Long-Term Variability of Distortion-Product Otoacoustic Emissions in Infants and Children and Its Relation to Pediatric Ototoxicity Monitoring

Objective: Distortion-product otoacoustic emissions (DPOAEs) provide a rapid, noninvasive measure of outer hair cell damage associated with chemotherapy and are a key component of pediatric ototoxicity monitoring. Serial monitoring of DPOAE levels in reference to baseline measures is one method for detecting ototoxic damage. Interpreting DPOAE findings in this context requires that test–retest differences be considered in relation to normal variability, data which are lacking in children. This study sought to (1) characterize normal test–retest variability in DPOAE level over the long time periods reflective of pediatric chemotherapy regimens for a variety of childhood ages and f2 primary frequencies using common clinical instrumentation and stimulus parameters; (2) develop level-shift reference intervals; and (3) account for any age-related change in DPOAE level or measurement error that may occur as the auditory system undergoes maturational change early in life. Design: Serial DPOAE measurements were obtained in 38 healthy children (25 females and 13 males) with normal hearing and ranging in age from one month to 10 years at the initial (baseline) visit. On average, children were tested 5.2 times over an observation period of 6.5 months. Data were collected in the form of DP grams, in which DPOAE level was measured for f2 ranging from 1.4 to 10 kHz, using a fixed f2/f1 ratio of 1.22 and stimulus level of 65/55 dB SPL for L1/L2. Age effects on DPOAE level and measurement error were estimated using Bayesian regression of the longitudinal data. The raw and model-based distribution of DPOAE test–retest differences were characterized using means and standard error of the measurement for several ages and f2’s. Results: DPOAE test–retest differences for the children in this study are at the high end of those previously observed in adults, as reflected in the associated shift reference intervals. Further, although we observe substantial child-specific variation in DPOAE level, the pattern of age-related changes is highly consistent across children. Across a wide range of f2’s, DPOAE level decreases by 3 to 4 dB from 1 to 13 months of age followed by a more gradual decline of

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Long-Term Variability of Distortion-Product Otoacoustic Emissions in Infants and Children and Its Relation to Pediatric Ototoxicity Monitoring

Objective: Distortion-product otoacoustic emissions (DPOAEs) provide a rapid, noninvasive measure of outer hair cell damage associated with chemotherapy and are a key component of pediatric ototoxicity monitoring. Serial monitoring of DPOAE levels in reference to baseline measures is one method for detecting ototoxic damage. Interpreting DPOAE findings in this context requires that test–retest differences be considered in relation to normal variability, data which are lacking in children. This study sought to (1) characterize normal test–retest variability in DPOAE level over the long time periods reflective of pediatric chemotherapy regimens for a variety of childhood ages and f2 primary frequencies using common clinical instrumentation and stimulus parameters; (2) develop level-shift reference intervals; and (3) account for any age-related change in DPOAE level or measurement error that may occur as the auditory system undergoes maturational change early in life. Design: Serial DPOAE measurements were obtained in 38 healthy children (25 females and 13 males) with normal hearing and ranging in age from one month to 10 years at the initial (baseline) visit. On average, children were tested 5.2 times over an observation period of 6.5 months. Data were collected in the form of DP grams, in which DPOAE level was measured for f2 ranging from 1.4 to 10 kHz, using a fixed f2/f1 ratio of 1.22 and stimulus level of 65/55 dB SPL for L1/L2. Age effects on DPOAE level and measurement error were estimated using Bayesian regression of the longitudinal data. The raw and model-based distribution of DPOAE test–retest differences were characterized using means and standard error of the measurement for several ages and f2’s. Results: DPOAE test–retest differences for the children in this study are at the high end of those previously observed in adults, as reflected in the associated shift reference intervals. Further, although we observe substantial child-specific variation in DPOAE level, the pattern of age-related changes is highly consistent across children. Across a wide range of f2’s, DPOAE level decreases by 3 to 4 dB from 1 to 13 months of age followed by a more gradual decline of

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Experience does not influence injury-related joint kinematics and kinetics in distance runners

Publication date: March 2018
Source:Gait & Posture, Volume 61
Author(s): Cristine E. Agresta, Jillian Peacock, Jeffrey Housner, Ronald F. Zernicke, Jessica Deneweth Zendler
PurposeIncreased running experience and more time spent running appears to be advantageous in reducing injury risk, although the reason behind this is unclear. It is plausible that more experience results in better running mechanics leading to less injuries. Running mechanics are often screened during clinical assessments and targeted for correction in gait retraining, particularly those thought to be global indicators of injury or those associated with elevated knee joint loading. Examining the biomechanics of runners who are less-injury prone can improve our understanding of the significance of faulty running mechanics in relation to injury. Our goal was to examine if running experience was correlated to differences in kinematics and kinetics associated with increased knee joint loading and running-related injury risk.MethodsOne hundred runners with varying experience ran on a pressure-sensing treadmill at a self-selected speed. Trunk and lower extremity kinematics, spatiotemporal measures, and ground reaction forces were collected. Multiple linear regression was used to assess the association between experience and three-dimensional hip kinematics, sagittal plane lower-extremity mechanics, and ground reaction forces while controlling for age and speed.ResultsIncreased running experience was not significantly associated with running mechanics. Increased age was significantly associated with reduced peak knee flexion and increased contact time. Running speed influenced several spatiotemporal, kinematic, and kinetic variables.ConclusionIncreased years of running experience does not appear to significantly influence running mechanics. However, age and running speed do influence biomechanical variables associated with injury in distance runners. Thus, there may be factors, other than running mechanics, that contribute to less risk in more experienced runners.



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Experience does not influence injury-related joint kinematics and kinetics in distance runners

Publication date: March 2018
Source:Gait & Posture, Volume 61
Author(s): Cristine E. Agresta, Jillian Peacock, Jeffrey Housner, Ronald F. Zernicke, Jessica Deneweth Zendler
PurposeIncreased running experience and more time spent running appears to be advantageous in reducing injury risk, although the reason behind this is unclear. It is plausible that more experience results in better running mechanics leading to less injuries. Running mechanics are often screened during clinical assessments and targeted for correction in gait retraining, particularly those thought to be global indicators of injury or those associated with elevated knee joint loading. Examining the biomechanics of runners who are less-injury prone can improve our understanding of the significance of faulty running mechanics in relation to injury. Our goal was to examine if running experience was correlated to differences in kinematics and kinetics associated with increased knee joint loading and running-related injury risk.MethodsOne hundred runners with varying experience ran on a pressure-sensing treadmill at a self-selected speed. Trunk and lower extremity kinematics, spatiotemporal measures, and ground reaction forces were collected. Multiple linear regression was used to assess the association between experience and three-dimensional hip kinematics, sagittal plane lower-extremity mechanics, and ground reaction forces while controlling for age and speed.ResultsIncreased running experience was not significantly associated with running mechanics. Increased age was significantly associated with reduced peak knee flexion and increased contact time. Running speed influenced several spatiotemporal, kinematic, and kinetic variables.ConclusionIncreased years of running experience does not appear to significantly influence running mechanics. However, age and running speed do influence biomechanical variables associated with injury in distance runners. Thus, there may be factors, other than running mechanics, that contribute to less risk in more experienced runners.



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Experience does not influence injury-related joint kinematics and kinetics in distance runners

Publication date: March 2018
Source:Gait & Posture, Volume 61
Author(s): Cristine E. Agresta, Jillian Peacock, Jeffrey Housner, Ronald F. Zernicke, Jessica Deneweth Zendler
PurposeIncreased running experience and more time spent running appears to be advantageous in reducing injury risk, although the reason behind this is unclear. It is plausible that more experience results in better running mechanics leading to less injuries. Running mechanics are often screened during clinical assessments and targeted for correction in gait retraining, particularly those thought to be global indicators of injury or those associated with elevated knee joint loading. Examining the biomechanics of runners who are less-injury prone can improve our understanding of the significance of faulty running mechanics in relation to injury. Our goal was to examine if running experience was correlated to differences in kinematics and kinetics associated with increased knee joint loading and running-related injury risk.MethodsOne hundred runners with varying experience ran on a pressure-sensing treadmill at a self-selected speed. Trunk and lower extremity kinematics, spatiotemporal measures, and ground reaction forces were collected. Multiple linear regression was used to assess the association between experience and three-dimensional hip kinematics, sagittal plane lower-extremity mechanics, and ground reaction forces while controlling for age and speed.ResultsIncreased running experience was not significantly associated with running mechanics. Increased age was significantly associated with reduced peak knee flexion and increased contact time. Running speed influenced several spatiotemporal, kinematic, and kinetic variables.ConclusionIncreased years of running experience does not appear to significantly influence running mechanics. However, age and running speed do influence biomechanical variables associated with injury in distance runners. Thus, there may be factors, other than running mechanics, that contribute to less risk in more experienced runners.



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Examining Motivation for Hearing Health Care in those with Self-Reported Hearing Problems and Hearing Thresholds within Normal Limits

At one point or another, you've likely seen a patient in your clinical practice who has reported substantial hearing difficulties but whose traditional audiometric test results are essentially within normal limits. When considering next steps and/or rehabilitation options for these individuals, have you been curious how motivated such individuals are for hearing health-care services? Is it any different than your patients who have had a measurable pure-tone hearing loss?

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Exploring Hearing Aid Problems: Perspectives of Hearing Aid Owners and Clinicians

Objectives: To gather perspectives of hearing aid owners and hearing healthcare clinicians with regard to problems that arise after hearing aid fitting and use these perspectives to generate a conceptual framework to gain a better understanding of these problems. Design: Participants included a group of 17 hearing aid owners and a group of 21 hearing healthcare clinicians; data collection occurred separately for each group. Participants each attended two group sessions in Perth, Western Australia, wherein they: (1) generated statements describing the problems associated with hearing aids and (2) grouped and rated the statements to identify key themes. Concept mapping was used to generate a conceptual framework. Results: Participants identified four concepts regarding hearing aid problems as follows: (1) hearing aid management; (2) hearing aid sound quality and performance; (3) feelings, thoughts, and behaviors; and (4) information and training. While hearing aid owners and clinicians generated similar results regarding the concepts derived, the clinicians reported that the problems identified had a greater negative impact on hearing aid success than did hearing aid owners. Conclusions: The magnitude and diversity of hearing aid problems identified in this study highlight the ongoing challenges that hearing aid owners face and suggest that current processes for hearing aid fitting can be improved. Problems relating to hearing aid management were most often deemed to have the greatest impact on hearing aid success and be the most preventable/solvable, and thus are a good starting point when addressing hearing aid–related problems.

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Comparison of Different Hearing Aid Prescriptions for Children

Objectives: To assess whether there are significant differences between speech scores for different hearing aid prescription methods, specifically DSL i/o, DSL V, and NAL-NL1, using age-appropriate closed- and open-set speech tests with young children, designed to avoid floor and ceiling effects. Design: Participants were 44 children with moderate or severe bilateral hearing loss, 8 aged 2 to 3 years, 15 aged 4 to 5 years, and 21 aged 6 to 9 years. Children wore bilateral hearing aids fitted with each prescription method in turn in a balanced double-blind design. The speech tests used with each child (and for some tests the levels) were chosen so as to avoid floor and ceiling effects. For the closed-set tests, the level used was selected for each child based on their hearing loss. The tests used were: (1) The closed-set Consonant Confusion Test of word identification; (2) The closed-set Chear Auditory Perception Test (CAPT) of word identification. This has separate sections assessing discrimination of consonants and vowels and detection of consonants; (3) The open-set Cambridge Auditory Word Lists for testing word identification at levels of 50 and 65 dBA, utilizing 10 consonant–vowel–consonant real words that are likely to be familiar to children aged 3 years or older; (4) The open-set Common Phrases Test to measure the speech reception threshold in quiet; (5) Measurement of the levels required for identification of the Ling 5 sounds, using a recording of the sounds made at the University of Western Ontario. Results: Scores for the Consonant Confusion Test and CAPT consonant discrimination and consonant detection were lower for the NAL-NL1 prescription than for the DSL prescriptions. Scores for the CAPT vowel-in-noise discrimination test were higher for DSL V than for either of the other prescriptions. Scores for the Cambridge Auditory Word Lists did not differ across prescriptions for the level of 65 dBA, but were lower for the NAL-NL1 prescription than for either of the DSL prescriptions for the level of 50 dBA. The speech reception threshold measured using the Common Phrases Test and the levels required for identification of the Ling 5 sounds were higher (worse) for the NAL-NL1 prescription than for the DSL prescriptions. Conclusions: The higher gains prescribed by the DSL i/o and DSL V prescription methods relative to NAL-NL1 led to significantly better detection and discrimination of low-level speech sounds.

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Experimental Simulation of Clinical Borderline Situations in Temporal Bone Specimens After Ossiculoplasty

Objectives: One reason for insufficient hearing improvement with a distinct air–bone gap after ossiculoplasty with implantation of partial or total ossicular replacement prostheses can be the dislocation or minimal shifting of the prosthesis. The aim of this study was the simulation of common clinical borderline situations with minimal shifting of the prosthesis in temporal bone specimens after ossiculoplasty. It was furthermore the goal to identify these specific situations through imaging by cone beam computed tomography (cbCT) and direct visual inspection using the operation microscope. Additionally, the functional status was evaluated using laser-Doppler vibrometry (LDV). Design: We used a total of four temporal bone specimens for this study. A reconstruction with a partial ossicular replacement prostheses was performed in three specimens and with a total ossicular replacement prostheses in one specimen, with good initial acoustic properties. Subsequently, one specific type of prosthesis failure was simulated in each specimen, respectively, by minimally shifting, tilting, or bending the prostheses from their initial positions. These changes were introduced step-by-step until a borderline situation just short of complete acoustic decoupling was reached. Each step was examined using both LDV and cbCT and observed through the operation microscope. Results: LDV was able to quantify the mechanic function of the ossicular chain after most of the manipulation steps by demonstrating the effect of any shifting of the prosthesis on the middle ear transfer function. However, in some situations, the middle ear transfer function was better with a visually more advanced failure of the prosthesis. In addition, cbCT showed most of the steps with excellent resolution and was able to delineate changes in soft tissue (e.g., cartilage covering). Conclusion: cbCT seems to be a promising imaging technique for middle ear problems. As cbCT and LDV exhibited slightly different advantages and disadvantages regarding the demonstration of borderline situations, the combination of both techniques allowed for a more precise evaluation of middle ear reconstructions. Knowledge of the specific characteristics of these methods and their possible combination might help otologists and otosurgeons to refine indications for revision surgery and improve their personal patient counseling.

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Closed-Set Speech Discrimination Tests for Assessing Young Children

Objective: The main objective of this study is to obtain data assessing normative scores, test–retest reliability, critical differences, and the effect of age for two closed-set consonant-discrimination tests. Design: The two tests are intended for use with children aged 2 to 8 years. The tests were evaluated using normal-hearing children within the appropriate age range. The tests were (1) the closed-set consonant confusion test (CCT) and (2) the consonant-discrimination subtest of the closed-set Chear Auditory Perception Test (CAPT). Both were word-identification tests using stimuli presented at a low fixed level, chosen to avoid ceiling effects while avoiding the use of background noise. Each test was administered twice. Results: All children in the age range 3 years 2 months to 8 years 11 months gave meaningful scores and were able to respond reliably using a computer mouse or a touch screen to select one of four response options displayed on a screen for each trial. Assessment of test–retest reliability showed strong agreement between the two test runs (interclass correlation ≥ 0.8 for both tests). The critical differences were similar to those for other monosyllabic speech tests. Tables of these differences for the CCT and CAPT are provided for clinical use of the measures. Performance tended to improve with increasing age, especially for the CCT. Regression equations relating mean performance to age are given. Conclusions: The CCT is appropriate for children with developmental age in the range 2 to 4.5 years and the CAPT is appropriate as a follow-on test from the CCT. If a child scores 80% or more on the CCT, they can be further tested using the CAPT, which contains more advanced vocabulary and more difficult contrasts. This allows the assessment of consonant perception ability and of changes over time or after an intervention.

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Contralateral Interference Caused by Binaurally Presented Competing Speech in Adult Bilateral Cochlear-Implant Users

Objectives: Bilateral cochlear implants (BI-CIs) are intended to improve sound localization and speech understanding in the presence of interfering sounds. For normal-hearing listeners, improved speech understanding in the presence of interfering sounds can be achieved with monaural head shadow and binaural unmasking. While some BI-CI listeners experience binaural unmasking under certain testing conditions, others appear to not. This study tested a group of BI-CI users with hearing histories that have been linked to poor binaural processing—early onset of deafness or long duration of deafness in just one ear. We predicted that these listeners would experience the opposite of binaural unmasking (i.e., contralateral interference) when trying to understand speech in the presence of a competing talker. Design: Nine adult BI-CI users who were deafened early in life or had an asymmetric hearing history (e.g., a much longer duration of deafness in one ear) participated in this study. The coordinate response measure corpus was used to assess speech understanding for a male target talker in quiet or in the presence of one male competing talker. Experiment 1 measured binaural unmasking in a paradigm that provided no head-shadow component. The target was always presented monaurally, while the interferer was presented either monaurally or diotically. Experiment 2 measured spatial release from masking in a paradigm that included both a head-shadow component and possible binaural-unmasking component. Nonindividualized head-related transfer functions were used to simulate talker locations in the front or 90° to the left or right. Results: In experiment 1, all nine listeners experienced contralateral interference (9 dB on average). Four listeners demonstrated roughly symmetric contralateral interference; five listeners experienced asymmetrical contralateral interference. In experiment 2, the listeners experienced only 1 dB of spatial release from masking on average; this small amount was possibly a result of the contralateral interference observed in experiment 1. The results were best explained by individual differences in speech understanding in quiet, which significantly correlated with the duration of deafness in the ipsilateral ear. Specifically, instances of asymmetrical contralateral interference could correspond to asymmetrical hearing histories. Conclusions: Bilateral cochlear implantation should provide a hearing benefit to the recipient. For the BI-CI listeners specifically recruited for this study, there seems to be a conflict with processing the auditory information across the two ears, which produced the opposite of the desired hearing benefit. This suggests that there may be a subset of potential BI-CI users for whom contralateral interference offsets much of the potential head-shadow benefit. If so, earlier implantation in the second implanted ear might have produced larger binaural benefits, which is important information for clinicians advising patients considering bilateral implantation.

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A Comparison of Distortion Product Otoacoustic Emission Properties in Ménière’s Disease Patients and Normal-Hearing Participants

Objectives: Postmortem examination of temporal bones of Ménière’s disease patients consistently show dilated endolymphatic spaces of the inner ear, for which the term endolymphatic hydrops has been coined. During the past decade, magnetic resonance imaging techniques for the inner ear appeared, advancing the diagnosis of Ménière’s disease. They require, however, a field-strength of at least 3 T, are costly and not universally available. Alternative, noninvasive, cost-effective tests with high sensitivity and specifity for endolymphatic hydrops are desirable. In this study, we test the suitability of distortion product otoacoustic emissions (DPOAEs) for endolymphatic hydrops detection. Previous measurements of the commonly recorded cubic DPOAEs mainly register cochlear hearing loss and are not specific for Ménière’s disease. Simultaneous recordings of cubic and quadratic DPOAEs might be more suitable to detect endolymphatic hydrops, because both DPOAE orders react differently to changes of the cochlear operating point as they might occur in Ménière’s disease patients. Design: Cubic and quadratic DPOAEs were recorded in normal-hearing participants (N = 45) and in the affected and unaffected ears of patients with a diagnosis of definite Ménière’s disease (N = 32). First, to assess the integrity of DPOAE-generating mechanisms, cubic DPOAE-grams were obtained with primary tone frequencies f2 between 1 and 8 kHz with primary tone levels l1 = 60 dB SPL and l2 = 50 dB SPL, and a fixed primary tone frequency ratio of 1.22. Then, cubic and quadratic DPOAEs were simultaneously recorded with primary tone levels l1 = l2 = 65 dB SPL and at primary tone frequencies f2 = 4 and 5 kHz, where f1 was successively varied such that the ratio f2/f1 ranged between 1.1 and 1.6 in 0.04 steps while quadratic and cubic DPOAE levels were extracted from the same recording. Results: Cubic DPOAEs were significantly reduced in the affected ears of Ménière’s disease patients, and slightly reduced in the unaffected ears of Ménière’s disease patients, relative to the ears of normal-hearing participants. In contrast, no significant changes could be seen in quadratic DPOAEs across the ears of normal-hearing participants and Ménière’s disease patients. Conclusions: We could identify a relatively good preservation of quadratic DPOAE levels in relation to a reduction of cubic DPOAE levels as a potential noninvasive diagnostic approach in the early stage of suspected Ménière’s disease. Future studies validating the differential diagnostic power of this parameter in control groups with nonhydropic forms of hearing loss are warranted.

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Cortical Processing of Vocal and Nonvocal Sounds in Cochlear-Implanted Children: An Electrophysiological Study

Objectives: For prelingually deaf children, cochlear implants (CIs) can restore auditory input to the auditory cortex and the ability to acquire spoken language. Language development is strongly intertwined with voice perception. The aim of this electrophysiological study was to investigate human voice processing using measures of cortical auditory evoked potentials (AEPs) in pediatric CI users. Design: Cortical AEPs were measured in 8 CI children (4 to 12 years old) with good auditory and language performance and 8 normal-hearing (NH) age-matched controls. The auditory stimuli were nonspeech vocal sounds (laughing, sighing, coughing) and environmental sounds (e.g., telephones, alarms, cars, bells, water, wind). Independent component analysis was used to minimize the CI artifact in cortical AEPs. Results: Fronto-temporal positivity to vocal sounds was found in NH children, with a significant effect in the 140 to 240 msec latency range. In CI children, there was a positive response to vocal sounds in the 170 to 250 msec latency range, with a more diffuse and anterior distribution than in the NH children. Conclusions: Cortical responses to vocal sounds were recorded in CI children. The topography and latency of response to voice differed from that of NH children. The results suggest that cortical reorganization for processing vocal sounds may occur in congenitally deaf children fitted with a CI.

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Auditory Cognitive Training for Pediatric Cochlear Implant Recipients

Objectives: Understanding speech in noise is the biggest challenge faced by individuals with cochlear implants (CIs). Improving speech-in-noise perception for pediatric CI recipients continues to remain a high priority for all stakeholders. This study was designed to investigate the efficacy of working memory training for improving speech-in-noise recognition for children with CIs. Design: Fourteen children with CIs (aged 6 to 15 years) received adaptive, home-based training on forward digit span task for 5 weeks, while 13 children with CIs participated in backward digit span training. Seventeen age-matched children with CIs in the control group received a placebo. Outcome measures included forward and backward digit span and speech recognition threshold for digit triplets in noise at pretraining, post-training, and 5-week follow-up. Performance measures from 26 age-matched children with normal hearing were also obtained only at the baseline session to generate normative standards for comparison. Results: Digit span scores were significantly higher at post-training and follow-up compared with pretraining for both forward- and backward-trained groups. Both trained groups showed similar training-induced shifts. The control group showed no such improvement. There was no effect of training on the speech recognition threshold. Children with CIs showed significantly lower digit span scores and a higher speech recognition threshold relative to children with normal hearing. Conclusions: Training improves working memory capacity as measured by digit spans for children with CIs. Training-induced improvements are stable for at least 5 weeks. Learning effects demonstrate near-transfer, from forward to backward digit span and vice versa, but failed to show far-transfer to speech-in-noise recognition. Current evidence is not supportive of cognitive training for improving speech-in-noise performance for children with CIs.

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Same or Different: The Overlap Between Children With Auditory Processing Disorders and Children With Other Developmental Disorders A Systematic Review

Objectives: Children diagnosed with auditory processing disorders (APD) experience difficulties in auditory functioning and with memory, attention, language, and reading tasks. However, it is not clear whether the behavioral characteristics of these children are distinctive from the behavioral characteristics of children diagnosed with a different developmental disorder, such as specific language impairment (SLI), dyslexia, attention-deficit hyperactivity disorder (ADHD), learning disorder (LD), or autism spectrum disorder. This study describes the performance of children diagnosed with APD, SLI, dyslexia, ADHD, and LD to different outcome measurements. The aim of this study was to determine (1) which characteristics of APD overlap with the characteristics of children with SLI, dyslexia, ADHD, LD, or autism spectrum disorder; and (2) if there are characteristics that distinguish children diagnosed with APD from children diagnosed with other developmental disorders. Design: A systematic review. Six electronic databases (Pubmed, CINAHL, Eric, PsychINFO, Communication & Mass Media Complete, and EMBASE) were searched to find peer-reviewed studies from 1954 to May 2015. The authors included studies reporting behaviors and performance of children with (suspected) APD and children diagnosed with a different developmental disorder (SLI, Dyslexia, ADHD, and LD). Two researchers identified and screened the studies independently. Methodological quality of the included studies was assessed with the American Speech-Language-Hearing Association’s levels-of-evidence scheme. Results: In total, 13 studies of which the methodological quality was moderate were included in this systematic review. In five studies, the performance of children diagnosed with APD was compared with the performance of children diagnosed with SLI: in two with children diagnosed with dyslexia, one with children diagnosed with ADHD, and in another one with children diagnosed with LD. Ten of the studies included children who met the criteria for more than one diagnosis. In four studies, there was a comparison made between the performances of children with comorbid disorders. There were no studies found in which the performance of children diagnosed with APD was compared with the performance of children diagnosed with autism spectrum disorder. Children diagnosed with APD broadly share the same characteristics as children diagnosed with other developmental disorders, with only minor differences between them. Differences were determined with the auditory and visual Duration Pattern Test, the Children’s Auditory Processing Performance Scale questionnaire, and the subtests of the Listening in Spatialized Noise-Sentences test, in which noise is spatially separated from target sentences. However, these differences are not consistent between studies and are not found in comparison to all groups of children with other developmental disorders. Conclusions: Children diagnosed with APD perform equally to children diagnosed with SLI, dyslexia, ADHD, and LD on tests of intelligence, memory or attention, and language tests. Only small differences between groups were found for sensory and perceptual functioning tasks (auditory and visual). In addition, children diagnosed with dyslexia performed poorer in reading tasks compared with children diagnosed with APD. The result is possibly confounded by poor quality of the research studies and the low quality of the used outcome measures. More research with higher scientific rigor is required to better understand the differences and similarities in children with various neurodevelopmental disorders.

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Spectral-Temporal Modulated Ripple Discrimination by Children With Cochlear Implants

Objectives: A postlingually implanted adult typically develops hearing with an intact auditory system, followed by periods of deafness (or near deafness) and adaptation to the implant. For an early implanted child whose brain is highly plastic, the auditory system matures with consistent input from a cochlear implant. It is likely that the auditory system of early implanted cochlear implant users is fundamentally different than postlingually implanted adults. The purpose of this study is to compare the basic psychophysical capabilities and limitations of these two populations on a spectral resolution task to determine potential effects of early deprivation and plasticity. Design: Performance on a spectral resolution task (Spectral-temporally Modulated Ripple Test [SMRT]) was measured for 20 bilaterally implanted, prelingually deafened children (between 5 and 13 years of age) and 20 hearing children within the same age range. Additionally, 15 bilaterally implanted, postlingually deafened adults, and 10 hearing adults were tested on the same task. Cochlear implant users (adults and children) were tested bilaterally, and with each ear alone. Hearing listeners (adults and children) were tested with the unprocessed SMRT and with a vocoded version that simulates an 8-channel cochlear implant. Results: For children with normal hearing, a positive correlation was found between age and SMRT score for both the unprocessed and vocoded versions. Older hearing children performed similarly to hearing adults in both the unprocessed and vocoded test conditions. However, for children with cochlear implants, no significant relationship was found between SMRT score and chronological age, age at implantation, or years of implant experience. Performance by children with cochlear implants was poorer than performance by cochlear implanted adults. It was also found that children implanted sequentially tended to have better scores with the first implant compared with the second implant. This difference was not observed for adults. An additional finding was that SMRT score was negatively correlated with age for adults with implants. Conclusions: Results from this study suggest that basic psychophysical capabilities of early implanted children and postlingually implanted adults differ when assessed in the sound field using their personal implant processors. Because spectral resolution does not improve with age for early implanted children, it seems likely that the sparse representation of the signal provided by a cochlear implant limits spectral resolution development. These results are supported by the finding that postlingually implanted adults, whose auditory systems matured before the onset of hearing loss, perform significantly better than early implanted children on the spectral resolution test.

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Pure-Tone Masking Patterns for Monopolar and Phantom Electrical Stimulation in Cochlear Implants

Objectives: Monopolar stimulation of the most apical electrode produces the lowest pitch sensation in cochlear implants clinically. A phantom electrode that uses out-of-phase electrical stimulation between the most apical and the neighboring basal electrode can produce a lower pitch sensation than that associated with the most apical electrode. However, because of the absence of contacts beyond the apical tip of the array, the ability to assess the spread of electrical excitation associated with phantom stimulation is limited in the typical cochlear implant subject with no residual hearing. In the present study, the spread of electrical excitation associated with monopolar and phantom stimulation of the most apical electrode was assessed using electrical masking of acoustic thresholds in cochlear implant subjects with residual, low-frequency, acoustic hearing. Design: Eight subjects with an Advanced Bionics cochlear implant and residual hearing in the implanted ear participated in this study (nine ears in total). Unmasked and masked thresholds for acoustic pure tones were measured at 125, 250, 500, 750, 1000, and 2000 Hz in the presence of monopolar and phantom electrode stimulation presented at the apical-most end of the array. The current compensation for phantom electrode stimulation was fixed at 50%. The two electrical maskers were loudness balanced. Differences between the unmasked and masked acoustic thresholds can be attributed to (1) the electrical stimulus–induced interference in the transduction/conduction of the acoustic signal through cochlear periphery and the auditory nerve and/or (2) masking at the level of the central auditory system. Results: The results show a significant elevation in pure-tone thresholds in the presence of the monopolar and phantom electrical maskers. The unmasked thresholds were subtracted from the masked thresholds to derive masking patterns as a function of the acoustic probe frequency. The masking patterns show that phantom stimulation was able to produce more masking than that associated with the monopolar stimulation of the most apical electrode. Conclusion: These results suggest that for some cochlear implant subjects, phantom electrode stimulation can shift the neural stimulation pattern more apically in the cochlea, which is consistent with reports that phantom electrode stimulation produces lower pitch sensations than those associated with monopolar stimulation of the most apical electrode alone.

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Chirp-Evoked Otoacoustic Emissions and Middle Ear Absorbance for Monitoring Ototoxicity in Cystic Fibrosis Patients

Objectives: The goal of this study was to investigate the use of transient-evoked otoacoustic emissions (TEOAEs) and middle ear absorbance measurements to monitor auditory function in patients with cystic fibrosis (CF) receiving ototoxic medications. TEOAEs were elicited with a chirp stimulus using an extended bandwidth (0.71 to 8 kHz) to measure cochlear function at higher frequencies than traditional TEOAEs. Absorbance over a wide bandwidth (0.25 to 8 kHz) provides information on middle ear function. The combination of these time-efficient measurements has the potential to identify early signs of ototoxic hearing loss. Design: A longitudinal study design was used to monitor the hearing of 91 patients with CF (median age = 25 years; age range = 15 to 63 years) who received known ototoxic medications (e.g., tobramycin) to prevent or treat bacterial lung infections. Results were compared to 37 normally hearing young adults (median age = 32.5 years; age range = 18 to 65 years) without a history of CF or similar treatments. Clinical testing included 226-Hz tympanometry, pure-tone air-conduction threshold testing from 0.25 to 16 kHz and bone conduction from 0.25 to 4 kHz. Experimental testing included wideband absorbance at ambient and tympanometric peak pressure and TEOAEs in three stimulus conditions: at ambient pressure and at tympanometric peak pressure using a chirp stimulus with constant incident pressure level across frequency and at ambient pressure using a chirp stimulus with constant absorbed sound power across frequency. Results: At the initial visit, behavioral audiometric results indicated that 76 of the 157 ears (48%) from patients with CF had normal hearing, whereas 81 of these ears (52%) had sensorineural hearing loss for at least one frequency. Seven ears from four patients had a confirmed behavioral change in hearing threshold for ≥3 visits during study participation. Receiver operating characteristic curve analyses demonstrated that all three TEOAE conditions were useful for distinguishing CF ears with normal hearing from ears with sensorineural hearing loss, with an area under the receiver operating characteristic curve values ranging from 0.78 to 0.92 across methods for frequency bands from 2.8 to 8 kHz. Case studies are presented to illustrate the relationship between changes in audiometric thresholds, TEOAEs, and absorbance across study visits. Absorbance measures permitted identification of potential middle ear dysfunction at 5.7 kHz in an ear that exhibited a temporary hearing loss. Conclusions: The joint use of TEOAEs and absorbance has the potential to explain fluctuations in audiometric thresholds due to changes in cochlear function, middle ear function, or both. These findings are encouraging for the joint use of TEOAE and wideband absorbance objective tests for monitoring ototoxicity, particularly, in patients who may be too ill for behavioral hearing tests. Additional longitudinal studies are needed in a larger number of CF patients receiving ototoxic drugs to further evaluate the clinical utility of these measures in an ototoxic monitoring program.

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Auditory and Audiovisual Close Shadowing in Post-Lingually Deaf Cochlear-Implanted Patients and Normal-Hearing Elderly Adults

Objectives: The goal of this study was to determine the effect of auditory deprivation and age-related speech decline on perceptuo–motor abilities during speech processing in post-lingually deaf cochlear-implanted participants and in normal-hearing elderly (NHE) participants. Design: A close-shadowing experiment was carried out on 10 cochlear-implanted patients and on 10 NHE participants, with two groups of normal-hearing young participants as controls. To this end, participants had to categorize auditory and audiovisual syllables as quickly as possible, either manually or orally. Reaction times and percentages of correct responses were compared depending on response modes, stimulus modalities, and syllables. Results: Responses of cochlear-implanted subjects were globally slower and less accurate than those of both young and elderly normal-hearing people. Adding the visual modality was found to enhance performance for cochlear-implanted patients, whereas no significant effect was obtained for the NHE group. Critically, oral responses were faster than manual ones for all groups. In addition, for NHE participants, manual responses were more accurate than oral responses, as was the case for normal-hearing young participants when presented with noisy speech stimuli. Conclusions: Faster reaction times were observed for oral than for manual responses in all groups, suggesting that perceptuo–motor relationships were somewhat successfully functional after cochlear implantation and remain efficient in the NHE group. These results are in agreement with recent perceptuo–motor theories of speech perception. They are also supported by the theoretical assumption that implicit motor knowledge and motor representations partly constrain auditory speech processing. In this framework, oral responses would have been generated at an earlier stage of a sensorimotor loop, whereas manual responses would appear late, leading to slower but more accurate responses. The difference between oral and manual responses suggests that the perceptuo–motor loop is still effective for NHE subjects and also for cochlear-implanted participants, despite degraded global performance.

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High-Frequency Distortion-Product Otoacoustic Emission Repeatability in a Patient Population

Objectives: Distortion-product otoacoustic emissions (DPOAEs) are repeatable over time at lower frequencies (≤8 kHz) and higher frequencies (>8 kHz) in healthy, normal-hearing subjects. The purpose of this study was to examine the repeatability of DPOAEs measured with high-frequency (HF) stimuli in a patient population. It was hypothesized that HF DPOAEs would be repeatable over four trials. Design: DPOAEs were measured in 40 cystic fibrosis (CF) patients (17 females and 23 males) with measurable behavioral thresholds and present DPOAEs for at least 2 of the high frequencies tested (8 to 16 kHz). A depth-compensated simulator sound pressure level (SPL) method of calibration was utilized. Each patient attended four trials, in which a complete set of data were collected. At each trial, three different DPOAE paradigms were completed. First, a discrete frequency sweep was measured between 8 and 16 kHz with a ratio (f2/f1) of 1.2 and levels of 65/50 dB SPL for L1/L2. Next, ratio and level sweeps were obtained at the two highest frequencies with a present DPOAE determined from the discrete frequency sweep, and the results were used to calculate DPOAE group delay and DPOAE detection thresholds, respectively. Ratio sweeps were collected with f2/f1 varied from 1.1 to 1.3 and stimulus levels of 60/45 dB SPL (L1/L2). Level sweeps were collected with an f2/f1 of 1.22 and L2 = 50 and L1 varied between 20 and 70 dB SPL. Differences and correlations between trials, SE of the measurement, and confidence intervals were calculated, as well as a repeated-measures analysis of variance. Results: DPOAE response and behavioral threshold variability in CF patients were not significantly different across four trials. It can be expected in 95% of CF patients that differences between trials of DPOAE levels, group delay, and detection thresholds and behavioral thresholds are less than 6.26 dB, 0.87 msec, 9.34 dB, and 9.60 dB, respectively. Conclusions: HF DPOAEs were repeatable across four test trials for all three paradigms measured in a group of CF patients. These results are encouraging for the measurement of HF DPOAEs to be monitored in those exposed to ototoxic agents.

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Auditory Acclimatization to Bilateral Hearing Aids: Effects on Sentence-in-Noise Processing Times and Speech-Evoked Potentials

Objectives: Using a longitudinal design, the present study sought to substantiate indications from two previous cross-sectional studies that hearing aid (HA) experience leads to improved speech processing abilities as quantified using eye-gaze measurements. Another aim was to explore potential concomitant changes in event-related potentials (ERPs) to speech stimuli. Design: Groups of elderly novice (novHA) and experienced (expHA) HA users matched in terms of age and working memory capacity participated. The novHA users were acclimatized to bilateral HA fittings for up to 24 weeks. The expHA users continued to use their own HAs during the same period. The participants’ speech processing abilities were assessed after 0 weeks (novHA: N = 16; expHA: N = 14), 12 weeks (novHA: N = 16; expHA: N = 14), and 24 weeks (N = 10 each). To that end, an eye-tracking paradigm was used for estimating how quickly the participants could grasp the meaning of sentences presented against background noise together with two similar pictures that either correctly or incorrectly depicted the meaning conveyed by the sentences (the “processing time”). Additionally, ERPs were measured with an active oddball paradigm requiring the participants to categorize word stimuli as living (targets) or nonliving (nontargets) entities. For all measurements, the stimuli were spectrally shaped according to individual real-ear insertion gains and presented via earphones. Results: Concerning the processing times, no changes across time were found for the expHA group. After 0 weeks of HA use, the novHA group had significantly longer (poorer) processing times than the expHA group, consistent with previous findings. After 24 weeks, a significant mean improvement of ~30% was observed for the novHA users, leading to a performance comparable with that of the expHA group. Concerning the ERPs, no changes across time were found. Conclusions: The results from this exploratory study are consistent with the view that auditory acclimatization to HAs positively impacts speech comprehension in noise. Further research is needed to substantiate them.

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Linguistic Context Versus Semantic Competition in Word Recognition by Younger and Older Adults With Cochlear Implants

Objectives: The increasing numbers of older adults now receiving cochlear implants raises the question of how the novel signal produced by cochlear implants may interact with cognitive aging in the recognition of words heard spoken within a linguistic context. The objective of this study was to pit the facilitative effects of a constraining linguistic context against a potential age-sensitive negative effect of response competition on effectiveness of word recognition. Design: Younger (n = 8; mean age = 22.5 years) and older (n = 8; mean age = 67.5 years) adult implant recipients heard 20 target words as the final words in sentences that manipulated the target word’s probability of occurrence within the sentence context. Data from published norms were also used to measure response entropy, calculated as the total number of different responses and the probability distribution of the responses suggested by the sentence context. Sentence-final words were presented to participants using a word-onset gating paradigm, in which a target word was presented with increasing amounts of its onset duration in 50 msec increments until the word was correctly identified. Results: Results showed that for both younger and older adult implant users, the amount of word-onset information needed for correct recognition of sentence-final words was inversely proportional to their likelihood of occurrence within the sentence context, with older adults gaining differential advantage from the contextual constraints offered by a sentence context. On the negative side, older adults’ word recognition was differentially hampered by high response entropy, with this effect being driven primarily by the number of competing responses that might also fit the sentence context. Conclusions: Consistent with previous research with normal-hearing younger and older adults, the present results showed older adult implant users’ recognition of spoken words to be highly sensitive to linguistic context. This sensitivity, however, also resulted in a greater degree of interference from other words that might also be activated by the context, with negative effects on ease of word recognition. These results are consistent with an age-related inhibition deficit extending to the domain of semantic constraints on word recognition.

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Deaf Culture: Exploring Deaf Communities in the United States

No abstract available

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[The relationship between the recurrence of benign paroxysmal positional vertigo and the level of bone mineral as well as estrogen in postmenopausal women].

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[The relationship between the recurrence of benign paroxysmal positional vertigo and the level of bone mineral as well as estrogen in postmenopausal women].

Zhonghua Er Bi Yan Hou Tou Jing Wai Ke Za Zhi. 2017 Dec 07;52(12):881-884

Authors: Zhang YM, Yang ZD, Li WX, Shi C, Yu YF

Abstract
Objective: To investigate the relationship between the recurrence of benign paroxysmal positional vertigo(BPPV) and the levels of bone mineral density(BMD) and estrogen in postmenopausal women. Methods: A total of 38 postmenopausal women with recurrent BPPV were recruited as study group, in the First Affiliated Hospital of Soochow University from December 2013 to June 2017. Meanwhile, 49 normal menopausal women were included as control. All patients were natural menopausal for over one year.The patients were diagnosed as BPPV based on results of Dix-Hallpike test and Roll-test, with at least two episodes of recurrent onset. In the subjects, BMD was measured by dual X-ray absorptiometry of lumbar vertebrae. Estrogen levels were obtained by testing serum estradiol (E2) levels in early morning fasting venous blood. In the present study, we compared the level of E2 and the value of BMD in two groups by SPSS 21.0. In the study group, patients with decreased BMD were divided into two groups: treatment and untreated group. The recurrence rate of BPPV was compared between the two groups within 12 months. Results: ①The averagel levels of E2 and BMD in the study group were (16.21±11.00)ng/L and -1.68±0.98) respectively, which were significantly lower than those in the control group (t value was 7.03 and 8.05 respectively, both P<0.05). The averagel levels of E2 and BMD incontrol group were(28.52±6.34)ng/L and -0.18±0.77 respectively. ②The number of patients with decreased BMD in the study group (30 cases) was more than that in control group (6 cases), and the difference was statistically significant (P<0.05). ③ The recurrence rate of BPPV in treatment group [17.6%(3/17)] was significantly lower than that of untreated group [61.5%(8/13)], and the difference was statistically significant (P<0.05). Conclusion: Recurrent BPPV in postmenopausal women usually accompany with low levels of estrogen and BMD. Active treatment is helpful for their recurrence of BPPV.

PMID: 29262443 [PubMed - in process]

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Innovative gene editing method may prevent deafness

In many cases, hearing loss is hereditary and caused by genetic mutations. Experiments with novel gene editing methods may lead to a preventive treatment.

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Clinical trials, ototoxicity grading scales and the audiologist’s role in therapeutic decision making

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Evaluation of otoscopy simulation as a training tool for real-time remote otoscopy

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Acoustic reflexes are common but not pervasive: evidence using a diagnostic middle ear analyser

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Delayed diagnosis of a patient with Usher syndrome 1C in a Louisiana Acadian family highlights the necessity of timely genetic testing for the diagnosis and management of congenital hearing loss.

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Delayed diagnosis of a patient with Usher syndrome 1C in a Louisiana Acadian family highlights the necessity of timely genetic testing for the diagnosis and management of congenital hearing loss.

SAGE Open Med Case Rep. 2017;5:2050313X17745904

Authors: Umrigar A, Musso A, Mercer D, Hurley A, Glausier C, Bakeer M, Marble M, Hicks C, Tsien F

Abstract
Advances in sequencing technologies and increased understanding of the contribution of genetics to congenital sensorineural hearing loss have led to vastly improved outcomes for patients and their families. Next-generation sequencing and diagnostic panels have become increasingly reliable and less expensive for clinical use. Despite these developments, the diagnosis of genetic sensorineural hearing loss still presents challenges for healthcare providers. Inherited sensorineural hearing loss has high levels of genetic heterogeneity and variable expressivity. Additionally, syndromic hearing loss (hearing loss and additional clinical abnormalities) should be distinguished from non-syndromic (hearing loss is the only clinical symptom). Although the diagnosis of genetic sensorineural hearing loss can be challenging, the patient's family history and ethnicity may provide critical information, as certain genetic mutations are more common in specific ethnic populations. The early identification of the cause of deafness can benefit patients and their families by estimating recurrence risks for future family planning and offering the proper interventions to improve their quality of life. Collaboration between pediatricians, audiologists, otolaryngologists, geneticists, and other specialists are essential in the diagnosis and management of patients with hearing disorders. An early diagnosis is vital for proper management and care, as some clinical manifestations of syndromic sensorineural hearing loss are not apparent at birth and have a delayed age of onset. We present a case of Usher syndrome (congenital deafness and childhood-onset blindness) illustrating the challenges encountered in the diagnosis and management of children presenting with congenital genetic sensorineural hearing loss, along with helpful resources for clinicians and families.

PMID: 29276601 [PubMed]

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The cost and utilisation patterns of a pilot sign language interpreter service for primary health care services in South Africa.

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The cost and utilisation patterns of a pilot sign language interpreter service for primary health care services in South Africa.

PLoS One. 2017;12(12):e0189983

Authors: Zulu T, Heap M, Sinanovic E

Abstract
BACKGROUND: The World Health Organisation estimates disabling hearing loss to be around 5.3%, while a study of hearing impairment and auditory pathology in Limpopo, South Africa found a prevalence of nearly 9%. Although Sign Language Interpreters (SLIs) improve the communication challenges in health care, they are unaffordable for many signing Deaf people and people with disabling hearing loss. On the other hand, there are no legal provisions in place to ensure the provision of SLIs in the health sector in most countries including South Africa. To advocate for funding of such initiatives, reliable cost estimates are essential and such data is scarce. To bridge this gap, this study estimated the costs of providing such a service within a South African District health service based on estimates obtained from a pilot-project that initiated the first South African Sign Language Interpreter (SASLI) service in health-care.
METHODS: The ingredients method was used to calculate the unit cost per SASLI-assisted visit from a provider perspective. The unit costs per SASLI-assisted visit were then used in estimating the costs of scaling up this service to the District Health Services. The average annual SASLI utilisation rate per person was calculated on Stata v.12 using the projects' registry from 2008-2013. Sensitivity analyses were carried out to determine the effect of changing the discount rate and personnel costs.
RESULTS: Average Sign Language Interpreter services' utilisation rates increased from 1.66 to 3.58 per person per year, with a median of 2 visits, from 2008-2013. The cost per visit was US$189.38 in 2013 whilst the estimated costs of scaling up this service ranged from US$14.2million to US$76.5million in the Cape Metropole District. These cost estimates represented 2.3%-12.2% of the budget for the Western Cape District Health Services for 2013.
CONCLUSIONS: In the presence of Sign Language Interpreters, Deaf Sign language users utilise health care service to a similar extent as the hearing population. However, this service requires significant capital investment by government to enable access to healthcare for the Deaf.

PMID: 29272272 [PubMed - in process]

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A new association between CDK5RAP2 microcephaly and congenital cataracts.

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A new association between CDK5RAP2 microcephaly and congenital cataracts.

Ann Hum Genet. 2017 Dec 22;:

Authors: Alfares A, Alhufayti I, Alsubaie L, Alowain M, Almass R, Alfadhel M, Kaya N, Eyaid W

Abstract
INTRODUCTION: Primary microcephaly type 3 is a genetically heterogeneous condition caused by a homozygous or compound heterozygous mutation in CDK5 regulatory subunit associated protein 2 (CDK5RAP2) and characterized by reduced head circumference (<5th percentile) with additional phenotypes varying from pigmentary abnormalities to sensorineural hearing loss. Until now, congenital cataracts have not been reported in patients with primary microcephaly type 3.
CLINICAL REPORT: We report multiple affected family members from a consanguineous Saudi family with microcephaly and congenital cataracts. We utilized a next-generation sequencing-based microcephaly gene panel that revealed a CDK5RAP2 variant (c.4055A>G; p.Glu1352Gly) as the most plausible candidate for the likely etiology in this family. Then we performed family segregation analysis using Sanger sequencing, autozygosity mapping, and whole exome sequencing, all of which revealed no other possible disease-causing variants.
CONCLUSION: Here we report on a new clinical manifestation of CDK5RAP2 and expand the phenotype of primary microcephaly type 3.

PMID: 29271474 [PubMed - as supplied by publisher]

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Natural History and Genotype-Phenotype Correlation in Female X-Linked Alport Syndrome.

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Natural History and Genotype-Phenotype Correlation in Female X-Linked Alport Syndrome.

Kidney Int Rep. 2017 Sep;2(5):850-855

Authors: Yamamura T, Nozu K, Fu XJ, Nozu Y, Ye MJ, Shono A, Yamanouchi S, Minamikawa S, Morisada N, Nakanishi K, Shima Y, Yoshikawa N, Ninchoji T, Morioka I, Kaito H, Iijima K

Abstract
Introduction: X-linked Alport syndrome (XLAS) is a hereditary disease characterized by progressive nephritis, hearing loss, and ocular abnormalities. Affected male patients usually progress to end-stage renal disease in early or middle adulthood, and disease severity is strongly correlated with genotype. However, the clinical course in female patients has rarely been reported.
Methods: We conducted a retrospective analysis of females with genetically proven XLAS (n = 275) and their affected female family members (n = 61) from 179 Japanese families. Patients suspected to have Alport syndrome from pathologic findings or a family history who were referred from anywhere in Japan for genetic diagnosis between 2006-2015 were included in this study. Clinical and laboratory data were collected from medical records at the time of registration for genetic analysis.
Results: Proteinuria was detected in 175 genetically proven patients (72.6%), and the median age for developing proteinuria was 7.0 years. Fifty-two of 336 patients developed end-stage renal disease with a median renal survival age of 65.0 years. No obvious genotype-phenotype correlation was observed. Additionally, targeted sequencing for podocyte-related genes in patients with severe phenotypes revealed no obvious variants considered to be modifier genes except for 1 patient with a COL4A3 gene variant.
Discussion: This study revealed that phenotypes in female XLAS patients may be severe, but genotype does not help to predict the disease severity. Clinicians must therefore pay careful attention to the clinical course and appropriate treatment in females with XLAS.

PMID: 29270492 [PubMed]

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Common founder effects of hereditary hemochromatosis, Wilson´s disease, the long QT syndrome and autosomal recessive deafness caused by two novel mutations in the WHRN and TMC1 genes.

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Common founder effects of hereditary hemochromatosis, Wilson´s disease, the long QT syndrome and autosomal recessive deafness caused by two novel mutations in the WHRN and TMC1 genes.

Hereditas. 2017;154:16

Authors: Olsson KS, Wålinder O, Jansson U, Wilbe M, Bondeson ML, Stattin EL, Raha-Chowdhury R, Williams R

Abstract
Background: Genealogy and molecular genetic studies of a Swedish river valley population resulted in a large pedigree, showing that the hereditary hemochromatosis (HH) HFE/p.C282Y mutation is inherited with other recessive disorders such as Wilson´s disease (WND), a rare recessive disorder of copper overload. The population also contain individuals with the Swedish long QT syndrome (LQTS1) founder mutation (KCNQ1/p.Y111C) which in homozygotes causes the Jervell & Lange Nielsen syndrome (JLNS) and hearing loss (HL).Aims of the study were to test whether the Swedish long QT founder mutation originated in an ancestral HFE family and if carriers had an increased risk for hemochromatosis (HH), a treatable disorder. We also aimed to identify the pathogenic mutation causing the hearing loss disorder segregating in the pedigree.
Methods: LQTS patients were asked about their ancestry and possible origin in a HH family. They were also offered a predictive testing for the HFE genotype. Church books were screened for families with hearing loss. One HH family had two members with hearing loss, who underwent molecular genetic analysis of the LQTS founder mutation, connexin 26 and thereafter exome sequencing. Another family with hearing loss in repeat generations was also analyzed for connexin 26 and underwent exome sequencing.
Results: Of nine LQTS patients studied, four carried a HFE mutation (two p.C282Y, two p.H63D), none was homozygous. Three LQTS patients confirmed origin in a female founder ( b 1694, identical to AJ b 1694, a HFE pedigree member from the Fax river. Her descent of 44 HH families, included also 29 families with hearing loss (HL) suggesting JLNS. Eleven LQTS probands confirmed origin in a second founder couple (b 1614/1605) in which the woman b 1605 was identical to a HFE pedigree member from the Fjällsjö river. In her descent there were not only 64 HH, six WND families, one JLNS, but also 48 hearing loss families. Most hearing loss was non syndromic and caused by founder effects of the late 16th century. One was of Swedish origin carrying the WHRN, c.1977delC, (p.S660Afs*30) mutation, the other was a TMC1(NM_138691),c.1814T>C,(p.L605P) mutation, possibly of Finnish origin.
Conclusions: Deep human HFE genealogies show HFE to be associated with other genetic disorders like Wilson´s disease, LQTS, JLNS, and autosomal recessive hearing loss. Two new homozygous HL mutations in WHRN/p.S660Afs*30 and TMC1/p.L605P were identified,none of them previously reported from Scandinavia. The rarity of JLNS was possibly caused by miscarriage or intrauterine death. Most hearing loss (81.7%) was seen after 1844 when first cousin marriages were permitted. However, only 10 (10.3%) came from 1st cousin unions and only 2 (2.0 %) was born out of wedlock.

PMID: 29270100 [PubMed - in process]

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Unique autosomal recessive variant of palmoplantar keratoderma associated with hearing loss not caused by known mutations.

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Unique autosomal recessive variant of palmoplantar keratoderma associated with hearing loss not caused by known mutations.

An Bras Dermatol. 2017;92(5 Suppl 1):154-158

Authors: Hegazi MA, Manou S, Sakr H, Camp GV

Abstract
Inherited Palmoplantar Keratodermas are rare disorders of genodermatosis that are conventionally regarded as autosomal dominant in inheritance with extensive clinical and genetic heterogeneity. This is the first report of a unique autosomal recessive Inherited Palmoplantar keratoderma -sensorineural hearing loss syndrome which has not been reported before in 3 siblings of a large consanguineous family. The patients presented unique clinical features that were different from other known Inherited Palmoplantar Keratodermas -hearing loss syndromes. Mutations in GJB2 or GJB6 and the mitochondrial A7445G mutation, known to be the major causes of diverse Inherited Palmoplantar Keratodermas -hearing loss syndromes were not detected by Sanger sequencing. Moreover, the pathogenic mutation could not be identified using whole exome sequencing. Other known Inherited Palmoplantar keratoderma syndromes were excluded based on both clinical criteria and genetic analysis.

PMID: 29267478 [PubMed - in process]

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GeneReviews®

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GeneReviews®

Book. 1993

Authors: Adam MP, Ardinger HH, Pagon RA, Wallace SE, Bean LJH, Mefford HC, Stephens K, Amemiya A, Ledbetter N

Abstract
CLINICAL CHARACTERISTICS: Pelizaeus-Merzbacher-like disease 1 (PMLD1) is a slowly progressive leukodystrophy that typically presents during the neonatal or early-infantile period with nystagmus, commonly associated with hypotonia, delayed acquisition of motor milestones, speech delay, and dysarthria. Over time the hypotonia typically evolves into spasticity that affects the ability to walk and communicate. Cerebellar signs (gait ataxia, dysmetria, intention tremor, head titubation, and dysdiadochokinesia) frequently manifest during childhood. Some individuals develop extrapyramidal movement abnormalities (choreoathetosis and dystonia). Hearing loss and optic atrophy are observed in rare cases. Motor impairments can lead to swallowing difficulty and orthopedic complications, including hip dislocation and scoliosis. Most individuals have normal cognitive skills or mild intellectual disability – which, however, can be difficult to evaluate in the context of profound motor impairment.
DIAGNOSIS/TESTING: The diagnosis of PMLD1 is established in a proband with suggestive clinical and neuroimaging findings and identification of biallelic pathogenic variants in GJC2 on molecular genetic testing.
MANAGEMENT: Treatment of manifestations: To date no definite treatment is available; treatment is mainly supportive and includes assuring adequate nutrition and providing standard treatment for developmental delay/cognitive impairment, neurologic complications (spasticity, ataxia, epilepsy, extrapyramidal movement disorders), communication difficulties, hearing loss, and visual impairment. Surveillance: Routine assessment of growth, weight gain, vision, and hearing. Routine monitoring of disease progression, spine for evidence of scoliosis and hips for evidence of dislocation, and needs related to physical therapy, communication, and swallowing/feeding.
GENETIC COUNSELING: PMLD1 is inherited in an autosomal recessive manner. At conception, each sib of an affected individual has a 25% chance of being affected, a 50% chance of being an asymptomatic carrier (heterozygote), and a 25% chance of being unaffected and not a carrier. Once the GJC2 pathogenic variants have been identified in an affected family member, carrier testing for at-risk relatives, prenatal testing for a pregnancy at increased risk, and preimplantation genetic diagnosis are possible.

PMID: 29276893

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Eosera Now Offers Earwax MD to Long-Term Care Facilities

​Eosera (https://earcaremd.com/), the maker of the topical cerumen dissolving drop Earwax MD, has established a partnership with nursing home and long-term care hospital health care product supplier VitalMedix to extend the availability of Earwax MD to long-term care facilities. Under this partnership, nursing homes and long-term care facilities in the United States will be able to purchase Earwax MD, which is already available in CVS nationwide, directly from VitalMedix to use in their offices starting this month. Elyse Dickerson, co-founder and CEO of Eosera, said hearing loss is common in the elderly, but it's largely avoidable. "Often, it is a direct result of severe earwax impaction, resulting from failure to keep the ears clean," Dickerson said. "In our ongoing conversations with health care professionals, we have identified this population as one that truly and urgently needs an effective solution to alleviate the discomfort related to earwax impaction, and they need it on premises." The dual-action Earwax MD solubilizes lipids and wax while simultaneously disrupting the sheets of keratinocytes that are continually shed from the ear canal. The product has won the company the 2017 Tech Fort Worth IMPACT Awards.​

Published: 12/22/2017 8:14:00 AM

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Visual Acuity and Cognition in Older Adults With and Without Hearing Loss: Evidence For Late-Life Sensory Compensation?

Objectives: Relationships between cognitive and sensory functioning become stronger with advancing age, and the debate on underlying mechanisms continues. Interestingly, the potential mechanism of compensation by the unaffected sensory modality has so far been investigated in younger age groups with congenital sensory impairment but not in older adults with late-life sensory loss. We compared associations between visual acuity and cognitive functioning in hearing-impaired older adults (HI), and sensory-unimpaired controls (UI). We expected stronger associations in the HI group as compared with the UI group. Design: Our study sample was drawn from the pools of outpatients from regional university clinics and city registries and consisted of n = 266 older adults (mean age = 82.45 years, SD = 4.76 years; HI: n = 116; UI: n = 150). For the assessment of cognitive performance, multiple established tests (e.g., subtests of the revised Wechsler Adult Intelligence Scale) were used. Moreover, objective visual acuity (distance vision) was assessed. Results: As expected, bivariate correlations between vision and cognitive abilities were stronger in the HI group compared with the UI group. In regression models controlling for age, sex, education, subjective health and number of chronic diseases, distance visual acuity was a significant predictor of general cognitive ability in the HI group only. Conclusions: Our findings suggest that visual acuity may play an important compensatory role for maintaining cognitive ability when hearing impairment sets in, which may reflect an adaptive process of late-life sensory compensation. ACKNOWLEDGMENTS: We want to thank PD Dr. Ingo Baumann, Prof. Dr. Hans Hörmann, Prof. Dr. Jost Jonas, Prof. Dr. Peter Plinkert, and Prof. Dr. Klaus Rohrschneider who provided outstanding support in generating the sensory impaired samples. Furthermore, we are very grateful for the support from the company KIND Hörgeräte which provided the audiometric assessment. We would like to thank our project staff and interviewers, particularly Nadine Langer and Christina Hunger, and our study participants who invested considerable time and energy to serve our research. We thank the German Research Foundation for supporting this research with a grant (WA 809/7–1) awarded to Hans-Werner Wahl. M.W. analyzed data for this publication and wrote the article. H.-W. W. and V. H. designed the study, were responsible for data collection, and provided critical revision and additions to all parts of the article. The authors have no conflicts of interest to disclose. Address for correspondence: Markus Wettstein, German Centre of Gerontology, Manfred-von-Richthofen-Straße 2, 12101 Berlin, Germany. E-mail: markus.wettstein@dza.de. Received October 27, 2016; accepted October 16, 2017. Copyright © 2017 Wolters Kluwer Health, Inc. All rights reserved.

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Evaluating the Performance of a Visually Guided Hearing Aid Using a Dynamic Auditory-Visual Word Congruence Task

Objectives: The “visually guided hearing aid” (VGHA), consisting of a beamforming microphone array steered by eye gaze, is an experimental device being tested for effectiveness in laboratory settings. Previous studies have found that beamforming without visual steering can provide significant benefits (relative to natural binaural listening) for speech identification in spatialized speech or noise maskers when sound sources are fixed in location. The aim of the present study was to evaluate the performance of the VGHA in listening conditions in which target speech could switch locations unpredictably, requiring visual steering of the beamforming. To address this aim, the present study tested an experimental simulation of the VGHA in a newly designed dynamic auditory-visual word congruence task. Design: Ten young normal-hearing (NH) and 11 young hearing-impaired (HI) adults participated. On each trial, three simultaneous spoken words were presented from three source positions (-30, 0, and 30o azimuth). An auditory-visual word congruence task was used in which participants indicated whether there was a match between the word printed on a screen at a location corresponding to the target source and the spoken target word presented acoustically from that location. Performance was compared for a natural binaural condition (stimuli presented using impulse responses measured on KEMAR), a simulated VGHA condition (BEAM), and a hybrid condition that combined lowpass-filtered KEMAR and highpass-filtered BEAM information (BEAMAR). In some blocks, the target remained fixed at one location across trials, and in other blocks, the target could transition in location between one trial and the next with a fixed but low probability. Results: Large individual variability in performance was observed. There were significant benefits for the hybrid BEAMAR condition relative to the KEMAR condition on average for both NH and HI groups when the targets were fixed. Although not apparent in the averaged data, some individuals showed BEAM benefits relative to KEMAR. Under dynamic conditions, BEAM and BEAMAR performance dropped significantly immediately following a target location transition. However, performance recovered by the second word in the sequence and was sustained until the next transition. Conclusions: When performance was assessed using an auditory-visual word congruence task, the benefits of beamforming reported previously were generally preserved under dynamic conditions in which the target source could move unpredictably from one location to another (i.e., performance recovered rapidly following source transitions) while the observer steered the beamforming via eye gaze, for both young NH and young HI groups. ACKNOWLEDGMENTS: E.R. helped to design the experiment, performed the experiment, analyzed/interpreted data, and wrote the article; V.B. assisted in interpretation of data and provided critical manuscript revision; C.R.M. assisted in interpretation of data and provided critical manuscript revision; T.S. helped to design and perform the experiment and assisted with analysis; G.K. designed the experiment, assisted in interpretation of data, and provided critical manuscript revision. The authors thank Lorraine Delhorne for her assistance with subject recruitment. Portions of this work were presented at the 39th meeting of the Association for Research in Otolaryngology in San Diego, CA, and at the 171st meetings of the Acoustical Society of American in Salt Lake City, UT. This work was supported by a grant from NIH/NIDCD (to G.K.) and by a grant from DoD/AFOSR (to G.K.). The authors report no conflicts of interest. Address for correspondence: Elin Roverud, Department of Speech, Language & Hearing Sciences, Boston University, 635 Commonwealth Avenue, Boston, MA 02215. E-mail: erover@bu.edu. Received April 25, 2017; accepted October 22, 2017. Copyright © 2017 Wolters Kluwer Health, Inc. All rights reserved.

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Social Development in Children With Early Cochlear Implants: Normative Comparisons and Predictive Factors, Including Bilateral Implantation

Objectives: There are mixed results regarding psychosocial development in children with cochlear implants (CIs) compared with children with normal hearing (NH), and the effect of bilateral CIs has not yet been investigated. This study aimed to investigate whether social development differed between NH children and those with early CIs, and to identify new predictors of psychosocial development in children with early CIs. Design: The psychosocial development, cognitive and language abilities of 159 children were measured as part of a longitudinal outcomes study of children with CIs. Parental involvement was also assessed. Results: The children in this study did not differ significantly from their peers with NH in terms of their psychosocial development, except with regard to Prosocial Behavior, with parents reporting significantly poorer development in this area. Having bilateral CIs predicted significantly fewer difficulties with psychosocial development, with earlier age at implantation increasing the effect size of the second CI for Emotional Symptoms and Peer Problems. Receptive language ability, higher parent education, later birth order, high parent involvement, and female gender predicted significantly fewer parent reports of psychosocial problems. Higher child cognitive ability and greater screen time predicted significantly greater reported psychosocial difficulties. Marginal effects modeling quantified the effects of changes in the predictive factors examined. Conclusions: Psychosocial development in these children with early CIs was found to be similar to that for peers with NH, with the exception of delayed Prosocial Behavior development. Having bilateral CIs predicted significantly better emotional outcomes. A number of new predictive factors were found for psychosocial development. The results of the marginal effects modeling should be shared with parents and professionals to increase their understanding of the impact of changes in predictive factors, particularly in terms of their own contribution to psychosocial outcomes for children with CIs. ACKNOWLEDGMENTS: The authors express their gratitude to the participating children and their families. The authors thank their collaborative partners: The Melbourne Cochlear Implant Clinic at the Royal Victorian Eye and Ear Hospital, The Shepherd Centre, The Hear and Say Centre, The Cora Barclay Centre, The Sydney Cochlear Implant Centre, The Hearing House in New Zealand and the Audiology Department at the Adelaide Women’s and Children’s Hospital. The authors thank the LOCHI study for sharing cognitive and language data for overlapping participants. This work was supported by an Australian Research Council Linkage Grant (Grant Number LP0989391), and Cochlear Ltd. All authors except Peter Busby, who is an employee of Cochlear Ltd, declare no conflict of interest. Address for correspondence: Julia Sarant, PhD, Department of Audiology and Speech Pathology, The University of Melbourne, 550 Swanston Street, Victoria 3010, Australia. E-mail: jsarant@unimelb.edu.au. Received September 17, 2016; accepted October 17, 2017. Copyright © 2017 Wolters Kluwer Health, Inc. All rights reserved.

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Looking Behavior and Audiovisual Speech Understanding in Children With Normal Hearing and Children With Mild Bilateral or Unilateral Hearing Loss

Objectives: Visual information from talkers facilitates speech intelligibility for listeners when audibility is challenged by environmental noise and hearing loss. Less is known about how listeners actively process and attend to visual information from different talkers in complex multi-talker environments. This study tracked looking behavior in children with normal hearing (NH), mild bilateral hearing loss (MBHL), and unilateral hearing loss (UHL) in a complex multi-talker environment to examine the extent to which children look at talkers and whether looking patterns relate to performance on a speech-understanding task. It was hypothesized that performance would decrease as perceptual complexity increased and that children with hearing loss would perform more poorly than their peers with NH. Children with MBHL or UHL were expected to demonstrate greater attention to individual talkers during multi-talker exchanges, indicating that they were more likely to attempt to use visual information from talkers to assist in speech understanding in adverse acoustics. It also was of interest to examine whether MBHL, versus UHL, would differentially affect performance and looking behavior. Design: Eighteen children with NH, eight children with MBHL, and 10 children with UHL participated (8–12 years). They followed audiovisual instructions for placing objects on a mat under three conditions: a single talker providing instructions via a video monitor, four possible talkers alternately providing instructions on separate monitors in front of the listener, and the same four talkers providing both target and nontarget information. Multi-talker background noise was presented at a 5 dB signal-to-noise ratio during testing. An eye tracker monitored looking behavior while children performed the experimental task. Results: Behavioral task performance was higher for children with NH than for either group of children with hearing loss. There were no differences in performance between children with UHL and children with MBHL. Eye-tracker analysis revealed that children with NH looked more at the screens overall than did children with MBHL or UHL, though individual differences were greater in the groups with hearing loss. Listeners in all groups spent a small proportion of time looking at relevant screens as talkers spoke. Although looking was distributed across all screens, there was a bias toward the right side of the display. There was no relationship between overall looking behavior and performance on the task. Conclusions: The present study examined the processing of audiovisual speech in the context of a naturalistic task. Results demonstrated that children distributed their looking to a variety of sources during the task, but that children with NH were more likely to look at screens than were those with MBHL/UHL. However, all groups looked at the relevant talkers as they were speaking only a small proportion of the time. Despite variability in looking behavior, listeners were able to follow the audiovisual instructions and children with NH demonstrated better performance than children with MBHL/UHL. These results suggest that performance on some challenging multi-talker audiovisual tasks is not dependent on visual fixation to relevant talkers for children with NH or with MBHL/UHL. Acknowledgments: The content of this article is the responsibility of the authors, and opinions of the authors do not necessarily represent the views of the National Institutes of Health. The authors thank Bob McMurray, Lori Leibold, and Ryan McCreery for discussions regarding this article and Christine Hammans for proofreading assistance. Supplemental digital content is available for this article. Direct URL citations appear in the printed text and are provided in the HTML and text of this article on the journal’s Web site (www.ear-hearing.com). This work was supported by NIH grants R03 DC009675, R03 DC009884, T32 DC000013, P20 GM109023, and P30 DC004662. The first author (D.L.) is a member of the Phonak Pediatric Advisory Board, but that membership has no conflicts with the content of this article. Portions of the findings have been presented at professional meetings. Address for correspondence: Dawna E. Lewis, Boys Town National Research Hospital, 555 N. 30th Street, Omaha, NE 68131. E-mail: dawna.lewis@boystown.org. Received March 16, 2016; accepted October 25, 2017. Copyright © 2017 Wolters Kluwer Health, Inc. All rights reserved.

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The Summating Potential Is a Reliable Marker of Electrode Position in Electrocochleography: Cochlear Implant as a Theragnostic Probe

Objective: For the increasing number of cochlear implantations in subjects with residual hearing, hearing preservation, and thus the prevention of implantation trauma, is crucial. A method for monitoring the intracochlear position of a cochlear implant (CI) and early indication of imminent cochlear trauma would help to assist the surgeon to achieve this goal. The aim of this study was to evaluate the reliability of the different electric components recorded by an intracochlear electrocochleography (ECochG) as markers for the cochleotopic position of a CI. The measurements were made directly from the CI, combining intrasurgical diagnostics with the therapeutical use of the CI, thus, turning the CI into a “theragnostic probe.” Design: Intracochlear ECochGs were measured in 10 Dunkin Hartley guinea pigs of either sex, with normal auditory brainstem response thresholds. All subjects were fully implanted (4 to 5 mm) with a custom six contact CI. The ECochG was recorded simultaneously from all six contacts with monopolar configuration (retroauricular reference electrode). The gross ECochG signal was filtered off-line to separate three of its main components: compound action potential, cochlear microphonic, and summating potential (SP). Additionally, five cochleae were harvested and histologically processed to access the spatial position of the CI contacts. Both ECochG data and histological reconstructions of the electrode position were fitted with the Greenwood function to verify the reliability of the deduced cochleotopic position of the CI. Results: SPs could be used as suitable markers for the frequency position of the recording electrode with an accuracy of ±1/4 octave in the functioning cochlea, verified by histology. Cochlear microphonics showed a dependency on electrode position but were less reliable as positional markers. Compound action potentials were not suitable for CI position information but were sensitive to “cochlear health” (e.g., insertion trauma). Conclusions: SPs directly recorded from the contacts of a CI during surgery can be used to access the intracochlear frequency position of the CI. Using SP monitoring, implantation may be stopped before penetrating functioning cochlear regions. If the technique was similarly effective in humans, it could prevent implantation trauma and increase hearing preservation during CI surgery. Diagnostic hardware and software for recording biological signals with a CI without filter limitations might be a valuable add-on to the portfolios of CI manufacturers. ACKNOWLEDGMENTS: The authors thank Dr. W.S. Konerding for comments on an earlier version of the manuscript and help with the statistical analysis. We are also grateful to MED-EL Comp. (Innsbruck, Austria) for providing the custom-build research cochlear implants for this study. This work was supported by the German Research Foundation Deutsche Forschungsgemeinschaft (DFG) Cluster of Excellence Hearing4 All, Hannover, Germany (DFG EXC 1077). The authors have no conflicts of interest to disclose. Address for correspondence: Peter Baumhoff, Department of Experimental Otology, VIANNA, Hannover, Germany. E-mail: baumhoff.peter@mh-hannover.de Received November 29, 2016; accepted October 8, 2017. Copyright © 2017 Wolters Kluwer Health, Inc. All rights reserved.

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Cost-Effective Hearing Conservation: Regulatory and Research Priorities

Hearing conservation programs (HCPs) mandated by the US Occupational Safety and Health Administration (OSHA) cost about $350/worker/year. Are they cost-effective? A cross-sectional model of the US adult population with and without HCPs incorporates (1) the American Medical Association’s method for estimating binaural hearing impairment and whole-person impairment; (2) the model of the International Organization for Standardization (ISO) for estimating both age-related and noise-induced hearing loss; and (3) an acceptable cost of $50,000 per quality-adjusted life year. The ISO model’s outputs were audiometric thresholds for groups of people with different age, sex, and noise exposure history. These thresholds were used to estimate cost per quality-adjusted life year saved for people in HCPs with different noise exposure levels. Model simulations suggest that HCPs may be cost-effective only when time-weighted average (TWA) noise exposures are ≥ 90 dBA. Enforcing existing regulations, requiring engineering noise control at high exposure levels, and using new methods that can document hearing protection device performance could improve cost-effectiveness. If the OSHA action level remains at 85 dBA-TWA, reducing the permissible exposure limit to the same level would simplify management and slightly improve cost-effectiveness. Research should evaluate employer compliance across industries, determine whether workers currently excluded from HCP regulations are at risk of noise-induced hearing loss, and develop cost-effective HCPs for mobile workers in construction, agriculture, and oil and gas drilling and servicing. Research on HCP cost-effectiveness could be extended to incorporate sensitivity analyses of the effects of a wider range of assumptions. ACKNOWLEDGMENTS: The author thanks Elliott Berger (3M Company) and Dennis Driscoll (Associates in Acoustics), who offered comments on a draft manuscript, and Howard Hoffman and Katalin Losonczy (National Institute on Deafness and other Communication Disorders, NIDCD), who provided statistical analyses of recent national health survey data sets collected by the National Center for Health Statistics via co-sponsorship and funding from NIDCD. The author has a private consulting practice (http://ift.tt/2pFajjj) in hearing conservation; no client was aware of this article prior to submission for publication. Address for correspondence: Robert A. Dobie, 214 Morton Street, San Antonio, TX 78209. E-mail: dobie@uthscsa.edu Copyright © 2017 Wolters Kluwer Health, Inc. All rights reserved.

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Verbal Learning and Memory After Cochlear Implantation in Postlingually Deaf Adults: Some New Findings with the CVLT-II

Objectives: Despite the importance of verbal learning and memory in speech and language processing, this domain of cognitive functioning has been virtually ignored in clinical studies of hearing loss and cochlear implants in both adults and children. In this article, we report the results of two studies that used a newly developed visually based version of the California Verbal Learning Test–Second Edition (CVLT-II), a well-known normed neuropsychological measure of verbal learning and memory. Design: The first study established the validity and feasibility of a computer-controlled visual version of the CVLT-II, which eliminates the effects of audibility of spoken stimuli, in groups of young normal-hearing and older normal-hearing (ONH) adults. A second study was then carried out using the visual CVLT-II format with a group of older postlingually deaf experienced cochlear implant (ECI) users (N = 25) and a group of ONH controls (N = 25) who were matched to ECI users for age, socioeconomic status, and nonverbal IQ. In addition to the visual CVLT-II, subjects provided data on demographics, hearing history, nonverbal IQ, reading fluency, vocabulary, and short-term memory span for visually presented digits. ECI participants were also tested for speech recognition in quiet. Results: The ECI and ONH groups did not differ on most measures of verbal learning and memory obtained with the visual CVLT-II, but deficits were identified in ECI participants that were related to recency recall, the buildup of proactive interference, and retrieval-induced forgetting. Within the ECI group, nonverbal fluid IQ, reading fluency, and resistance to the buildup of proactive interference from the CVLT-II consistently predicted better speech recognition outcomes. Conclusions: Results from this study suggest that several underlying foundational neurocognitive abilities are related to core speech perception outcomes after implantation in older adults. Implications of these findings for explaining individual differences and variability and predicting speech recognition outcomes after implantation are discussed. ACKNOWLEDGMENTS: Data collection and analysis were supported by the National Institutes of Health, National Institute on Deafness and Other Communication Disorders (NIDCD) Career Development Award 5K23DC015539-02, and the American Otological Society Clinician-Scientist Award to A.C.M. This research was also supported by NIDCD research grant DC-000111 and the IU Grant Linking University-wide Expertise award to David Pisoni and NIDCD research grant DC-015257 to David Pisoni and William Kronenberger. ResearchMatch, used to recruit some NH participants, is supported by National Center for Advancing Translational Sciences Grant UL1TR001070. The authors acknowledge Susan Nittrouer for providing some testing materials used in this study, Beth Miles-Markley for her administrative support, and Leticia DeLeon, Gabrielle Grose, and Eleanor Gulick for their assistance in data scoring for this study. We also thank Terren Green for her help in preparing the final manuscript for publication and overseeing the research project. All authors contributed to the completion of this research project. A.C.M., D.B.P., W.G.H., L.R.H., and M.S.H. conceptualized and designed the original study and research protocol. A.C.M. and D.B.P. supervised the data collection at both sites. D.B.P., A.C.M., and W.G.K. drafted, edited, and revised the final manuscript. D.B.P., A.C.M., W.G.K., K.D.M., and H.X. carried out the statistical analyses. D.B.P., A.C.M., L.R.H., and K.D.M. prepared the graphics and figures. All authors of this article were involved in discussing the major results of the study and commented on the final manuscript in various stages of preparation. The authors declare no conflicts of interest. Address for correspondence: David B. Pisoni, Speech Research Laboratory, Department of Psychological and Brain Sciences, Indiana University, 1101 East 10th Street, Bloomington, IN 47405, USA. E-mail: Pisoni@indiana.edu Received June 23, 2017; accepted October 5, 2017. Copyright © 2017 Wolters Kluwer Health, Inc. All rights reserved.

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