Comparison of the Motor Performance and Vestibular Function in Infants with a Congenital Cytomegalovirus Infection or a Connexin 26 Mutation: A Preliminary Study.

OBJECTIVES: Hearing-impaired children are at risk for vestibular damage and delayed motor development. Two major causes of congenital hearing loss are cytomegalovirus (CMV) infection and connexin (Cx) 26 mutations. Comparison of the motor performance and vestibular function between these specific groups is still underexplored. The objective of this study was to investigate the impact of congenital (c)CMV and Cx26 on the motor performance and vestibular function in 6 months old infants. DESIGN: Forty children (mean age 6.7 months; range 4.8 to 8.9 months) participated in this cross-sectional design and were recruited from the Flemish CMV registry. They were divided into five age-matched groups: normal-hearing control, asymptomatic cCMV, normal-hearing symptomatic cCMV, hearing-impaired symptomatic cCMV, and hearing-impaired Cx26. Children were examined with the Peabody Developmental Motor Scales-2 and cervical vestibular-evoked myogenic potential (cVEMP) test. RESULTS: Symptomatic hearing-impaired cCMV children demonstrated a significantly lower gross motor performance compared with the control group (p = 0.005), the asymptomatic cCMV group (p = 0.034), and the Cx26 group (0.016). In this symptomatic hearing-impaired cCMV group, 4 out of 8 children had absent cVEMP responses that were related to the weakest gross motor performance. The Cx26 children showed no significant delay in motor development compared with the control children and none of these children had absent cVEMP responses. CONCLUSIONS: The weakest gross motor performance was found in symptomatic hearing-impaired cCMV-infected children with absent cVEMP responses. These results suggest that abnormal saccular responses are a major factor for this delayed motor development, although more work is needed including comprehensive vestibular function testing to verify this.

Early motor development of children with a congenital cytomegalovirus infection.

BACKGROUND: Congenital cytomegalovirus (cCMV) infection is the most important etiology of non-hereditary childhood hearing loss and an important cause of neurodevelopmental delay. The current study aimed to investigate the early motor development of symptomatic and asymptomatic cCMV infected children with and without sensorineural hearing loss (SNHL). METHODS: Sixty-four children with a cCMV infection, without cerebral palsy, were compared to a control group of 107 normal hearing children. They were assessed around the ages of 6, 12, and 24 months with the Peabody Developmental Motor Scales-2 (PDMS-2), Alberta Infant Motor Scales (AIMS), and Ghent Developmental Balance Test (GDBT). The cCMV infected children were subdivided into a symptomatic (n=26) and asymptomatic cCMV group (n=38) but also into a cCMV group with SNHL (n=19) and without SNHL (n=45). RESULTS: Symptomatic cCMV infected children and cCMV infected children with SNHL performed significantly weaker for all gross motor outcome measures. CONCLUSION: A congenital CMV infection is a risk factor for a delay in the early motor development. Follow-up will be necessary to gain insight into the exact cause of this motor delay and to define the predictive value of early motor assessment of cCMV infected children.

Examining the impact of cochlear implantation on the early gross motor development of children with a hearing loss.

OBJECTIVE: As deaf children are now implanted at a very early age, the influence of a cochlear implant (CI) on the early motor development of children with a hearing loss becomes relevant. DESIGN: Forty-eight children with a hearing loss were included in this controlled prospective follow-up study and were subdivided into a CI group (n = 23) receiving a CI during the follow-up period and a control group (n = 25) receiving no CI during the follow-up period. All children were assessed around the ages of 6 (T1), 12 (T2), 18 (T3), and 24 (T4) months with a motor test battery consisting of the Peabody Developmental Motor Scales-2 (PDMS-2), Alberta Infant Motor Scales (AIMS) (only at T1 and T2), and Ghent Developmental Balance Test (GDBT) (only at T3 and T4). In addition, collic vestibular-evoked myogenic potential testing was performed in all children. Group differences in PDMS-2 Gross Motor Quotient (GMQ), Fine Motor Quotient, AIMS z score, and GDBT z score were analyzed using Linear Mixed Model (LMM) analysis for repeated measures. RESULTS: For PDMS-2 GMQ, the LMM revealed significant effects for group (p = 0.04), test moment (p < 0.001), and for the interaction between these two factors (p = 0.035). Contrasts indicated that the CI group showed a greater deterioration in PDMS-2 GMQ between T2 and T3 compared with that showed by the control group (p = 0.002). The LMM for PDMS-2 Fine Motor Quotient and AIMS z score showed no significant effects. For GDBT z score, the LMM pointed out significant effects for group (p = 0.013) and test moment (p < 0.001), but no significant interaction between these two factors. Contrasts indicated that the CI group performed significantly weaker than the control group at both test moments (T3 and T4; all p < 0.012) and that both groups showed a significant recovery in GDBTz scores between T3 and T4 (all p < 0.012). CONCLUSIONS: This study shows that the trajectory of gross motor development can be changed in children with a hearing loss after a cochlear implantation. Implanted children show a drop in their gross motor performance within the age range of 6 to 18 months, at which period the majority of the implantations took place, with a tendency of recovery toward the age of 2 years. However, longer follow-up will be necessary to trace whether the implanted children catch up their motor delay in comparison with nonimplanted children with a hearing loss at later age.

Association between vestibular function and motor performance in hearing-impaired children.

OBJECTIVE: The clinical balance performance of normal-hearing (NH) children was compared with the balance performance of hearing-impaired (HI) children with and without vestibular dysfunction to identify an association between vestibular function and motor performance. STUDY DESIGN: Prospective study. SETTING: Tertiary referral center. PATIENTS: Thirty-six children (mean age, 7 yr 5 mo; range, 3 yr 8 mo-12 yr 11 mo) divided into three groups: NH children with normal vestibular responses, HI children with normal vestibular responses, and HI children with abnormal vestibular function. INTERVENTIONS: A vestibular test protocol (rotatory and collic vestibular evoked myogenic potential testing) in combination with three clinical balance tests (balance beam walking, one-leg hopping, one-leg stance). MAIN OUTCOME MEASURES: Clinical balance performance. RESULTS: HI children with abnormal vestibular test results obtained the lowest quotients of motor performance, which were significantly lower compared with the NH group (p < 0.001 for balance beam walking and one-leg stance; p = 0.003 for one-leg hopping). The balance performance of the HI group with normal vestibular responses was better in comparison with the vestibular impaired group but still significantly lower compared with the NH group (p = 0.020 for balance beam walking; p = 0.001 for one-leg stance; not significant for one-leg hopping). CONCLUSION: These results indicate an association between vestibular function and motor performance in HI children, with a more distinct motor deterioration if a vestibular impairment is superimposed to the auditory dysfunction.

Middle and inner ear malformations in two siblings exposed to valproic acid during pregnancy: a case report.

Valproic acid (VPA) is a known teratogenic drug. Exposure to VPA during the pregnancy can lead to a distinct facial appearance, a cluster of major and minor anomalies and developmental delay. In this case report, two siblings with fetal valproate syndrome and a mild conductive hearing loss were investigated. Radiologic evaluation showed middle and inner ear malformations in both children. Audiologic, vestibular and motor examination was performed. This is the first case report to describe middle and inner ear malformations in children exposed to VPA.

Rotatory and collic vestibular evoked myogenic potential testing in normal-hearing and hearing-impaired children.

OBJECTIVES: Vertigo and imbalance are often underestimated in the pediatric population, due to limited communication abilities, atypical symptoms, and relatively quick adaptation and compensation in children. Moreover, examination and interpretation of vestibular tests are very challenging, because of difficulties with cooperation and maintenance of alertness, and because of the sometimes nauseatic reactions. Therefore, it is of great importance for each vestibular laboratory to implement a child-friendly test protocol with age-appropriate normative data. Because of the often masked appearance of vestibular problems in young children, the vestibular organ should be routinely examined in high-risk pediatric groups, such as children with a hearing impairment. Purposes of the present study were (1) to determine age-appropriate normative data for two child-friendly vestibular laboratory techniques (rotatory and collic vestibular evoked myogenic potential [cVEMP] test) in a group of children without auditory or vestibular complaints, and (2) to examine vestibular function in a group of children presenting with bilateral hearing impairment. DESIGN: Forty-eight typically developing children (mean age 8 years 0 months; range: 4 years 1 month to 12 years 11 months) without any auditory or vestibular complaints as well as 39 children (mean age 7 years 8 months; range: 3 years 8 months to 12 years 10 months) with a bilateral sensorineural hearing loss were included in this study. All children underwent three sinusoidal rotations (0.01, 0.05, and 0.1 Hz at 50 degrees/s) and bilateral cVEMP testing. RESULTS: No significant age differences were found for the rotatory test, whereas a significant increase of N1 latency and a significant threshold decrease was noticeable for the cVEMP, resulting in age-appropriate normative data. Hearing-impaired children demonstrated significantly lower gain values at the 0.01 Hz rotation and a larger percentage of absent cVEMP responses compared with normal-hearing children. Seventy-four percent of hearing-impaired children showed some type of vestibular abnormality when examined with a combination of rotatory and cVEMP testing, in contrast to an abnormality rate of 60% with cVEMP and a rate of 49% with rotatory testing alone. CONCLUSIONS: The observed pediatric age correlations underscore the necessity of age-appropriate normative data to guarantee accurate interpretation of test results. The high percentages of abnormal vestibular test results in hearing-impaired children emphasize the importance of vestibular assessment in these children because the integrity of the vestibular system is a critical factor for motor and psychological development.

Gait analysis with cognitive-motor dual tasks to distinguish fallers from nonfallers among rehabilitating stroke patients.

OBJECTIVES: To evaluate fall risk in stroke patients based on single- and dual-task gait analyses, and to investigate the difference between 2 cognitive tasks in the dual-task paradigm. DESIGN: Prospective cohort study. SETTING: Rehabilitation hospitals. PARTICIPANTS: Subacute stroke patients (N=32), able to walk without physical/manual help with or without walking aids, while performing a verbal task. INTERVENTIONS: Not applicable. MAIN OUTCOME MEASURES: Functional gait measures were Functional Ambulation Categories (FAC) and use of a walking aid. Gait measures were evaluated by an electronic walkway system under single- and dual-task (DT) conditions. For the single-task, subjects were instructed to walk at their usual speed. One of the DTs was a verbal fluency dual task, whereby subjects had to walk while simultaneously enumerating as many different animals as possible. For the other DT (counting dual task), participants had to walk while performing serial subtractions. After inclusion, participants kept a 6-month falls diary. RESULTS: Eighteen (56.3%) of the 32 included patients fell. Ten (31.3%) were single fallers (SFs), and 8 (25%) were multiple fallers (MFs). Fallers (Fs) more frequently used a walking aid and more frequently needed an observatory person for walking safely (FAC score of 3) than nonfallers (NFs). Two gait decrement parameters in counting dual task could distinguish potential Fs from NFs: decrement in stride length percentage (P=.043) and nonparetic step length percentage (P=.047). Regarding the division in 3 groups (NFs, SFs, and MFs), only MFs had a significantly higher percentage of decrement for paretic step length (P=.023) than SFs. CONCLUSIONS: Examining the decrement of spatial gait characteristics (stride length and paretic and nonparetic step length) during a DT addressing working memory can identify fall-prone subacute stroke patients.

The influence of a vestibular dysfunction on the motor development of hearing-impaired children.

OBJECTIVES/HYPOTHESIS: To identify the predictive ability of vestibular function test results on motor performance among hearing-impaired children. STUDY DESIGN: Cross-sectional study. METHODS: Fifty-one typically developing children and 48 children with a unilateral (n = 9) or bilateral hearing impairment (n = 39) of more than 40 dB HL between 3 and 12 years were tested by the Movement Assessment Battery for Children-Second Edition (M ABC-2), clinical balance tests, posturography, rotatory chair testing, and vestibular evoked myogenic potential (VEMP). From the group of hearing-impaired children, 23 had cochlear implants. RESULTS: Balance performance on M ABC-2, clinical balance tests, as well as the sway velocity assessed by posturography in bipedal stance on a cushion with eyes closed and in unilateral stance differed significantly between both groups. Presence of a VEMP response is an important clinical parameter because comparison of the motor performance among hearing-impaired children between those with present and absent VEMPs showed significant differences in balance performance. The three most important predictor variables on motor performance by bivariate regression analyses are the vestibular-ocular reflex (VOR) gain value of the rotatory chair test at 0.01 and 0.05 Hz frequency, as well as the VEMP asymmetry ratio. Multivariate regression analyses suggest that the VOR asymmetry value of the rotatory chair test at 0.05 Hz and the etiology of the hearing loss seem to have additional predictive value. CONCLUSIONS: Hearing-impaired children are at risk for balance deficits. A combination of rotatory chair testing and VEMP testing can predict the balance performance.

Ghent developmental balance test: a new tool to evaluate balance performance in toddlers and preschool children.

BACKGROUND: Balance is a fundamental component of movement. Early identification of balance problems is important to plan early intervention. The Ghent Developmental Balance Test (GDBT) is a new assessment tool designed to monitor balance from the initiation of independent walking to 5 years of age. OBJECTIVE: The purpose of this study was to establish the psychometric characteristics of the GDBT. METHODS: To evaluate test-retest reliability, 144 children were tested twice on the GDBT by the same examiner, and to evaluate interrater reliability, videotaped GDBT sessions of 22 children were rated by 3 different raters. To evaluate the known-group validity of GDBT scores, z scores on the GDBT were compared between a clinical group (n = 20) and a matched control group (n = 20). Concurrent validity of GDBT scores with the subscale standardized scores of the Movement Assessment Battery for Children-Second Edition (M-ABC-2), the Peabody Developmental Motor Scales-Second Edition (PDMS-2), and the balance subscale of the Bruininks-Oseretsky Test-Second Edition (BOT-2) was evaluated in a combined group of the 20 children from the clinical group and 74 children who were developing typically. RESULTS: Test-retest and interrater reliability were excellent for the GDBT total scores, with intraclass correlation coefficients of .99 and .98, standard error of measurement values of 0.21 and 0.78, and small minimal detectable differences of 0.58 and 2.08, respectively. The GDBT was able to distinguish between the clinical group and the control group (t(38) = 5.456, P<.001). Pearson correlations between the z scores on GDBT and the standardized scores of specific balance subscales of the M-ABC-2, PDMS-2, and BOT-2 were moderate to high, whereas correlations with subscales measuring constructs other than balance were low. CONCLUSIONS: The GDBT is a reliable and valid clinical assessment tool for the evaluation of balance in toddlers and preschool-aged children.

Prediction of falling among stroke patients in rehabilitation.

OBJECTIVE: To identify risk factors and predict falling in stroke patients. To determine the strength of general vs mobility screening for this prediction. DESIGN: Prospective study. SUBJECTS: Patients in the first 6 months after stroke. METHODS: The following assessments were carried out: an interview concerning civil state and fall history, Mini-Mental State Examination, Geriatric Depression Scale, Falls Efficacy Scale (FES), Star Cancellation Task (SCT), Stroop test, Berg Balance Scale, Functional Ambulation Categories (FAC), Motricity Index, grip and quadriceps strength, Modified Ashworth Scale, Katz scale, and a 6-month fall follow-up. RESULTS: Sixty-five patients were included for analysis. Thirty -eight (58.5%) reported falling. Risk factors were: being single (odds ratio (OR) 4.7; 95% confidence interval (95% CI) 1.2-18.3), SCT-time (OR 1.2; 95% CI 1.0-1.3), grip strength on unaffected side (US) (OR 0.1; 95% CI 0.0-0.8), FAC 3 vs FAC 4-5 (OR 8.1; 95% CI 1.5-43.2), and walking aid vs none (OR 5.1; 95% CI 1.4-17.8). These parameters were included in predictive models, which finally implied a general model (I) with inclusion of SCT-time, FAC category and use of walking aid. A mobility model (II) included: FAC category and strength (US). These models showed a sensitivity of 94.1% and 76.3%, respectively. CONCLUSION: Several assessments and both prediction models showed acceptable accuracy in identifying fall-prone patients. A purely physical model can be used; however, looking beyond mobility aspects adds value. Further validation of these results is required.

Construct validity of the assessment of balance in children who are developing typically and in children with hearing impairments.

BACKGROUND: Children with hearing impairments have a higher risk for deficits in balance and gross motor skills compared with children who are developing typically. As balance is a fundamental ability for the motor development of children, a valid and reliable assessment to identify weaknesses in balance is crucial. OBJECTIVE: The purpose of this study was to investigate the construct validity of posturography and clinical balance tests in children with hearing impairments and in children who are developing typically. METHODS: The study involved 53 children with typical development and 23 children with hearing impairments who were between 6 and 12 years of age and without neuromotor or orthopedic disorders. All participants completed 3 posturography tests (modified Clinical Test of Sensory Interaction of Balance [mCTSIB], unilateral stance, and tandem stance) and 4 clinical balance tests (one-leg stance with eyes open and with eyes closed, balance beam walking, and one-leg hopping). RESULTS: Three conditions of the mCTSIB, unilateral stance, and 2 clinical balance tests were able to distinguish significantly between the 2 groups. Children with hearing impairments showed more difficulties in balance tasks compared with children who were developing typically when 1 or 2 types of sensory information were eliminated or disturbed. The study showed only low to moderate correlations among the different methods of evaluating balance. CONCLUSIONS: Clinical balance tests and posturography offer different but complementary information. An assessment protocol for balance consisting of posturography and clinical balance tasks is proposed. Static and dynamic balance abilities could not be differentiated and seem not to be a valid dichotomy.