Development Of A Health Related Quality Of Life Instrument For Children With Clefting PDF Download

Introduction: Approximately 100 children are born each year in New Zealand with an orofacial cleft (OFC). Children born with OFC require numerous surgical and non-surgical treatments from infancy to adulthood to re-establish as near to normal appearance and function as possible. In New Zealand this treatment is state funded and is provided by specialist clinicians working within five dedicated multidisciplinary cleft teams. To date there has not been any nationwide assessment of contemporary treatment outcomes. Objectives: To investigate: (1) The primary surgical outcomes of children aged 9-10 years who were born with either a complete unilateral (CUCLP) or complete bilateral cleft lip and palate (CBCLP) in New Zealand by assessing both dental arch relationships using a categorical and continuous scale and craniofacial morphology including soft tissue profile. (2) The bone infill outcomes following the secondary alveolar bone grafting for children with cleft of the alveolus using a categorical and continuous scale. (3) the dental health status at age 5 and 12 years and (4) the self-reported oral health related quality of life (OHRQoL) of children aged 10-11 years with OFC. Materials and Methods: Subjects born with an OFC from 1st January 2000 onwards who received their primary surgery in New Zealand were identified from the New Zealand Cleft Audit database. Records were collected up to 31st September 2017 to assess: (1) Dental arch relationships outcomes using 3D digital models from subjects with CULCP (n=100) were assessed using the GOSLON Yardstick and subjects with CBCLP (n=32) using the Bauru- BCLP (BCLP) Yardstick. Both the CULCP and CBCLP were also scored using a 100mm visual analogue scale (VAS). (2) Craniofacial morphology including soft tissue profile outcomes using lateral cephalograms from subjects with CULP (n=76) and subjects with CBCLP (n=23) using 13 hard tissue and 8 soft tissue landmarks. Records were collected up to December 2014 to assess bone infill outcomes following secondary alveolar grafts using intraoral radiographs (n=45) scored by the modified Kindelan index and a 100mm VAS. Dental records (n=554) were collected up to December 2016 for 478 children with OFC aged 5 (n=333) and 12 years (n=221) and were assessed for dental health status (caries experience). OHRQoL questionnaires were collected between January 2015 and December 2017 for children with OFC (n=185) aged 10-11 years using both 8-item and 16-item impact short form versions of the Child Perception Questionnaires (CPQ11-14-ISF and P-CPQ). Results: (1) Dental arch relationships of the CUCLP models showed 46% were poor/very poor, 28% fair and 26% were good/very good relationships. With the CBCLP models, 43.7% were poor/very poor, 28.1% fair and 25.0% had good/very good relationships. The mean CUCLP VAS score was 50.5mm (SD 19.9mm) while the mean CBCLP VAS score was 40.0mm (SD 22.0mm). These findings were similar to the less favourable outcomes reported by those centres which participated in large multicentre outcome comparative studies overseas. (2) The craniofacial morphology and soft tissue profile assessment found differences between CUCLP/CBCLP, gender and ethnicity. The CUCLP had greater midface and mandibular retrusion compared to CBCLP while Pacific and Māori had more retrusive midface profiles and Pacific more prominent mandibles. A sub-analysis of New Zealand European CUCLP results found they were most closely aligned to the centres from the Eurocleft and Americleft studies that had less favourable outcomes. (3) The amount of bone infill revealed only 69% of the grafts had good or satisfactory bone infill outcomes. The remaining 31% had unsatisfactory or complete failure. Those patients aged 10-11 years had significantly better outcomes compared to those aged 10 and 11 years at time of surgery. The contemporary secondary alveolar bone grafting bone infill outcomes in New Zealand are poor when referenced to recent international studies. (4) The dental health status of 5-year-old children with OFC revealed an increased dental caries experience to that reported by the Ministry of Health national data but similar for 12- year-olds. A higher (49.6%) caries prevalence (1 + dmft) and mean 5-year-old dmft (2.3 +/- 3.6) was found in relation to national data (prevalence 40.4% and mean dmft 1.8). The 12- year-old (37.6%) caries prevalence (1 + DMFT) and mean DMFT (0.8 +/- 1.4) was similar to national data (prevalence 37.3% and mean DMFT 0.9). Those children with caries (1+dmf/DMF) had means of 4.8 +/- 3.8 at age 5 and 2.1 +/- 1.4 and age 12 years. No differences were found by sex or cleft type. Increased caries experience was associated with children of Pacific and Māori ethnicity, and those not receiving community water fluoridation. (5) The OHRQoL of children with OFC found higher (worse) scores were recorded for children of Pacific ethnicity and children with more severe OFC (CLP). The study findings indicate children with OFC generally have worse OHRQoL than children without OFC. Conclusions: The investigation of some key outcomes of cleft care in New Zealand has revealed findings which do not compare favourably with other multicentre studies. These findings indicate an urgent review of the provisions of care for children with OFC in New Zealand, in particular the delivery of primary surgery and preventive dental health measures, is required to improve outcomes. The review should also emphasise a commitment to collect standardised records as part of each patient's standard of care to allow for ongoing monitoring of outcomes.