The wide variation in infant maxillary morphology and cleft size of children with unilateral cleft lip and palate (UCLP) and isolated cleft palate (CP) raise concerns about their possible influences on treatment outcome. The studies in this thesis aimed to investigate the relation between cleft size in infancy and crossbite at 5 years of age (Paper I); the impact of primary surgery on cleft size and maxillary arch dimensions from infancy to 5 years of age (Paper II); associations between cleft size, maxillary arch dimensions and facial growth in both UCLP and CP children (Paper III); and, to evaluate the relation between infant cleft size and nasal airway size and function in adults treated for UCLP (Paper IV). In homogenously treated groups of children with UCLP and CP, dental casts were used to measure cleft size and maxillary arch dimensions from infancy up to 5 years of age, and for crossbite recording at 5 years. Serial lateral cephalometric radiographs taken between 5 and 19 years of age in the same groups were used to study facial growth. Nasal airway size and function were evaluated by acoustic rhinometry, rhinomanometry, peak nasal inspiratory flow and odour test in a group of adults treated for UCLP. The main findings were: crossbite was a frequent malocclusion at 5 years of age in children with UCLP and large cleft widths at the level of the cuspid points in infancy were associated with less anterior and posterior crossbite in this group (Paper I). Cleft widths decreased after lip closure and/or soft palate closure in both UCLP and CP children. Initially, UCLP children had wider maxillary arch dimensions, but after hard palate closure, the transverse growth was reduced, and at 5 years, they had smaller maxillary arch widths than CP children had (Paper II). Maxillary arch depths and cleft widths in infancy were correlated with maxillary protrusion and sagittal jaw relationships in both UCLP and CP children (Paper III), but cleft width in infancy was not correlated with nasal airway size and function in adults treated for UCLP (Paper IV).

Mémoire numérisé par la Division de la gestion de documents et des archives de l'Université de Montréal.

Mémoire numérisé par la Division de la gestion de documents et des archives de l'Université de Montréal

Background. The treatment of patients born with cleft lip and palate has been gradually modified over the years as the surgical procedures have developed and improved. Multidisciplinary team care has evolved and provided improved care with enhanced results. Clefts in the alveolus can be reconstructed by alveolar bone grafting or by periosteoplasty. The main goal is to repair and close the alveolar cleft and create a continuous alveolar processes so that the teeth can erupt. Aims. This thesis has several aims: to investigate the impact of dental status and initial cleft width on the outcome of Secondary alveolar bone grafting (SABG) in patients born with unilateral cleft lip and palate (UCLP) at the 10-year follow-up (Studies I and II); to compare the outcomes of primary periosteoplasty (PPP) with those of SABG in patients born with unilateral cleft lip and alveolus (CLA) (Study III); to evaluate clinical and radiographic conditions and identify factors important for the final treatment outcomes after SABG ( Study IV); to evaluate two radiographic methods, i.e. occlusal radiographs and cone beam tomography (CBCT)) for assessing alveolar bone height ( study IV). Results. In UCLP patients, SABG achieved excellent results in terms of bone height; tended to reduce with time, correlated with dental status and dental restoration factors. Occlusal radiographs correspond well with the CBCT, for evaluating alveolar bone height in cleft area. The width of the initial cleft does not seem to affect the success of SABG. Finally, patients with CLA treated with PPP at the time of lip repair have inferior bone formation outcomes in the cleft area compared with patients treated with SABG at the time of mixed dentition. Conclusion.  Poor dental status and malpositioning negatively affect the long-term survival of bone in the alveolar cleft. The initial cleft width affects certain dental status factors. In adults with UCLP, the alveolar bone height in the cleft was correlated to the presence of gingival inflammation and restorations at 20 years follow-up. Specially designed maintenance therapy is beneficial, after complex dental restorations in the cleft area. SABG is preferred to PPP for the reconstruction of alveolar clefts.

O objetivo deste trabalho foi avaliar a morfologia periodontal vestibular e lingual do canino permanente movimentado para a região enxertada em fissuras alveolares unilaterais com comprometimento do rebordo alveolar por meio de mensurações realizadas em imagens de exames de tomografia computadorizada cone-beam (TCCB). Esses valores foram comparados com o canino e incisivo lateral presentes no lado não comprometido pela fissura. A amostra consistiu de exames de TCCB de 30 pacientes com fissura unilateral em fase de contenção pós-tratamento ortodôntico. Foram realizados exames de TCCB com protocolo de FOV de 6cm, voxel de 0,25mm por 26,9 segundos. Os exames foram avaliados por apenas um ortodontista duas vezes em um intervalo de 30 dias para calcular o erro do método. A padronização da posição da cabeça foi determinada com o plano palatino paralelo ao solo. Foram realizados cortes axiais na altura da trifurcação do dente 16 para mensurar a espessura da tábua óssea vestibular e lingual do canino do lado da fissura (CF), canino do lado sem fissura (CsF) e incisivo lateral do lado sem fissura (IL). Para a avaliação do nível da crista óssea vestibular e lingual, foram realizados cortes parassagitais passando pelo centro da coroa de cada dente avaliado: CF, CsF e IL. Calculou-se a distância da junção cemento-esmalte (JCE) à crista óssea alveolar vestibular e lingual. Para a comparação dos valores do CF com o CsF e IL, foram utilizados o teste t e o teste de Wilcoxon a depender da distribuição de normalidade. A espessura da tábua óssea vestibular do CF se mostrou significantemente menos espessa do que a do CsF (p=0,002) e do que a do IL (p=0,001). Além disso, a distância da JCE à crista óssea se apresentou estatisticamente maior para o CF (p<0,001). A movimentação do canino superior permanente para a área de fissura alveolar enxertada consiste em uma opção terapêutica viável. No entanto, a condução do tratamento ortodôntico demanda cautela, incluindo uma movimentação lenta e a verificação da qualidade da mucosa ceratinizada. / The purpose of this research was to assess the buccal and lingual periodontal morphology of the permanent canine moved to the grafted region in unilateral alveolar clefts with commitment of the alveolar bone by measuring images of conebeam computed tomography (CBCT) exams. These values were compared to the canine and lateral incisor present in the non-clefted side. The sample comprised CBCT exams of 30 patients with unilateral cleft lip and/or palate during retention period after orthodontic treatment. The protocol used for the CBCT exam was FOV of 6cm, voxel of 0,25mm for 26.9 seconds. The exams were assessed by one orthodontist two times with an interval of 30 days to assess intraoperator reliability. The standard position of the head was determined with the palatal plane parallel to the horizontal plane. Axial slices of the trifurcation of the right permanent molar were obtained to assess the buccal and lingual alveolar bone thickness of the canine in the clefted side (CF), the canine in the non-clefted side (CsF) and the lateral incisor in the non-clefted side (IL). To assess the buccal and lingual bone crest height, parassagital slices were obtained passing through the center of the crown of each evaluated teeth. It was calculated the distance form the cemento-enamel junction (CEJ) to the buccal and lingual alveolar bone crest. The comparison among the CF values with the CsF and IL was performed with the t test or the Wilcoxon test, depending on the distribution of normality. The buccal alveolar bone thickness of the CF is statistically thinner than the buccal alveolar bone thickness of the CsF (p=0,002) and of the IL (p=0,001). Other than that, the distance between the CEJ and the alveolar crest was significant higher for the CF (p<0,001). The movement of the upper permanent canine to the grafted alveolar consists in a viable therapeutic option. However, the orthodontic treatment demands caution, including a very slow movementation and the verification of the quality of the keratinized mucosa.

O objetivo deste trabalho foi avaliar a morfologia periodontal vestibular e lingual do canino permanente movimentado para a região enxertada em fissuras alveolares unilaterais com comprometimento do rebordo alveolar por meio de mensurações realizadas em imagens de exames de tomografia computadorizada cone-beam (TCCB). Esses valores foram comparados com o canino e incisivo lateral presentes no lado não comprometido pela fissura. A amostra consistiu de exames de TCCB de 30 pacientes com fissura unilateral em fase de contenção pós-tratamento ortodôntico. Foram realizados exames de TCCB com protocolo de FOV de 6cm, voxel de 0,25mm por 26,9 segundos. Os exames foram avaliados por apenas um ortodontista duas vezes em um intervalo de 30 dias para calcular o erro do método. A padronização da posição da cabeça foi determinada com o plano palatino paralelo ao solo. Foram realizados cortes axiais na altura da trifurcação do dente 16 para mensurar a espessura da tábua óssea vestibular e lingual do canino do lado da fissura (CF), canino do lado sem fissura (CsF) e incisivo lateral do lado sem fissura (IL). Para a avaliação do nível da crista óssea vestibular e lingual, foram realizados cortes parassagitais passando pelo centro da coroa de cada dente avaliado: CF, CsF e IL. Calculou-se a distância da junção cemento-esmalte (JCE) à crista óssea alveolar vestibular e lingual. Para a comparação dos valores do CF com o CsF e IL, foram utilizados o teste t e o teste de Wilcoxon a depender da distribuição de normalidade. A espessura da tábua óssea vestibular do CF se mostrou significantemente menos espessa do que a do CsF (p=0,002) e do que a do IL (p=0,001). Além disso, a distância da JCE à crista óssea se apresentou estatisticamente maior para o CF (p<0,001). A movimentação do canino superior permanente para a área de fissura alveolar enxertada consiste em uma opção terapêutica viável. No entanto, a condução do tratamento ortodôntico demanda cautela, incluindo uma movimentação lenta e a verificação da qualidade da mucosa ceratinizada. / The purpose of this research was to assess the buccal and lingual periodontal morphology of the permanent canine moved to the grafted region in unilateral alveolar clefts with commitment of the alveolar bone by measuring images of conebeam computed tomography (CBCT) exams. These values were compared to the canine and lateral incisor present in the non-clefted side. The sample comprised CBCT exams of 30 patients with unilateral cleft lip and/or palate during retention period after orthodontic treatment. The protocol used for the CBCT exam was FOV of 6cm, voxel of 0,25mm for 26.9 seconds. The exams were assessed by one orthodontist two times with an interval of 30 days to assess intraoperator reliability. The standard position of the head was determined with the palatal plane parallel to the horizontal plane. Axial slices of the trifurcation of the right permanent molar were obtained to assess the buccal and lingual alveolar bone thickness of the canine in the clefted side (CF), the canine in the non-clefted side (CsF) and the lateral incisor in the non-clefted side (IL). To assess the buccal and lingual bone crest height, parassagital slices were obtained passing through the center of the crown of each evaluated teeth. It was calculated the distance form the cemento-enamel junction (CEJ) to the buccal and lingual alveolar bone crest. The comparison among the CF values with the CsF and IL was performed with the t test or the Wilcoxon test, depending on the distribution of normality. The buccal alveolar bone thickness of the CF is statistically thinner than the buccal alveolar bone thickness of the CsF (p=0,002) and of the IL (p=0,001). Other than that, the distance between the CEJ and the alveolar crest was significant higher for the CF (p<0,001). The movement of the upper permanent canine to the grafted alveolar consists in a viable therapeutic option. However, the orthodontic treatment demands caution, including a very slow movementation and the verification of the quality of the keratinized mucosa.

Unilateral cleft lip and palate (UCLP) is a craniofacial malformation with functional and aesthetical impact on the face and the upper airways. The aims of the current thesis were to evaluate Quality of life (QoL) in adults treated for UCLP  (I), to objectively evaluate nasal form and function and to search for possible differences in residual nasal deformity and impairment of function between patients operated according to one-stage and two-stage palate closure (II) as well as to evaluate the relationship between professional and lay rating and patient satisfaction with nasolabial appearance (III) and to identify factors associated with lower levels of QoL and less satisfaction with nasal appearance among adults treated for UCLP (IV). Analyses of data from a homogenous population of UCLP patients treated at Uppsala University Hospital form the basis of this thesis. The mean follow-up time after primary surgery was 35 years (20-47 years) and participation rate was 79% (n=86). An age and gender matched control group of 68 people without clefts were evaluated according to the same protocol. The evaluation protocol included the Short Form 36 questionnaire (SF-36), rhinomanometry, acoustic rhinometry, odor test, peak nasal inspiratory flow test and photographies of faces. For the SF-36 data, age- and gender-matched norm data of 1385 people from the Swedish population were used. Unilateral cleft lip and palate affected QoL differently depending on gender and age of the patient. Younger patients were affected more negatively than older patients in several subscales. However, except for lower values in the Mental Health subscale, QoL was similar among UCLP patients and norm data. Objectively measured nasal function was extensively affected among adults treated for UCLP. No difference in impairment of nasal function was found between one-stage and two-stage palate closure protocols on the cleft side. Judgment of nasolabial appearance differed between professionals, lay people and patients. Large infant cleft width was associated with less satisfaction with nasal appearance and male gender was associated with lower levels of mental QoL. Correlation between high nasal breathing resistance and low levels of physical QoL was found. In conclusion, this thesis provides a platform for future research for optimal evaluation of cleft treatment outcome.

Läpp-, käk och gomspalt (LKG) kan medföra svårigheter med tal, artikulation och röst. Enligt tidigare forskning har vuxna individer behandlade för LKG liknande förekomst av röstavvikelser som kontrollpersoner utan spalt. Syftet med denna studie var att undersöka grad av röstavvikelser hos vuxna patienter behandlade för enkelsidig LKG och kartlägga eventuella samband mellan röstavvikelser och kirurgiska metoder för gomslutning, kön och operation med eller utan svalglambå samt jämföra röstresultat med data från individer utan LKG. Sjuttio patienter behandlade för enkelsidig genomgående LKG deltog i studien, varav 45 patienter hade opererats med gomslutning i en seans och 25 patienter hade opererats i två seanser. Elva av patienterna i patientgruppen hade genomgått svalglambåoperation. I studien ingick även en åldersmatchad kontrollgrupp med 63 individer utan LKG. Röstinspelningar utvärderades med perceptuell röstbedömning med Stockholm Voice Evaluation Approach (SVEA) av två erfarna logopeder. Skattning skedde individuellt av randomiserade och blindade inspelningar. Inter- och intrabedömarreliabilitet beräknades. Grad av avvikelse i röstkvalité var ca. 5/100 i patientgrupp och kontrollgrupp vilket är lägre jämfört med tidigare studier. “Knarr” fanns lägre i patientgruppen i jämförelse med kontrollgrupp. Skattningar av övriga röstparametrar skiljde sig inte mellan patientgrupp och kontrollgrupp. Patienter opererade med gomslutning i en eller två seanser hade inga skillnader i röstparametrar. Enstaka små skillnader fanns mellan könen. Ingen skillnad fanns mellan patienter som genomgått svalglambåoperation och de som inte gjort det. / Cleft lip and palate (CLP) may cause impairments in speech, articulation and voice. Treatment of patients with CLP include different types of palatoplasty. Recent studies have found a comparable prevalence rate of dysphonia in adult patients treated for CLP and controls without cleft. The purpose of this study was to investigate the rate of dysphonic deviation in adult patients treated for unilateral CLP and determine, if any, the relationship between voice quality outcome and differences in surgical procedures, gender, palatoplasty with or without pharyngeal flap surgery and compare the voice characteristics with age related normative data. Seventy patients treated for unilateral CLP with one- or two-stage palatoplasty participated in this study, of which 45 patientshad undergone one-stage palatoplasty and 25 patients two-stage palatoplasty. Eleven ofthe patients had also undergone pharyngeal flap surgery. Data from an age matched non-cleft group consisted of 63 participants. Sound recordings of participants' voices were perceptually assessed by two speech-language pathologists. Individual ratings of randomized and blinded sound recordings were performed. Inter- and intra-raterreliability was calculated. The study showed a lower degree of dysphonia in patients treated for unilateral CLP than earlier studies. Among patients treated for unilateral CLP and the non-cleft group, a degree of approximately 5/100 voice quality deviation was found. ”Vocal fry” was found significantly lower in patients treated for CLP than the non-cleft group. Measures of voice parameters did not differ significantly between patients and non-cleft groups. Patients that had undergone palatoplasty in one or two stages showed no significant differences in voice parameters. Minor statistical significance was found in a few of the measured voice parameters as related to gender. Between patients that had undergone pharyngeal flap surgery and those that had not no differences were found.

Doctor Educationis / This study was a cephalometric and dental investigation of the treatment outcomes of UCLP children treated at the Red Cross Children's Hospital (RCCH) with respect to craniofacial morphology and dental arch relationship. The quality of the outcome for the RCCH group was compared with the outcomes reported for the Six-Centre International Study (Melsted et al., 1992; Mars et al., 1992). The sample consisted of 20 (11 females, 9 males) consecutively treated UCLP children who had cephalometric and dental records taken between the ages of8 to 11years (mean 10.13 ± 1.2 years). The cephalometric analysis described by Melsted et al. (1992) was used to evaluate the skeletal and soft tissue morphology. The quality of the dental arch relationship was measured according to the Gosion Yardstick (Mars et al., 1987). The treatment outcome of children treated at the RCCH was evaluated with respect to craniofacial form and dental arch relationship. When comparing the mean cephalometric skeletal parameters of the RCCH to the six centres in the Eurocleft study, a significant difference was found between the RCCH group and centre D for most of the variables. A significant increase in the upper incisor inclination and maxillary inclination was found in the RCCH patients compared to the European centres. The difference in the soft tissue parameters was limited to the relative protrusion of the nose and the sagittal soft tissue variable sss-ns-pgs. The analysis of the Goslon scores showed a significant difference between the RCCH group and centres C, D, and F. According to the Goslon score, 85% of the RCCH patients had good to satisfactory dental arch relationship, which was comparable to that recorded for centres A(92%), B(89%) and C(94). In conclusion, the results of the cephalometric analysis and the Goslon Yardstick showed a significant difference between the RCCH group and centre D. The GosIon score indicated good quality of the dental arch relationship, which faired favourably with the better centres in the Six Centre Study.