups, but overall the infants with unilateral cleft lip and palate in both groups had significantly lower mean z scores for weight-for-age and height-for-age than the reference during the first 14 months, and had lower mean values for weight-for-length after soft palate closure. CONCLUSION: Infant orthopedics with the aim of improving feeding and consequent nutritional status in infants with unilateral cleft lip and palate can be abandoned.     

Craniofacial morphology in children with operated complete unilateral cleft lip and palate

The objective of the study was to compare the craniofacial characteristics of children with operated unilateral complete cleft lip and palate (UCLP) with those of noncleft children. Lateral and posteroanterior cephalometric radiographs of subjects with UCLP and controls who were matched for sex, age, and ethnic origin were analyzed and compared. There were a total of 21 subjects with UCLP (10 boys and 11 girls) from the Orthodontic Department of Selcuk University and 15 controls (7 boys and 8 girls) from the Orthodontic Department of Ankara University. The nasal bone length in the group with cleft did not differ significantly from that in the control group. Subjects with UCLP included in the study appeared to have shorter faces, smaller mandibulae, greater lateral orbital width, and smaller sella turcicae compared with controls. Unilateral cleft lip and palate affects the intracranial morphology as well as dentofacial structures.     

Changes in soft tissue thickness after Le Fort I osteotomy in different cleft types

The soft tissue thickness before and after Le Fort I osteotomy was evaluated in 46 cleft patients. The sample consisted of 10 patients with isolated cleft palate (CP, mean age 25.5 years); 10 patients with bilateral cleft lip and palate (BCLP, mean age 21.7 years); and 26 patients with unilateral cleft lip and palate (UCLP, mean age 22.9 years). Patients with bimaxillary surgery, simultaneous rhinoplasty, or V-Y plasty of the upper lip were excluded. Soft tissue changes were analyzed by cephalograms taken shortly before surgery and at 6 months postoperatively. Horizontal advancement varied from 4.1 mm in the UCLP group to 5 mm in the BCLP group. The vertical lengthening varied from 3.7 mm in the CP group to 7.2 mm in the BCLP group. In all cleft types, thinning of the subnasal area, superior labial sulcus, and upper lip (anterior nasal spine-subnasale, point A-soft tissue point A, and prosthion-labrale superius) took place. Significant thinning of the upper lip occurred in the UCLP and BCLP patients. Surgical changes of the lower lip and mandibular area were small and insignificant. There were significant differences in soft tissue thicknesses between different types of clefts. The subnasal area and superior labial sulcus were significantly thicker in the CP group than in the BCLP or UCLP groups, both pre- and postoperatively. The upper lip was thickest in the BCLP group preoperatively but thickest in the CP group postoperatively. The upper lip was thinnest in the UCLP group both before and after the operation.     

Evaluation of cheiloplasty and palatoplasty on palate surface area in children with oral clefts: longitudinal study

This study evaluated the palatal surface area in children with different oral clefts after primary surgeries and at five years of age. This longitudinal study was composed by 216 digital models: unilateral complete cleft lip (UCL), unilateral complete cleft lip and palate (UCLP), and complete cleft palate (CP). The models were analysed at four time periods: T1 (before cheiloplasty), T2 (before palatoplasty), T3 (after palatoplasty); and T4 – (at five years of age). Area of the dental arches was measured through stereophotogrammetry software. Measurements evaluated with Student’s test and ANOVA followed by the Tukey test (p<0.05) (AQ 1). In the UCL group, the palatal surface area significantly increased among phases. In the primary surgery periods, UCLP and CP significantly decreased (p<0.001). Palatal area in the UCLP group was significantly greater than the CP group. Overall, no statistically significant differences occurred among groups. At T4, the area of the palate in the UCL group was significantly greater than the UCLP group and no significant differences occurred between UCLP and CP groups. This study suggests that cheiloplasty did not inhibit the growth of the palatal surface area in children with UCL and UCLP. Palatoplasty significantly decreased the palatal area in children with UCLP and CP, demonstrating a significant negative effect of palatal repair on maxillary growth. At five years, children with UCLP and CP had a significantly smaller palate area than those with UCL.     

Mandibular morphology in complete unilateral cleft lip and palate.

OBJECTIVE: The purpose of this study was to evaluate and compare mandibular morphology and spatial position in children with complete unilateral cleft lip and palate (UCLP) treated at two different cleft centers (Hannover and Brussels) following different surgical treatment protocols. PATIENTS: A total of 62 Caucasian children (40 boys, 22 girls) with nonsyndromic complete unilateral cleft lip and palate (UCLP) were evaluated by means of conventional cephalometric analysis at approximately the age of 10 years. Data of both cleft groups were compared with a control, noncleft group (n = 40) matched according to age and sex. INTERVENTIONS: The Hannover children with cleft (n = 36) underwent lip repair at a mean age of 5.83 +/- 1.16 months. The hard and soft palates were closed at a mean age of 29.08 +/- 4.68 and 32.25 +/- 4.29 months, respectively. The Brussels children with cleft (n = 26) were treated according to the Malek surgical protocol with soft palate repair at a mean age of 3.04 +/- 0.20 months and simultaneous lip and hard palate repair at a mean age of 6.15 +/- 0.68 months. RESULTS: Statistical analysis (analysis of variance with post hoc Tukey's test) showed a significant (p =.001) smaller mandibular ramus length (Co-Go) in the Brussels cleft group, compared with the control group. The Hannover-Brussels comparison data revealed that the S-N-B angle was significantly (p =.047) less in the Brussels cleft group. CONCLUSIONS: The influence of surgical procedures in patients with UCLP might not be restricted to the maxilla but could influence mandibular spatial position to the cranial base. Because of these positional changes of the mandible, both cleft groups showed facial balance.     

Spectral analysis of prespeech sounds (spontaneous cries) in infants with unilateral cleft lip and palate (UCLP): a pilot study.

OBJECTIVE: The objectives of the present study were: (1) to analyze the cry features of infants with cleft lip and palate (UCLP) by means of spectral analysis, (2) to describe changes of the acoustic parameters from birth until 9 months of age, and (3) to compare these data with existing cry data of infants without cleft (control group). DESIGN: The study was designed on a interdisciplinary, prospective, and longitudinal basis. SETTING: Interdisciplinary study: (1) Institute of Anthropology at the Humboldt-University, Berlin; (2) Heidelberg University Hospital: Interdisciplinary Cleft Palate and Craniofacial Center. PATIENTS AND METHOD: The cry parameters of five patients with complete unilateral cleft lip, alveolar ridge, and hard and soft palate were analyzed from birth to 9 months of age. The patients were treated with the same protocol. At the age of 24 months, sensomotor development was assessed using the KIPHARD test. Perceptual judgment of speech, performed after 36 months of life, included nasal resonance, nasal emission of air, articulation disorders, and speech intelligibility. MAIN OUTCOME MEASURE: The cry parameters of fundamental frequency (F(0)), pitch period perturbation quotient (PPQ), and cry duration (Tsam) were analyzed. RESULTS: Contrary to the expectation that laryngeal parameters are not affected by vocal tract malformations, differences of cry parameters were found between the patients with UCLP and the non-cleft group. Particularly, the F(0) and its short-time variability (PPQ) were affected. CONCLUSIONS: The preliminary results of this study showed that F(0) and PPQ of spontaneous cries are influenced in patients with UCLP, and a cry analysis might become a noninvasive tool for early detection of an at-risk status for neuromuscular development and prediction of an at-risk status for later speech and language acquisition in infants with cleft lip and palate. Future research strategies are outlined.     

Early maxillary orthopedics in CLP cases: guidelines for surgery.

Two samples of complete cleft cases between five and seven years of age are compared (11 UCLP + 9 BCLP versus 21 UCLP + 12 BCLP). The same early orthopedic procedures were applied to all cases, but surgical management differed for the two groups: "classical" surgery (lip 3 months, palate 2--2 1/2 years) versus delayed conservative surgery (lip 6 months, soft palate 18 months, hard palate 6--8 years). It is evident that early maxillary orthopedic treatment is of little consequence for long-term development unless concomitant surgery complies with growth and functional requirements. In contrast to "classical" surgery, palatal closure in two steps is more favourable to skeletal growth and does not interfere to any relevant degree with speech development.     

A comparison of craniofacial form in Northern Irish children with unilateral cleft lip and palate treated with different primary surgical techniques.

OBJECTIVE: The quality of outcomes in patients with unilateral cleft lip and palate (UCLP) was compared between two senior plastic surgeons. One surgeon carried out a one-stage Wardill-Kilner palate repair. The other surgeon employed a vomer flap hard palate repair followed by a von Langenbeck soft palate closure (Oslo protocol). DESIGN: Retrospective analysis. PATIENTS: Thirty-four children (mean age 9.7 years) born with complete skeletal UCLP in Northern Ireland from 1983 to 1991 who received primary repair surgery from one of the two surgeons. MAIN OUTCOME MEASURES: Cephalometric analysis was used to determine the craniofacial form and soft tissue profile. The quality of the dental arch relationships was independently assessed using the Goslon ranking system. RESULTS: Twenty-nine of the 34 subjects had good or satisfactory arch relationships. Comparison between the surgeons revealed that the Wardill-Kilner group had a greater proportion of Goslon grades of greater than 3, indicating poor arch relationships. However, this difference failed to reach statistical significance. There were no significant cephalometric differences between patients treated by the two surgeons. CONCLUSIONS: Although the difference was not statistically significant, the highest proportion of patients likely to require orthognathic surgery was found in those treated using the Wardill-Kilner technique.