Long-term Follow-up of Bilateral Cleft Lip and Palate: Incidence of Speech-Correcting Surgeries and Fistula Formation.

While bilateral cleft lip and palate (BCLP) constitutes a clinical challenge for the whole cleft team, the ideal surgical protocol remains obscure. This study presents the long-term burden of care in terms of secondary surgeries, defined as fistula repair and speech-correcting surgeries (SCS), in a single center. Outcomes of two surgical protocols utilized over the years were also compared.A retrospective single-center analysis of 81 non-syndromic children with complete BCLP born between 1990 and 2010. Two surgical protocols comprising single-stage and two-stage (delayed hard palate closure) procedures were compared. Outcome was analyzed at the time of alveolar bone grafting (ABG) and post-ABG.Altogether 54 children (66.7%) had underwent secondary surgery by the time of bilateral ABG. At this point, 38.3% (n = 31) of patients had received SCS and 49.4% (n = 40) had undergone fistula repair. The corresponding incidences at the end of follow-up were 46.9% (n = 38) and 53.1% (n = 43). No significant difference emerged in SCS incidence between the 2 protocols; however, prior to ABG the single-stage protocol had a significantly lower need for fistula repair. Regarding the location of fistulas, some differences were observed, with the single-stage procedure more associated with anterior fistulas.BCLP has a high surgical burden of care in terms of secondary surgeries, defined as SCS and fistula repair. In our experience, the single-stage protocol, particularly the two-flap technique, offers better results in the management of BCLP than the two-stage approach with a short delay in hard palate closure.

Speech of Patients With Unilateral Complete Cleft Lip and Palate: Comparison of Three Different Surgical Protocols for Primary Repair.

BACKGROUND AND AIMS: Between 1997 and 2014, 3 protocols have been used in out cleft unit for primary repair of unilateral cleft lip and palate. During the Scandcleft randomized controlled trial closing the soft palate and lip at 4 months and the hard palate at 12 months (Protocol 1) was compared with closing the entire palate at 12 months (Protocol 2). Protocol 3 comprises closure of the lip and hard palate with a vomer flap at 4 months and the soft palate at 10 months. The purpose of this study was to compare subsequent velopharyngeal competence at age of 3 and 5 years. PATIENTS AND METHODS: The study consisted of 160 non-syndromatic patients with a unilateral cleft lip and palate. Protocol 3 was retrospectively compared with Protocols 1 and 2 within the previously published Scandcleft study. RESULTS: At 3 years of age, normal or borderline competent velopharyngeal function was found in 68% of patients in Protocol 1, 74% of patients in Protocol 2, and 72% of patients in Protocol 3. At 5 years of age, the corresponding figures were 84%, 82%, and 92%. 21% of patients in Protocol 1, 4% in Protocol 2, and 23% in Protocol 3 had palatal reoperations before the age of 5 years. CONCLUSION: No significant differences emerged in velopharyngeal competence at age 3 years between the 3 protocols. Palatal reoperations were performed earlier in patient groups 1 and 3, explaining the difference in the velopharyngeal competence rate at the 5-year time-point.

Anterior crossbite, dental arch dimensions, and later need for orthognathic surgery in 6-year-old children with unilateral cleft lip and palate.

Objective : Six-year-old children with unilateral cleft lip and palate were examined to compare the prevalence of anterior crossbite and dental arch dimensions of those who later needed orthognathic surgery with the prevalence of those who did not. Design : Retrospective longitudinal study. Patients : A total of 68 consecutive nonsyndromic patients with unilateral cleft lip and palate (44 boys, 24 girls). Main Outcome Measures : Children with unilateral cleft lip and palate whose palates had been closed in one stage by the Veau-Wardill-Kilner or Cronin-Brauer V-Y pushback techniques were analyzed from dental casts taken at a mean age of 6.1 years (range, 5.7 to 6.8 years) before orthodontic treatment or bone grafting. The need for orthognathic surgery in these patients was determined from hospital records at the mean age of 18.2 years (range, 15.6 to 20.2 years). Student's t test and chi-square test were used in statistical analyses. Results : The prevalence of anterior crossbite was 62% (one or both central incisors in full crossbite). The prevalence was higher (75% versus 53%) in children later needing orthognathic surgery (28 of 68, 41%), but the difference was not significant. Nor were there significant differences in dental arch measurements between children who later needed osteotomies and those who did not or between the two modifications of the primary palatal pushback operations. Conclusions : The prevalence of anterior crossbite and the dental arch dimensions did not differ between 6-year-old children with unilateral cleft lip and palate who later needed orthognathic surgery and those who did not.

Cone beam computed tomography in the assessment of alveolar bone grafting in children with unilateral cleft lip and palate.

OBJECTIVES: To quantify the treatment outcome of secondary alveolar bone grafting (SABG) in individuals with unilateral cleft lip and palate using cone beam computed tomography (CBCT) and to reveal needs for improvement in surgical technique. MATERIAL AND METHODS: CBCT images taken 6 months after SABG of 35 patients were analysed. Vertical and horizontal bone supports of the grafted bone at three levels of the roots of the adjacent teeth were classified, the height of the nasal floor was compared with the unaffected side, and the inter- and intraexaminer reproducibility of these evaluations was assessed. RESULTS: The grafted bone filled the defect in all three vertical measurement levels in 34 per cent. The labiopalatal thickness of the grafted bone was good in at least one-third of the root length in 66 per cent and fair in 34 per cent. Typically, the bone graft was deficient in the apical and palatal direction. Clear asymmetry in the nasal floor was found in 72 per cent. Kappa values indicated excellent agreement for all but one measured parameter. LIMITATIONS: This is a preliminary study involving only a limited number of study subjects. CONCLUSIONS: Our results showed mainly a good or fair treatment outcome. Deficiency of the bone graft was observed mostly in the apical and palatal areas of the defect. Asymmetry of the nasal floor was observed frequently. Careful insertion of the bone graft towards the palatal and apical direction of the cleft is recommended.

Embryology and epidemiology of microtia.

The auricle derives from six hillocks arising from the first and second branchial arches. Different hillocks give rise to different parts of the pinna. In the course of embryonic development, the auricle migrates postero-cranially as the mandible enlarges. Auricular malformations, such as microtia, are thought to be related to cell death of the first and second arch derivatives. The prevalence and characteristics of microtia vary in different populations. The prevalence ranges from 0.83 to 17.4 per 10,000. Microtia is more common in males, and right-sided dominance varies from 57 to 67%. The prevalence of aural atresia or stenosis varies from 55 to 93%. Microtia has been associated with numerous risk factors including race and gender. Genetic factors are likely to have an effect at least in some patients with microtia.

[Microtia--not just an ear problem]. / Mikrotia--ei ainoastaan korvaongelma.

Microtia can be defined as a malformation of the auricle with varying severity. In the majority of patients it is combined with atresia or stenosis of the external auditory canal. The prevalence of microtia in Finland is approx. 4 out of 10000. Associated anomalies should be actively seeked and excluded. Approximately 70% of the patients seem to be non-syndromatic. Patients with microtia are known to more than usual have other structural abnormalities. With the exception of the mildest cases, the development of facial structure and dental occlusion should be monitored in all microtia patients, attempting to actively exclude any associated diseases.

Craniofacial cephalometric morphology in 6-year-old children with isolated cleft lip, isolated submucous cleft palate, and combined cleft lip and submucous cleft palate.

One hundred and twenty-one cleft children (67 with isolated cleft lip (CL), 32 with isolated submucous cleft palate (SMCP), and 22 with combined cleft lip and submucous cleft palate (CL + SMCP)) were compared retrospectively from lateral cephalograms taken at a mean age of 6.2 years (range 5.5-7.9). None of the children had had their palates repaired or been operated on to treat velopharyngeal insufficiency (VPI). The children with CL + SMCP and CL had similar morphology. They had greater maxillary length, greater maxillary and mandibular prominence, less vertical growth pattern, less deep nasopharyngeal airways, and thinner upper lips than those with isolated SMCP. After 6 years of age one patient with CL + SMCP, none with CL, and 16 with SMCP needed operations for VPI. This small series suggests that children with CL + SMCP and SMCP have different morphology. Although CL + SMCP is a combination of two types of clefts, it seems to be associated with similar morphology to CL.

Craniofacial morphology in children with van der Woude syndrome and isolated cleft palate.

OBJECTIVE: To compare cephalometrically 6-year-old children with van der Woude syndrome and cleft palate (VWS) to children with isolated cleft palate alone (CP). DESIGN: A retrospective case-control study. PATIENTS AND SETTING: Forty-four children with VWS were compared to 73 children with CP using lateral cephalograms. The mean age of the children with VWS was 6.6 years (range = 5.9-8.2) and that of the children with CP, 6.2 years (range = 5.7-6.7). Palatal closure had been done at a mean age of 1.4 years (range = 0.8-2.2), mostly with the Veau-Wardill-Killner or the Cronin pushback surgical techniques. The data was collected over a 30-year period. MAIN OUTCOME MEASURE: Linear and angular measurements were obtained from lateral cephalograms. A Student's t-test was used in the statistical analysis. RESULTS: The craniofacial morphology in children with VWS and CP was similar, but those with VWS had slightly smaller diameters of the lower pharyngeal airway. The maxilla and mandible were well related to each other, although a little retrusive in relation to the cranial base. The soft tissue profile reflected the skeletal relationships, no significant protrusion of the lower lip was noted. CONCLUSIONS: Six-year-old children with VWS and CP have similar craniofacial morphology.

Early placement of ventilation tubes in cleft lip and palate patients: does palatal closure affect tube occlusion and short-term outcome?

OBJECTIVES: Otitis media with effusion is almost universal in children with cleft palate due to the poor function of the Eustachian tube. Our study investigates the functioning of ventilation tubes placed at the time of primary cleft surgery (4 months of age) and at the time of secondary surgery (12 months of age). We compared two different surgical protocols: (Leg A) closure of the lip and soft palate at the age of 3-4 months (primary surgery) and closure of the hard palate at the age of 12 months (secondary surgery), and (Leg C) closure of the lip at the age of 3-4 months (primary surgery) and closure of the hard and soft palate at the age of 12 months (secondary surgery). METHODS: A retrospective review of the medical records of 97 Finnish children with unilateral cleft lip and palate (UCLP) included in the Scandcleft study and randomized into two groups. RESULTS: The majority (63%) of cleft (lip and) palate children benefit from early placement of ventilation tubes, and this group is even larger with early closure of the soft palate (86%; p=0.02). Closure of the soft palate at four months of age also reduces the frequency of OME in ears with the tube extruded or occluded, thus indicating better function of the Eustachian tube (p=0.02). CONCLUSIONS: Early tympanostomy tube placement should be considered in children with cleft lip and palate, even prior to palatal closure.

A comparison of craniofacial cephalometric morphology and the later need for orthognathic surgery in 6-year-old cleft children.

OBJECTIVES: To compare 6-year-old children with unilateral cleft lip and palate (UCLP) and children with unilateral cleft lip and a separate cleft palate (CL+CP), cephalometrically and to evaluate the need for orthognathic surgery of these children in later life. MATERIALS AND METHODS: One hundred and seven cleft children (67 UCLP, 40 CL+CP) were compared retrospectively using lateral cephalograms taken at a mean age of 6.2 years (range 5.7-7.3). The need for maxillary or bimaxillary osteotomies in these patients was evaluated from the hospital records at the mean age of 18.1 years (range 15.6-20.5). RESULTS: The craniofacial morphology of children with UCLP and CL+CP was similar except for a more retrusive maxilla in children with UCLP. The frequency of orthognathic surgery was 40% (27/67) in the UCLP group and 18% (7/40) in the CL+CP group. The patients who needed orthognathic surgery (UCLP and CL+CP combined) had smaller mean values of ANB angle at the age of 6 years when compared to those who did not (1.8 versus 3.9°). None of the children whose ANB angle was greater than 4.5 needed orthognathic surgery whereas all the children whose ANB angle was less than -1 needed osteotomies. CONCLUSION: Six-year-old children with UCLP have more retruded maxillae and require orthognathic surgery later in life more often than the children with CL+CP.

Craniofacial and pharyngeal cephalometric morphology in seven-year-old boys with unoperated submucous cleft palate and without a cleft.

OBJECTIVE: To evaluate cephalometrically the craniofacial and pharyngeal morphology in 7-year-old boys with unoperated submucous cleft palate and to compare the findings with the morphology of 7-year-old boys without clefts. SETTING AND PATIENTS: Thirty-two boys with unoperated submucous cleft palate and 49 boys without a cleft were compared retrospectively from lateral cephalograms taken at the mean age of 7 years (range, 5.5 to 8.6 years). DESIGN: A retrospective case-control study. OUTCOME MEASURE: Linear and angular measurements were obtained from lateral cephalograms. A Student's t test was used in the statistical analysis. RESULTS: The maxilla of the boys with submucous cleft palate was shorter and slightly more retrusive in relation to the cranial base than that of boys without clefts. Also, the mandible of the boys with submucous cleft palate was smaller, with a steeper mandibular plane. The relationship between the jaws was similar in both groups; although, those without clefts showed higher values for soft tissue maxillary prominence. In the pharyngeal area, the boys with submucous cleft palate had larger nasopharyngeal depths, smaller hypopharyngeal depths, and shorter soft palates than the boys without a cleft. CONCLUSIONS: This small study suggests that the boys with unoperated submucous cleft palate have minor distinctive morphological features in the maxillary, mandibular, and pharyngeal areas.

Dental arches in submucous cleft palate: comparison of six-year-old boys with unoperated submucous cleft palate, with operated cleft of the soft palate, and without a cleft.

OBJECTIVE: To evaluate the dental arch dimensions in 6-year-old boys with unoperated submucous cleft palate (SMCP), and to compare their dental arch dimensions with those of boys without clefts and boys with clefts of the soft palate (CP). MATERIAL AND METHODS: The sizes of dental arches in 97 boys were compared retrospectively from dental casts taken at a mean age of 6.4 years (range 5.2-8.4). The material included 34 boys without clefts, 30 with unoperated SMCP, and 33 with CP. In children with CP, palatal closure had been done by the pushback technique at a mean age of 1.5 years. RESULTS: The dental arch dimensions of the boys with SMCP were similar to those of boys without clefts, except for a decreased maxillary arch length. There was no difference in maxillary arch length between the boys with SMCP and those with CP. The boys with CP had smaller maxillary and mandibular dental arch widths than the boys without clefts and with SMCP. There were no differences in the number of boys with permanent incisors between the three groups. CONCLUSION: Six-year-old boys with unoperated SMCP have similar dental arch dimensions as boys without clefts, except for a decreased maxillary arch length.

Dental arches in six-year-old children with operated and unoperated submucous cleft palate and isolated cleft palate.

The sizes of dental arches in 129 children with cleft palate were evaluated retrospectively from dental casts taken at the mean age 6.2 years (range 5.2-7.5). The material included 61 children with submucous cleft palate (SMCP) and 68 children with isolated cleft palate (ICP). Twenty of the children with SMCP were not operated on, while 41 had had surgical treatment, either palatal repair (n = 16, mean age at operation: 1.6 years, range 0.8-3.9) or pharyngeal flap (VPP) surgery (n = 25, mean age at operation: 4.5 years, range 2.6-6.2). In children with ICP, one-stage hard-palate and soft-palate closure had been done at the mean age of 1.5 years (range 1.0-2.1). Decreased maxillary intermolar widths were seen in children with SMCP after VPP, and especially after palatal repair. The children with ICP had the smallest maxillary dental arch widths. No significant differences were observed in the maxillary arch length or mandibular intermolar arch dimensions in children with SMCP or ICP. Surgery is associated with decreased maxillary intermolar arch widths in children with SMCP. Children with ICP had smaller maxillary dental arch widths than children SMCP.

Craniofacial cephalometric morphology in six-year-old girls with submucous cleft palate and isolated cleft palate.

Ninety-three girls with cleft palate (53 submucous cleft palate (SMCP) and 40 isolated cleft palate (ICP)), mean age 6.2 years (range 5.5-7.5), were compared retrospectively from lateral cephalograms. Forty-three patients with SMCP had had surgical treatment at the mean age of 3.4 years (range 0.9-6.8), 10 of the SMCP patients were unoperated. Twenty-six patients with ICP had clefts of the hard and soft palate, and 14 had clefts of the soft palate only. Palatal closure of ICP had been done at the mean age of 1.5 years (range 1.0-2.1). The skeletal craniofacial morphology was similar in SMCP and ICP. The maxilla and mandible were well related to each other but slightly retrusive in relation to the cranial base. The slight skeletal retrusion was significantly more masked by soft tissue in patients with SMCP. The patients with SMCP showed higher values for soft tissue maxillary and mandibular prominence.

Dental abnormalities in permanent dentition in children with submucous cleft palate.

Seventy-three children with submucous cleft palate (38 girls and 35 boys), mean age 8.2 years (range 7.7-9.5), were studied retrospectively from orthopantomograms. Dental abnormalities in permanent dentition were found in 26 patients (36%). Missing teeth, mainly lower 2nd premolars, upper lateral incisors, and upper 2nd premolars, were found in 12 patients (16%). Most of the patients had 1 or 2 missing teeth, 2 had 3 missing teeth. In 5 patients hypodontia was associated with another dental abnormality. Other dental abnormalities included peg-shaped lateral incisors in 7 patients (10%), ectopic eruption of upper 1st molars in 6 patients (8%), transposition of upper canines and 1st premolars in 3 patients (4%), supernumerary teeth in 2 patients (3%), and palatally displaced upper canines in 1 patient (1%). As children with submucous cleft palate have a tendency towards increased frequency of missing teeth and other dental abnormalities, the need for thorough clinical and radiological dental examination is emphasized.