Cochlear implants and deafness: a global case study to increase policy awareness and action on an under-resourced health issue.

There has been a major growth in global health networks in recent decades to address health issues including tobacco use, alcohol abuse, and maternal mortality. Most conditions that incur high costs have provoked networks of advocates working to mitigate the impact, increase investment in research, and establish campaigns. Global health networks often work simultaneously across policy, knowledge creation, and advocacy. Until recently there has been limited activity in global health networks and advocacy addressing hearing loss and deafness which has contributed to its relatively low visibility with policymakers compared to other health conditions. This discussion paper reports on a global consultation that explored the views of advocacy groups and individuals on advocacy for the management of hearing loss, and cochlear implantation (CI). It focussed on stakeholders' views of current advocacy endeavours, opportunities and barriers, and the possible development of a global advocacy network to improve access to cochlear implantation and the supporting services. The subsequent development of a global health network, the Cochlear Implant International Community of Action (CIICA) is discussed and the conditions necessary for the successful development of health networks are explored. This paper will be of interest to those wishing to understand the factors influencing the development of health networks and advocacy.

Hypnotherapy for irritable bowel syndrome: an audit of one thousand adult patients.

BACKGROUND: Gut-focused hypnotherapy improves the symptoms of irritable bowel syndrome (IBS) with benefits being sustained for many years. Despite this, the technique has not been widely adopted by healthcare systems, possibly due to relatively small numbers in published studies and uncertainty about how it should be provided. AIM: To review the effect of hypnotherapy in a large cohort of refractory IBS patients. METHODS: One thousand IBS patients fulfilling Rome II criteria, mean age 51.6 years (range 17-91 years), 80% female, receiving 12 sessions of hypnotherapy over 3 months, were studied. The primary outcome was a 50 point reduction in the IBS Symptom Severity Score. The fall in scores for Noncolonic Symptoms, Quality of Life and Anxiety or Depression, were secondary outcomes. The Federal Drug Administration's recommended outcome of a 30% or more reduction in abdominal pain was also recorded. RESULTS: Overall, 76% met the primary outcome which was higher in females (females: 80%, males: 62%, P < 0.001) and those with anxiety (anxious: 79%, non-anxious: 71%, P = 0.010). The mean reduction in other scores was: IBS Symptom Severity Score, 129 points (P < 0.001), Noncolonic Symptom Score, 65 (P < 0.001) and Quality of Life Score, 66 (P < 0.001). Sixty-seven per cent reported a 30% or more reduction in abdominal pain scores. Pain days fell from 18 to 9 per month. Patients with anxiety and depression fell from 63% to 34% and 25% to 12% respectively (P < 0.001). Outcome was unaffected by bowel habit subtype. CONCLUSION: These results provide further evidence that gut-focused hypnotherapy is an effective intervention for refractory IBS.

Parents' views on the quality of life of their children 2-3 years after cochlear implantation.

OBJECTIVE: Cochlear implants for children are known to have impact on the lives of recipients and their families in a variety of ways. To obtain a clearer picture of these benefits, we explored the quality of life of 36 Finnish children and their families 2-3 years after unilateral cochlear implantation. METHODS: The studied children were, on average 5 years old, and had received their implant at the median age of 2 years:5 months (range 1:6 to 12:3). Most (67%) of the children used speech, eight (22%) used speech and signs, and four (11%) used sign language as their main communication mode. A third of the children had concomitant problems in addition to their profound hearing impairment. A validated closed-set questionnaire "Children with cochlear implants: parental perspectives" (available, e.g., at http://www.earfoundation.org.uk/research/questionnaires.html) was used to find out parents' views and experiences on implantation and explore life after it. RESULTS: Parents were most satisfied with improved/expanded social relations, improved communication (the development of spoken language), general functioning with the help of hearing and improved self-reliance of the child. Benefit of cochlear implantation was also detected with the Categories of Auditory Performance (CAP), which was concordant with views of the parents on the progress of their child in the areas of communication and education. When deciding on implantation, the parents particularly expected auditory information to enhance their child's safety in traffic, joining socially the hearing world, and better employment prospects as adults. Concerning the process of implantation, parents especially valued the know-how and fluent services of the implant centre, positive attitude within the family and information received from other families during the time they were considering the implant decision. Parents also found it important that they have the possibility to influence the communication mode that is used in their child's educational setting. CONCLUSIONS: Parents report that cochlear implants affect their children in a wide variety of ways that cannot be summarized by a single scale. A broader descriptive framework is required to capture their experiences adequately.

Estimating time and out-of-pocket costs incurred by families attending a pediatric cochlear implant programme.

OBJECTIVES: The objectives of this study are to estimate time and out-of-pocket costs incurred by families attending a pediatric cochlear implant programme. In addition, qualitative data examine the intangible costs faced by families. METHODS: Data was collected during semi-structured face-to-face interviews with parents of children with a cochlear implant attending a clinic visit at Nottingham Cochlear Implantation Programme (NPCIP), UK. Information supplied included socio-demographic characteristics, mode of travel, out-of-pocket expenses, time foregone, and details of companions. Quantitative data was stored and analysed in SPSS version 11.5. RESULTS: Two hundred and sixteen face-to-face interviews were conducted with parents of children implanted for between 1 month and 13 years. Time and out-of-pocket costs were significantly higher for those in the first 2 years of the programme, mean UK pound 3090 per annum compared to UK pound 2159 per annum for those in years >2-5 and UK pound 1815 per annum for those in years >5 (P<0.001). The biggest component of this was time costs, although the sensitivity analysis revealed that these were also most variable depending on the methods used to estimate them. The largest out-of-pocket cost incurred by families was travel costs which accounted for 44%, although 16% of families received some financial help with travel costs. The qualitative findings are consistent with these findings, also showing that over time the financial and intangible costs incurred as a result of cochlear implantation decline. CONCLUSIONS: This study is the first to obtain primary data on the time and out-of-pocket costs incurred by families attending a pediatric cochlear implant programme in the UK. It finds that these costs are greatest for those families in the first 2 years of the programme and/or who live furthest from the programme.

Innovation and funding specialist services. Cochlear implantation.

This paper traces the innovative development of the Nottingham Cochlear Implant Programmes. The paediatric programme was the first to be established in the UK in 1989 and remains the largest programme in the UK today, whilst the adult programme developed later, in 1994. The first section of the paper describes trends in service development whilst the second section makes detailed reference to the history of funding arrangements which enabled the programme to become established. The third part of the paper examines the (de)merits of locality purchasing versus centralised purchasing for specialist services, using cochlear implantation as way of illustration. The paper aims to provide an informative history of the development of the service in Nottingham and from this background create debate as to the most appropriate future funding mechanism for cochlear implantation in particular and specialist services in general.

Willingness-to-pay for pediatric cochlear implantation.

OBJECTIVE: This paper presents the results of the first willingness-to-pay (WTP) study to be undertaken on cochlear implantation. It aims to measure the values parents place on the UK having a pediatric cochlear implantation (PCI) programme. METHODS: Face-to-face semi-structured interviews were conducted with parents of children from the Nottingham Pediatric Cochlear Implant programme, whom had been implanted for a period ranging from 1 month to 13 years. Parents willingness-to-pay for the UK to have a pediatric cochlear implantation programme were elicited using a bidding process question format and via a discrete choice question. To see if income was a significant determinant of willingness-to-pay an analysis of variance (ANOVA) was undertaken in the statistical package SPSS version 10. RESULTS: Two hundred and sixteen parents were interviewed over the period July 2001-August 2002, representing over 130h of interviewing. The mean and median willingness-to-pay values elicited were UK pound 127 and 50 per month, respectively (UK pound 2001/2002). Willingness-to-pay was positively related to income (P<0.020). When the income constraint was removed, 99% of parents choose the implant over having the money the implant would cost to spend in some other way to benefit their child. CONCLUSIONS: Parents of implanted children were willing to pay substantial monthly amounts for pediatric cochlear implantation. Most parents saw no alternative to pediatric cochlear implantation that could improve their child's quality of life to the same extent. Willingness-to-pay was sensitive to income as expected suggesting that the values elicited are both valid and influenced by a respondent's budget constraint.

Evidence of improving cost-effectiveness of pediatric cochlear implantation.

OBJECTIVES: To examine the cost-effectiveness of pediatric cochlear implantation over time. METHODS: A prospective study based on ninety-eight children implanted between 1989 and 1996 at Nottingham's Paediatric Cochlear Implantation Programme, UK. The influence of outcomes and other variables on total costs was examined using multivariate regression analysis. RESULTS: Having controlled for potential confounding variables, total cost was negatively related to year of implant and positively related to the number of hours of rehabilitation (p=.000). CONCLUSIONS: Having controlled for outcomes (Categories of Auditory Performance and Speech Intelligibility Rating), the cost-effectiveness improved over time. This finding may be due to a learning curve and have policy implications.

Variations in gains in auditory performance from pediatric cochlear implantation.

OBJECTIVE: To investigate variations in gains in auditory performance in children with cochlear implants. STUDY DESIGN: The auditory performance of 98 children was measured with the Category of Auditory Performance survey instrument. All data were collected prospectively. Variables used to explain gain in Category of Auditory Performance were age at implantation, sex, the duration of "untreated" deafness, the year in which implantation occurred, health care inputs, and cause of hearing impairment. The data were analyzed by ordered probabilistic regression analysis. RESULTS: Gain in Category of Auditory Performance was observed to be negatively related to age at implantation, the year in which implantation took place, and the number of medical consultations the child received. None of the other variables were significant determinants of gain in Category of Auditory Performance. CONCLUSION: This study demonstrates the value of examining incremental gain from implantation rather than simply examining outcome levels. It was found that pediatric implantation is subject to diminishing returns. This suggests that further relaxation of entry criteria to implant programs should be undertaken only after careful consideration. The study also confirmed that age at implantation is an important determinant of outcomes. Greater gain in Category of Auditory Performance was experienced by those who underwent implantation at a younger age. This finding has implications for screening, as well as for purchasers and providers of implant services, highlighting the importance of responding in a timely fashion to identified need.

Functionally equivalent ages and hearing levels of children with cochlear implants measured with pre-recorded stimuli.

Three groups of children undertook an interactive computer-based closed-set test of the ability to identify pre-recorded spoken words presented acoustically. The test was completed by 31/39 children with profound hearing loss who had used the Nucleus Spectra-22 cochlear-implant system for at least one year (Group A); by 30 children with normal hearing (Group B); and by 22 children with severe-profound hearing loss who used acoustic hearing aids (Group C). Among the implanted children, those who were younger when implanted and who had used their devices for longer produced higher scores (multiple-r = 0.68). Logistic regression functions were fitted to the data from Group B to describe the relationship between performance and age, and to the data from Group C to describe the relationships between performance and average hearing level (AHL) and aided threshold. By use of the regression equations, the performance of each implanted child was converted into a functionally equivalent (FE) age, an FE AHL and an FE aided threshold. Despite high variability leading to wide confidence intervals, these transformations showed that: (1) mean FE age (3.4 years) lagged mean chronological age (7.4 years), but some implanted children performed within the range expected for children with normal hearing of the same age; (2) mean FE AHL was 94 dB compared with a mean pre-implant AHL of 117 dB; (3) mean FE aided threshold was 45 dB(A) compared with a mean pre-implant aided threshold of 99 dB(A). These results confirm that implantation of appropriate candidates leads to functionally better hearing than would be expected with acoustic hearing aids. The results also demonstrate that many implanted children can participate in interactive tests with pre-recorded speech, thus providing robust data for comparison with future performance.

Pediatric cochlear implantation: the parents' perspective.

OBJECTIVE: To analyze parental views on cochlear implantation, before and in the years following implantation, to determine whether the results from the intervention met their expectations. DESIGN: Prospective longitudinal study to assess parental perspectives of an unselected group of children with cochlear implantation. SETTING: Tertiary referral pediatric cochlear implant center in the United Kingdom. SUBJECTS: Forty-three parents of children with cochlear implantation. INTERVENTION: A specifically designed questionnaire was administered to assess preimplant expectations and observed changes and concerns at 1, 2, and 3 years following implantation. Three key domains were evaluated: (1) communication with others, (2) listening to speech without lipreading, and (3) the development of speech and language. RESULTS: Preoperative expectations were met or surpassed at each of the follow-up intervals. In the area of communication, 35 (81%) parents expected a definite improvement preoperatively, and 3 years following implantation, 42 (98%) actually saw such an improvement. The respective numbers in the area of listening to speech were 15 (35%) and 38 (88%), and for speech development, 37 (86%) and 37 (86%). Speech development was the major area of concern at all intervals. CONCLUSION: This study demonstrates the ability of cochlear implantation to meet or surpass parental expectations in 3 important outcome domains: communication, listening to speech, and the development of speech and language.

Use of the telephone in prelingually deaf children with a multichannel cochlear implant.

OBJECTIVE: To assess progress in the use of the telephone in a group of prelingually deaf children after cochlear implantation. SETTING: Tertiary referral pediatric cochlear implant center in the U. K. STUDY DESIGN: A prospective study was undertaken on a consecutive group of 150 congenital and prelingually deaf children up to 5 years after implantation. The study group was confined to prelingually deaf children aged less than 7 years at the time of implantation. No child was lost to follow-up, and there were no exclusions from the study. At the time of the study, 129 children had reached the 1-year stage, and 91, 68, 40, and 23 had reached the 2-, 3-, 4-, and 5-year intervals, respectively. METHODS: A specifically designed profile was used to assess the telephone use of the implanted children. Regression analysis was used to assess the correlation between the results of the telephone profile with the outcomes of the Iowa sentence test and connected discourse tracking. RESULTS: After implantation, prelingually deaf children showed significant progress in telephone use over time, not reaching a plateau at the 5-year interval (median score 27 with maximum score available 34). The results of the telephone profile showed significant correlations with the other tests of speech perception (correlation coefficients from 0.47 to 0.79, all statistically significant p < 0.0001). CONCLUSION: The telephone profile provided a useful method of monitoring children's telephone use. The profile was easily administered, and it was sensitive in assessing the progress of prelingually deaf children with cochlear implants. Outcomes from the profile were highly correlated with results from other widely used closed- and open-set tests.

Indirect costs, cost-utility variations and the funding of paediatric cochlear implantation.

OBJECTIVE: To demonstrate the existence of variations in cost-utility associated with indirect costs in paediatric cochlear implantation; to illustrate the implications of this for purchasing decisions and; to posit a potential solution to anomalies in purchasing that may otherwise result when services are publicly funded. METHODS: Data was taken from published sources on the cost of implantation, outcomes measured in terms of quality adjusted life years (QALY) and in savings in education costs associated with paediatric cochlear implantation. Cost-utility ratios across education authorities were calculated using a single centre's implantation costs. RESULTS: Variations in savings across education authorities show that the cost per QALY gain associated with paediatric cochlear implantation can vary between approximately $12,000 and $18,000 assuming an exchange rate of $1.45 = 1 pound sterling for the same implant centre. These variations have the potential to produce situations in which less efficient implant centres are preferred by purchasers over more efficient ones or in which candidates with poorer outcomes are selected for funding over candidates with superior outcomes. It is important that savings associated with education be taken into consideration in evaluations intended to inform purchasing decisions regarding implantation. Equally it is important that potentially anomalous decisions be avoided if evaluations are to remain credible. It is argued that this may be achievable if public funding for implantation is determined at a national level and ring-fenced i.e. devoted exclusively to use in cochlear implantation.

Determinants of speech perception in children after cochlear implantation.

BACKGROUND: Cochlear implants provide access to the speech signal in those profoundly deaf children who derive no material benefit from acoustic hearing aids. Speech perception after implantation can vary widely--we have analysed the contribution of several factors. METHODS: We examined 40 children with mean age at implantation of 52 months who were either born deaf or became deaf before 3 years. All patients received the same multichannel implant system and were followed up for 5 years. We used connected discourse tracking (CDT) as the measure of speech perception. The effect of five potential predictors (age at implantation, number of inserted electrodes, origin of deafness, mode of communication, and socioeconomic group) on speech perception was analysed. FINDINGS: The mean number of words per minute perceived increased from 0 before implantation to 44.8 (SD 24.3) 5 years after implantation. Repeated-measures ANOVA showed that children significantly progressed over time (p=0.001). Age at implantation was a significant covariate (p=0.01) and mode of communication was a significant between-individuals factor (p=0.04). INTERPRETATION: Young age at intervention and oral communication mode are the most important known determinants of later speech perception in young children after cochlear implantation. Early identification of candidate children necessitates implementation of universal neonatal screening programmes for hearing impairment.

Approach to communication, speech perception and intelligibility after paediatric cochlear implantation.

The aim of this study was to explore the relationship between approach to communication, speech perception and speech intelligibility after cochlear implantation of young children with profound early deafness. A prospective speech perception and speech intelligibility assessment was undertaken on a consecutive group of implanted children. There were 46 children at the three-year, 26 at the four-year and 20 at the five-year intervals. All had been born deaf or deafened before the age of three and received cochlear implants before the age of seven. Their speech perception ability and the intelligibility of their speech were measured before cochlear implantation and annually thereafter. The children's communication had been classified by their teachers of the deaf at each interval into one of two categories: those using an oral approach and those using a signing approach. Results revealed that at all intervals, those children classified as using oral communication significantly exceeded those using signed communication on measures of speech perception and intelligibility (p<0.05). When those children who had changed from signed to oral communication were compared at the three-year interval with those who used oral communication throughout, there was no significant difference in their results. However, it remains to be explored whether children use oral communication after cochlear implantation because they are doing well, or whether they do well because they are using oral communication.

A cost-utility analysis of pediatric cochlear implantation.

OBJECTIVE/HYPOTHESIS: The aim of this study was to explore the cost-utility of pediatric cochlear implantation, incorporating savings associated with education into the analysis. METHODS: The costs of pediatric cochlear implantation were based on the full costs levied to purchasers, inclusive of complications and maintenance, by a large pediatric cochlear implant program in the United Kingdom. After implantation, profoundly hearing-impaired children have been found to develop hearing threshold levels equivalent to severely hearing-impaired children who wear hearing aids. An independent study calculated the educational costs for severely hearing-impaired and profoundly hearing-impaired children. From this study, savings in educational costs that would result from enabling the profoundly hearing-impaired to function as severely hearing-impaired were determined. Cost-utility was established conservatively by applying to children the known gains in utility reported by adults with cochlear implants. RESULTS: The discounted costs of creating a pediatric cochlear implant user and of maintaining the child over the first 12 years were 48,757 pound silver($78,011). The discounted difference in education costs associated with a profoundly hearing-impaired child (HL >95 dB) as compared with a severely hearing-impaired child (HL 70-95 dB) over the same period was 26,781 pound silver($42,850). These represent the potential savings in educational costs associated with pediatric cochlear implantation. Assuming implantation at age 4 years, the discounted net average cost of pediatric cochlear implantation over compulsory school years (ages 4-16) was 21,976 pound silver ($35,162). Cochlear implants have been shown to improve the quality of life in adults by 0.23 points per annum (where quality of life is rated on a scale from 0 to 1). Applying this weight to children receiving implantation at age 4 years, and assuming a life expectancy of 74 years, the quality-adjusted life-year (QALY) gain is calculated to be 16.33. The cost per undiscounted QALY gain was estimated to be 1,345.70 pound silver ($2153.12) and per discounted QALY gain to be 10,341 pound silver ($16,545.60). CONCLUSION: This study provides evidence, based on conservative assumptions, to support the view that pediatric cochlear implantation is a cost-effective health care intervention in profoundly hearing-impaired young children.

The development of auditory perception in children following cochlear implantation.

The time course for the development of auditory perception in prelingually deaf children following cochlear implantation may extend over many years, thus making long-term studies necessary to evaluate any such outcome. However, few such studies exist in the literature. We prospectively followed-up a consecutive group of 133 prelingually deaf children up to 6 years following implantation. All children were prelingually deaf with age at onset of deafness < 3 years and age at implantation < 8 years. The aetiology of deafness was meningitis for 45 children (34%), congenital deafness for 77 children (58%) and other causes for 11 children (8%). All were implanted with a Nucleus-22 multi-channel cochlear implant and followed the same rehabilitation programme. No child was lost to follow-up and there were no exclusions from the study. Prelingually deaf children showed significant improvement in the auditory perception with implant experience. 82% of children who reached the 6-year interval could understand conversation without lip-reading. The respective percentage in the 4-year interval was 70%. The long-term results of cochlear implantation reveal that the majority of prelingually deaf children, when implanted before the age of 8 years, will develop significant auditory perception.

Cochlear implants in young children: the relationship between speech perception and speech intelligibility.

OBJECTIVE: To determine the relationship between measures of speech perception and speech production after cochlear implantation of young children with profound congenital and prelingual deafness. DESIGN: A prospective study was undertaken on a consecutive group of children with profound deafness. There were 126 children at the preimplantation interval and 71, 50, 26, and 20 children, respectively, at the 2, 3, 4, and 5 yr follow-up after implantation. Speech perception and speech intelligibility were assessed using hierarchical rating scales. Spearman rank correlation coefficients were used to determine the statistical correlations. All patients were either congenitally deaf or deafened before the age of 3 yr and were implanted before age 7 yr. The patients all received the Nucleus multichannel cochlear implant system with the most appropriate speech encoding strategy. RESULTS: Speech intelligibility at 5 yr was strongly correlated with speech perception at the 2, 3, 4, and 5 yr intervals after implantation (Spearman coefficients 0.77, 0.81, 0.58, 0.58; p < or = 0.01). Speech intelligibility at the 2, 3, and 4 yr intervals also correlated in a similar manner with earlier speech perception abilities (p < or = 0.01). CONCLUSIONS: The results suggest that speech intelligibility between 2 and 5 yr after implantation in young children with congenital and prelingual profound deafness can be predicted by measures of earlier speech perception.

Age at implantation: its importance in pediatric cochlear implantation.

OBJECTIVE: To assess the influence of age at implantation on speech perception and speech intelligibility following pediatric cochlear implantation. STUDY DESIGN: A prospective study was undertaken on a consecutive group of 126 congenital and prelingually deaf children up to 4 years after implantation. The study group was confined to prelingually deaf children less than 7 years of age at the time of implantation. All had implantation with the same multichannel cochlear implant system. No child was lost to follow-up, and there were no exclusions from the study. METHODS: The Iowa Matrix Closed Set Sentence test, connected discourse tracking, categories of auditory performance, and speech intelligibility rating were used to assess the speech perception (closed and open set) and speech intelligibility of the children with implants. Regression analysis and Spearman rank correlation coefficients were used to assess the correlation between the outcome measures with age at implantation. The setting was a tertiary referral pediatric cochlear implant center in the United Kingdom. RESULTS: Age at implantation positively correlated with preimplantation assessment performance and with most of the outcome measures up to 24 months following implantation. However, at the 3-and 4-year intervals following implantation, age at implantation was found to be a strong negative predictor of all the outcomes studied (correlation coefficients ranging from -0.44 to -0.58, all statistically significant [P<.05]). CONCLUSIONS: The results of the present study provide strong evidence that prelingually deaf children should receive implants as early as possible to facilitate the later development of speech perception skills and speech intelligibility and thus maximize the health gain from the intervention. However, because of the wide variation in individual outcomes, age alone should not be used as a criterion to decide implant candidacy.

Educational placement of deaf children following cochlear implantation.

This study examined the educational placements, before cochlear implantation, of 121 deaf children, and the educational placements, two years after implantation, of the 48 children who had reached that stage, looking at the influence of age at implantation and duration of deafness on the placement of these children. In addition, it compared the educational placements of those given implants prior to schooling, and those given implants when already in an educational setting. Categories used were pre-school, school for the deaf, unit or resource base within a mainstream school and full-time mainstream provision. Age at implantation and duration of deafness were found to be significant predictors of placement two years after implantation. The duration of deafness of children in schools for the deaf or units was twice that of children in mainstream education. Fifty-three per cent of children who were in pre-school at the time of implantation were in mainstream schools two years after implantation, whereas only 6% of those who were already in educational placements at the time of implantation were in mainstream education. This difference was statistically significant. The results indicate that children who are given implants early, before an educational decision has been made, are more likely to go to mainstream schools than those given implants when already in an educational setting.