Societal costs of permanent childhood hearing loss at teen age: a cross-sectional cohort follow-up study of universal newborn hearing screening.

OBJECTIVE: To investigate the effects in adolescence of bilateral permanent childhood hearing loss (PCHL) > 40 dB and of exposure to universal newborn hearing screening (UNHS) on societal costs accrued over the preceding 12 months. DESIGN SETTING PARTICIPANTS: An observational cohort study of a sample of 110 adolescents aged 13-20 years, 73 with PCHL and 37 in a normally hearing comparison group (HCG) closely similar in respect of place and date of birth to those with PCHL, drawn from a 1992-1997 cohort of 157 000 births in Southern England, half of whom had been exposed to a UNHS programme. INTERVENTION: Birth in periods with and without UNHS. OUTCOME MEASURES: Resource use and costs in the preceding 12-month period, estimated from interview at a mean age of 16.9 years and review of medical records. Effects on costs were examined in regression models. RESULTS: Mean total costs for participants with PCHL and the HCG were £15 914 and £5883, respectively (difference £10 031, 95% CI £6460 to £13 603), primarily driven by a difference in educational costs. Compared with the HCG, additional mean costs associated with PCHL of moderate, severe and profound severity were £5916, £6605 and £18 437, respectively. The presence of PCHL and an additional medical condition (AMC) increased costs by £15 385 (95% CI £8532 to £22 238). An increase of one unit in receptive language z-score was associated with £1616 (95% CI £842 to £2389) lower costs. Birth during periods of UNHS was not associated with significantly lower overall costs (difference £3594, 95% CI -£2918 to £10 106). CONCLUSIONS: The societal cost of PCHL was greater with more severe losses and in the presence of AMC and was lower in children with superior language scores. There was no statistically significant reduction in costs associated with birth in periods with UNHS. TRIAL REGISTRATION NUMBER: ISRCTN03307358, pre-results.

Predicting reading ability in teenagers who are deaf or hard of hearing: A longitudinal analysis of language and reading.

BACKGROUND: Deaf and hard of hearing (D/HH) children and young people are known to show group-level deficits in spoken language and reading abilities relative to their hearing peers. However, there is little evidence on the longitudinal predictive relationships between language and reading in this population. AIMS: To determine the extent to which differences in spoken language ability in childhood predict reading ability in D/HH adolescents. METHODS: and procedures: Participants were drawn from a population-based cohort study and comprised 53 D/HH teenagers, who used spoken language, and a comparison group of 38 normally hearing teenagers. All had completed standardised measures of spoken language (expression and comprehension) and reading (accuracy and comprehension) at 6-10 and 13-19 years of age. OUTCOMES: and results: Forced entry stepwise regression showed that, after taking reading ability at age 8 years into account, language scores at age 8 years did not add significantly to the prediction of Reading Accuracy z-scores at age 17 years (change in R2 = 0.01, p = .459) but did make a significant contribution to the prediction of Reading Comprehension z-scores at age 17 years (change in R2  = 0.17, p < .001). CONCLUSIONS: and implications: In D/HH individuals who are spoken language users, expressive and receptive language skills in middle childhood predict reading comprehension ability in adolescence. Continued intervention to support language development beyond primary school has the potential to benefit reading comprehension and hence educational access for D/HH adolescents.

Language and reading comprehension in middle childhood predicts emotional and behaviour difficulties in adolescence for those with permanent childhood hearing loss.

BACKGROUND: Permanent childhood hearing loss (PCHL) is associated with an elevated level of emotional and behaviour difficulties (EBD). In children and adolescents with PCHL, EBD has been found to be linked to language ability in children with PCHL. The present study was designed to test whether childhood language and/or reading comprehension abilities of children with PCHL predict subsequent EBD in adolescence. METHODS: Language comprehension (LC) and reading comprehension (RC) were measured at ages 6-10 years (Time 1) and 13-20 years (Time 2) in participants with PCHL who preferred to communicate using spoken language (n = 57) and a hearing comparison group (n = 38). EBD was measured at both time points by parent and by teacher ratings on the Strengths and Difficulties Questionnaire. RESULTS: Within the PCHL group there were negative correlations between EBD scores and concurrent LC and RC scores at Time 1 and at Time 2. Cross-lagged latent variable models fitted to the longitudinal data indicated that the associations between LC, RC and teacher-rated EBD were more likely to arise from the impact of LC and RC on behaviour rather than the other way around. CONCLUSIONS: In those with PCHL, poor language and reading comprehension in middle childhood increased the risk of emotional and behaviour difficulties at school in the teenage years. The results suggest that effective language and literacy interventions for children with hearing loss may also bring benefits to their mental health.

Emotional and behaviour difficulties in teenagers with permanent childhood hearing loss.

OBJECTIVES: It is known that during the middle childhood years those with permanent childhood hearing loss (PCHL) are at increased risk of showing emotional and behaviour difficulties (EBD). It has yet to be established whether this risk continues into the late teenage years. There is a paucity of longitudinal studies on the association between PCHL and EBD. METHODS: The Strengths and Difficulties Questionnaire (SDQ) was used to measure EBD based on parent, teacher and self-ratings in 76 teenagers with PCHL and 38 in a hearing comparison group (HCG) from a population sample of children that was followed up from birth to adolescence. RESULTS: On parent-rated SDQ, the PCHL group had significantly higher Total Difficulties score than the HCG (Standardised mean difference (SMD) = +0.39, 95%CI 0.00 to 0.79). Amongst the PCHL group the presence of disabilities other than hearing loss had a substantial impact on the level of parent-rated EBD (SMD = +1.68, 1.04 to 2.33). There was a relationship between receptive language ability and EBD in both the HCG (r = -0.33, 95%CI -0.59 to -0.01) and the PCHI group (r = -0.33, 95%CI -0.53 to -0.02). The effect of PCHL on EBD became non-significant when receptive language was included as a covariate (F = 0.12, df = 1,95, p = 0.729). Early confirmation of hearing loss (i.e. before 9 months of age) did not have a significant effect on EBD scores (SMD = +0.31, 95%CI -0.15 to 0.77). CONCLUSIONS: PCHL continues to be associated with elevated EBD scores as measured by parent rated SDQ into the late teenage years but the degree of this elevation is less than in childhood and is not apparent on teacher or self-ratings. Poor receptive language ability appeared to account for these elevated EBD scores in the group with PCHL. Particular attention needs to be paid to the mental health of children and adolescents with PCHL that is accompanied by other disabilities and to those with poor receptive language ability. However, the majority of teenagers with PCHL do not show clinically significant elevated levels of EBD.

Language Outcomes in Deaf or Hard of Hearing Teenagers Who Are Spoken Language Users: Effects of Universal Newborn Hearing Screening and Early Confirmation.

OBJECTIVES: This study aimed to examine whether (a) exposure to universal newborn hearing screening (UNHS) and b) early confirmation of hearing loss were associated with benefits to expressive and receptive language outcomes in the teenage years for a cohort of spoken language users. It also aimed to determine whether either of these two variables was associated with benefits to relative language gain from middle childhood to adolescence within this cohort. DESIGN: The participants were drawn from a prospective cohort study of a population sample of children with bilateral permanent childhood hearing loss, who varied in their exposure to UNHS and who had previously had their language skills assessed at 6-10 years. Sixty deaf or hard of hearing teenagers who were spoken language users and a comparison group of 38 teenagers with normal hearing completed standardized measures of their receptive and expressive language ability at 13-19 years. RESULTS: Teenagers exposed to UNHS did not show significantly better expressive (adjusted mean difference, 0.40; 95% confidence interval [CI], -0.26 to 1.05; d = 0.32) or receptive (adjusted mean difference, 0.68; 95% CI, -0.56 to 1.93; d = 0.28) language skills than those who were not. Those who had their hearing loss confirmed by 9 months of age did not show significantly better expressive (adjusted mean difference, 0.43; 95% CI, -0.20 to 1.05; d = 0.35) or receptive (adjusted mean difference, 0.95; 95% CI, -0.22 to 2.11; d = 0.42) language skills than those who had it confirmed later. In all cases, effect sizes were of small size and in favor of those exposed to UNHS or confirmed by 9 months. Subgroup analysis indicated larger beneficial effects of early confirmation for those deaf or hard of hearing teenagers without cochlear implants (N = 48; 80% of the sample), and these benefits were significant in the case of receptive language outcomes (adjusted mean difference, 1.55; 95% CI, 0.38 to 2.71; d = 0.78). Exposure to UNHS did not account for significant unique variance in any of the three language scores at 13-19 years beyond that accounted for by existing language scores at 6-10 years. Early confirmation accounted for significant unique variance in the expressive language information score at 13-19 years after adjusting for the corresponding score at 6-10 years (R change = 0.08, p = 0.03). CONCLUSIONS: This study found that while adolescent language scores were higher for deaf or hard of hearing teenagers exposed to UNHS and those who had their hearing loss confirmed by 9 months, these group differences were not significant within the whole sample. There was some evidence of a beneficial effect of early confirmation of hearing loss on relative expressive language gain from childhood to adolescence. Further examination of the effect of these variables on adolescent language outcomes in other cohorts would be valuable.

The impact of universal newborn hearing screening on long-term literacy outcomes: a prospective cohort study.

OBJECTIVE: To determine whether the benefits of universal newborn hearing screening (UNHS) seen at age 8 years persist through the second decade. DESIGN: Prospective cohort study of a population sample of children with permanent childhood hearing impairment (PCHI) followed up for 17 years since birth in periods with (or without) UNHS. SETTING: Birth cohort of 100 000 in southern England. PARTICIPANTS: 114 teenagers aged 13-19 years, 76 with PCHI and 38 with normal hearing. All had previously their reading assessed aged 6-10 years. INTERVENTIONS: Birth in periods with and without UNHS; confirmation of PCHI before and after age 9 months. MAIN OUTCOME MEASURE: Reading comprehension ability. Regression modelling took account of severity of hearing loss, non-verbal ability, maternal education and main language. RESULTS: Confirmation of PCHI by age 9 months was associated with significantly higher mean z-scores for reading comprehension (adjusted mean difference 1.17, 95% CI 0.36 to 1.97) although birth during periods with UNHS was not (adjusted mean difference 0.15, 95% CI -0.75 to 1.06). The gap between the reading comprehension z-scores of teenagers with early compared with late confirmed PCHI had widened at an adjusted mean rate of 0.06 per year (95% CI -0.02 to 0.13) during the 9.2-year mean interval since the previous assessment. CONCLUSIONS: The benefit to reading comprehension of confirmation of PCHI by age 9 months increases during the teenage years. This strengthens the case for UNHS programmes that lead to early confirmation of permanent hearing loss. TRIAL REGISTRATION NUMBER: ISRCTN03307358.

Emotional and behavioural difficulties in children and adolescents with hearing impairment: a systematic review and meta-analysis.

The aim of this study is to estimate the extent to which children and adolescents with hearing impairment (HI) show higher rates of emotional and behavioural difficulties compared to normally hearing children. Studies of emotional and behavioural difficulties in children and adolescents were traced from computerized systematic searches supplemented, where appropriate, by studies referenced in previous narrative reviews. Effect sizes (Hedges' g) were calculated for all studies. Meta-analyses were conducted on the weighted effect sizes obtained for studies adopting the Strength and Difficulties Questionnaire (SDQ) and on the unweighted effect sizes for non-SDQ studies. 33 non-SDQ studies were identified in which emotional and behavioural difficulties in children with HI could be compared to normally hearing children. The unweighted average g for these studies was 0.36. The meta-analysis of the 12 SDQ studies gave estimated effect sizes of 0.23 (95% CI 0.07, 0.40), 0.34 (95% CI 0.19, 0.49) and -0.01 (95% CI -0.32, 0.13) for Parent, Teacher and Self-ratings of Total Difficulties, respectively. The SDQ sub-scale showing consistent differences across raters between groups with HI and those with normal hearing was Peer Problems. Children and adolescents with HI have scores on emotional and behavioural difficulties measures about a quarter to a third of a standard deviation higher than hearing children. Children and adolescents with HI are in need of support to help their social relationships particularly with their peers.

The effect of early confirmation of hearing loss on the behaviour in middle childhood of children with bilateral hearing impairment.

AIM: To determine if the benefit of early confirmation of permanent childhood hearing impairment (PCHI) on children's receptive language development is associated with fewer behavioural problems. METHOD: Follow-up of a total population cohort of 120 children with PCHI of moderate or greater severity (≥ 40 decibels relative to hearing threshold level) (67 males, 53 females; mean age 7 y 11 mo, range 5 y 5 mo-11 y 8 mo) and 63 hearing children (37 males, 26 females; mean age 8 y 1 mo, range 6 y 4 mo-9 y 10 mo). The main outcome measures were the Strengths and Difficulties Questionnaire (SDQ) completed by teachers and parents and the Vineland Adaptive Behaviour Scales (VABS) which are completed on the basis of a parental interview. RESULTS: Children with PCHI had lower standard scores than hearing children on the Daily Living Skills (p=0.001) and the Socialisation (p=0.001) scales of the VABS. They had significantly higher Total Behaviour Problem scores on the parent-rated (p=0.002) and teacher-rated SDQ (p=0.03). Children for whom PCHI was confirmed by 9 months did not have significantly fewer problems on the behavioural measures than those confirmed after that age (p=0.635 and p=0.196). INTERPRETATION: Early confirmation has a beneficial effect on receptive language development but no significant impact in reducing behavioural problems in children with PCHI.

Narrative skills following early confirmation of permanent childhood hearing impairment.

AIM: the aim of this study was to compare spoken language production in children with permanent childhood hearing impairment (PCHI) whose PCHI was confirmed either early or late. METHOD: audio-taped spoken narrative was assessed for syntax, phonology, morphology, and narrative in transcripts from a population-based sample of 89 children (49 males, 40 females; age mean age 7y 7mo, SD1y 1mo, range 6y 6mo-10y 9mo) with bilateral PCHI (≥ 40dB hearing loss) and a comparison group of 63 children (37 males, 26 females; mean age 8y 1mo; SD 1y) with normal hearing. Of the 89 children with PCHI, 41 (21 males, 20 females) had their hearing impairment confirmed by the age of 9 months. All children with PCHI were tested with hearing aids in place, including 16 with cochlear implants. The group of children whose PCHI had been confirmed by age 9 months was compared with the group with later confirmation of PCHI using regression models on the outcome measures. RESULTS: compared with those with late-confirmed PCHI, children with early-confirmed PCHI used significantly more sentences (mean difference 2.7; 95% confidence interval [CI] 0.49-5.24; p=0.019) and categories of high-pitched morphological markers (mean difference 6.64; 95% CI 1.96-11.31; p=0.006). The number of categories of low-pitched morphological markers, phonological simplifications, and sentences with multiple clauses did not differ between groups. The odds ratios (95% CI) of superior narrative structure and narrative content in children whose PCHI was confirmed early were 3.03 (1.09-8.46; p=0.034) and 4.43 (1.52-12.89; p=0.006) respectively. INTERPRETATION: early confirmation compared with late confirmation of PCHI was associated with benefit to narrative skills and to certain expressive aspects of syntax and morphology, but not expressive phonology.

The relationship between language development and behaviour problems in children with hearing loss.

BACKGROUND: There are well-replicated findings that link poor development on a range of communication skills with increased behavioural problems. This paper examines this relationship in children with hearing loss. METHOD: One hundred and twenty children with hearing loss (67 boys, 53 girls) and 63 hearing children (37 boys, 26 girls) with a mean age of 8 years from eight districts in Southern England were assessed for receptive and expressive language skills. The relationships between these measures and an aggregate of parent- and teacher-reported behaviour problems in the children were investigated. RESULTS: Children with hearing loss had higher levels of behaviour problems compared to hearing children. Once the language abilities of children with hearing loss are taken into account, the negative effects of hearing loss on behaviour disappear. CONCLUSIONS: Behaviour problems are found more commonly in children with hearing loss and the level of behaviour problems is highest amongst those children with hearing loss with the least developed language capabilities.

Health status and health-related quality of life preference-based outcomes of children who are aged 7 to 9 years and have bilateral permanent childhood hearing impairment.

OBJECTIVE: The objective of this study was to describe the health status and health-related quality of life preference-based outcomes of children with diagnosed bilateral permanent childhood hearing impairment and a comparison group of English-speaking children with normal hearing. METHODS: We studied 120 children who were aged 7 to 9 years and had bilateral permanent childhood hearing impairment of moderate or greater severity, identified from a cohort of 156,733 children who were born in 8 districts of southern England, and 63 English-speaking children with normal hearing and the same place of birth and age at assessment. Principal caregivers were interviewed by using the Health Utilities Index Mark III questionnaire for proxy-assessed usual health status assessment. Levels of function within each of the 8 attributes of the Health Utilities Index Mark III (cognition, vision, hearing, speech, ambulation, dexterity, emotion, and pain) were recorded. RESULTS: Bilateral permanent childhood hearing impairment is associated with significantly increased proportions of suboptimal levels of function and significantly lower single-attribute utility scores in 6 of the 8 attributes of the Health Utilities Index Mark III: vision, hearing, speech, ambulation, dexterity, and cognition. Compared with the children with normal hearing, the mean multiattribute utility score for the children with hearing impairment was significantly lower for both the whole group and the moderate, severe, and profound severity subgroups. The differences in the distributions of the multiattribute utility scores between the children with hearing impairment as a group and the children with normal hearing and between each of the severity subgroups and the children with normal hearing all were statistically significant. CONCLUSIONS: This study provides rigorous evidence of an association between bilateral permanent childhood hearing impairment and diminished health status and health-related quality of life preference-based outcomes during midchildhood.

Language ability in children with permanent hearing impairment: the influence of early management and family participation.

OBJECTIVE: The goal was to examine the relationships between management after confirmation, family participation, and speech and language outcomes in the same group of children with permanent childhood hearing impairment. METHODS: Speech, oral language, and nonverbal abilities, expressed as z scores and adjusted in a regression model, and Family Participation Rating Scale scores were assessed at a mean age of 7.9 years for 120 children with bilateral permanent childhood hearing impairment from a 1992-1997 United Kingdom birth cohort. Ages at institution of management and hearing aid fitting were obtained retrospectively from case notes. RESULTS: Compared with children managed later (> 9 months), those managed early (< or = 9 months) had higher adjusted mean z scores for both receptive and expressive language, relative to nonverbal ability, but not for speech. Compared with children aided later, a smaller group of more-impaired children aided early did not have significantly higher scores for these outcomes. Family Participation Rating Scale scores showed significant positive correlations with language and speech intelligibility scores only for those with confirmation after 9 months and were highest for those with late confirmed, severe/profound, permanent childhood hearing impairment. CONCLUSIONS: Early management of permanent childhood hearing impairment results in improved language. Family participation is also an important factor in cases that are confirmed late, especially for children with severe or profound permanent childhood hearing impairment.

Language ability after early detection of permanent childhood hearing impairment.

BACKGROUND: Children with bilateral permanent hearing impairment often have impaired language and speech abilities. However, the effects of universal newborn screening for permanent bilateral childhood hearing impairment and the effects of confirmation of hearing impairment by nine months of age on subsequent verbal abilities are uncertain. METHODS: We studied 120 children with bilateral permanent hearing impairment identified from a large birth cohort in southern England, at a mean of 7.9 years of age. Of the 120 children, 61 were born during periods with universal newborn screening and 57 had hearing impairment that was confirmed by nine months of age. The primary outcomes were language as compared with nonverbal ability and speech expressed as z scores (the number of standard deviations by which the score differed from the mean score among 63 age-matched children with normal hearing), adjusted for the severity of the hearing impairment and for maternal education. RESULTS: Confirmation of hearing impairment by nine months of age was associated with higher adjusted mean z scores for language as compared with nonverbal ability (adjusted mean difference for receptive language, 0.82; 95 percent confidence interval, 0.31 to 1.33; and adjusted mean difference for expressive language, 0.70; 95 percent confidence interval, 0.13 to 1.26). Birth during periods with universal newborn screening was also associated with higher adjusted z scores for receptive language as compared with nonverbal ability (adjusted mean difference, 0.60; 95 percent confidence interval, 0.07 to 1.13), although the z scores for expressive language as compared with nonverbal ability were not significantly higher. Speech scores did not differ significantly between those who were exposed to newborn screening or early confirmation and those who were not. CONCLUSIONS: Early detection of childhood hearing impairment was associated with higher scores for language but not for speech in midchildhood.

The economic costs of congenital bilateral permanent childhood hearing impairment.

OBJECTIVE: The objective of this study was to estimate the economic costs of bilateral permanent childhood hearing impairment (PCHI) in the preceding year of life for children aged 7 to 9 years. METHODS: A cost analysis was conducted by using a birth cohort of children born between 1992 and 1997 in 8 districts of Southern England, of which half had been born into populations exposed to universal newborn screening (UNS). Unit costs were applied to estimates of health, social, and broader resource use made by 120 hearing-impaired children and 63 children in a normally hearing comparison group. Associations between societal costs per child and severity of hearing impairment, language ability score, exposure to UNS, and age of confirmation were analyzed, including adjustment for potential confounders in a linear regression model. RESULTS: The mean societal cost in the preceding year of life at 7 to 9 years of age was 14092.5 pound sterling for children with PCHI, compared with 4206.8 pound sterling for the normally hearing children, a cost difference of 9885.7 pound sterling. After adjusting for severity and other potential confounders in a linear regression model, mean societal costs among children with PCHI were reduced by 2553 pound sterling for each unit increase in the z score for receptive language. Using similar regression models, exposure to a program of UNS was associated with a smaller cost reduction of 2213.2 pound sterling, whereas costs were similar between children whose PCHI was confirmed at <9 or >9 months. CONCLUSIONS. The study provides rigorous evidence of the annual health, social, and broader societal cost of bilateral PCHI in the preceding year of life at 7 to 9 years of age and shows that it is related to its severity and has an inverse relationship with language abilities after adjustment for severity.