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. 2019 Dec 17;10:1294. doi: 10.3389/fneur.2019.01294

Table 2.

Studies addressing central neurological conditions (Sensorineural hearing loss and cochlear implant).

References Country Study design Participants Age Etiology and severity Comorbidities Vestibular assessment tools Main results, outcomes and prevalence NCO score
Sokolov et al. (40) Canada Cohort 20 children with unilateral SNHL Mean age: 8.8 y.o. Unilateral SNHL; severe-profound SNHL (PTA = 96 dB), moderate-severe SNHL (PTA = 67 dB); mild-moderate SNHL None specified Caloric test
vHIT
cVEMP
oVEMP
Abnormal vestibular function found in 12/20 (60%) patients.
Abnormal utricle response found in 4/12 (33%) of cases, through oVEMP testing.
Abnormal saccular response absents in 3/18 (17%) of cases, through cVEMP testing.
Overall otolothic dysfunction shown in 4/19 (21%) patients through either cVEMP and/or oVEMP testing.
Abnormal horizontal canal function observed in 7/20 (35%) cases through vHIT testing and observed in 9/19 (48%) cases through calorictesting.
5/9
Raj and Gupta (21) India Cross sectional 50 children with SNHL Mean age: 5.48 y.o.
Age range: 4–9y.o
Congenital profound
and severe SNHL
None reported Warm air caloric test Abnormal vestibular function found in 9/48 (18.75%) cases through caloric testing 7/10
Thierry et al. (24) France Cohort, retrospective 43 children with unilateral CI Mean age: 2.9 y.o.
Age range: 6–15.1y.o.
Etiology of SNHL:
genetic mutations, infections, Waardenburg syndrome, meningitis, Kallman syndrome, or idiopathic
None specified HIT
Bithermal caloric test
VEMP
Decreased ipsilateral vestibular function post-CI observed in 8/43 (18.6%) children.
Abnormal contralateral vestibular function found in 3/43 (7%) cases.
Worsening of vestibular function post-CI experienced by 2/12 (16.7&) patients.
Improvement of vestibular function found in 4/12 (4/12)cases.
4/9
Wolter et al. (43) Canada Cohort, retrospective 187
Group 1: 22 children who experienced CI failure
Group 2: 165 children who did not experience CIfailure
Not specified Etiology of SNHL: Meningitis, cochleovestibular anomaly, Usher syndrome,
Connexin-26 mutation, Cytomegalovirus
None specified Bithermal caloric test
Rotational head impulse test/vHIT
VEMP
Abnormal horizontal canal function found in 18/22 (81.8%) children with CI failure vs. 78/165 (47.3%) children without CI failure through bithermal caloric testing.
Abnormal high frequency horizontal canal function found in 16/22 (72.2%) children with CI failure vs. 57/165 (34.5%) of children without CI failure through vHIT and/or high frequency rotational chair testing.
Abnormal saccular function observed in 18/22 (81.8%) children with CI failure vs. 76/165 (46.1%) children without failure through VEMPtesting.
6/9
Devroede et al. (41) Belgium Cohort, Retrospective 26 children with unilateral CI, before and after Mean age: 6.75 y.o
Age range: 1–13y.o
SNHL as part of a clinical syndrome, genetic mutations, post meningitis, CMV infection, auditory neuropathy spectrum disorder, or idiopathic
± unilateral CI
± bilateral /contralateral CI
None reported Caloric test
VEMP
Pre-contralateral implantation, 2/26 (8%) showed bilateral areflexia, 16/26 (61%) showed hyporeflexia (i.e., 69% presented with hyporeflexia).
Otholitic functioning was abnormal in 5/24 (21%) patients' pre-contralateral implantation, and in 9/24 (37%) post-contralateral implantation, as recorded through VEMP
Horizontal canal function changed in 32% of the patients tested through caloric stimulation
7/9
Cushing et al. (46) Canada Cross sectional Children
Group 1: 119 children with unilateral CIs
Group 2:
34 childrenpre-CI
Mean age: 12.95 y.o
Age range: 3.6–20y.o
Profound SNHL with unilateral CI or before implantation procedure None reported Caloric test
Rotatory chair test
VEMP
VOR
Abnormal horizontal canal function found in: 69/139 (50%) through caloric testing, of which 18/69 (26%) reflect mild to moderate unilateral abnormalities, and 51/139 (37%) severe hypofunction or areflexia.
Abnormal horizontal canal function found in: 64/139 (47%) through rotatory chair testing.
Bilateral reduction in VOR seen in 29% (40/139)
Absent saccular function bilaterally in 32/135 (21%) and unilaterally in 40/135 (30%) through VEMP.
All children with meningitis (n = 11) and 46% with radiologic cochleovestibular anomalies (n = 31) had horizontal canal dysfunction, whereas 45 and 46%, respectively, displayed saccular dysfunction.
10/10
Schwab and Kontorinis (47) Germany Case control Group 1: 40 children with SNHL
Group 2l: 40 normal-hearingchildren
Age range: 4–20 y.o, Deaf of hearing -impaired children admitted for CI exam None specified Caloric test
SOT, MCT,ADT
Abnormal vestibular function found in 16/33 (40%) cases through caloric testing. Hypoexcitabililty of vestibular function found in 27/66 (41%) tested ears, whereas hyperexcitability found in 2/66 (3%) tested ears 4/9
Jafari and Asad Malayeri (48) Iran Case control Group 1: 30 children with SNHL
Group 2: 30 healthychildren
Group 1: Mean age: 6.93 y.o
Age range: 6–9 y.o
Group 2: Mean age: 7.18 y.o
Age range: 6–9y.o
SNHL congenital or early acquired bilateral profound SNHL None specified VEMP, ABR, BOT-2, balance subtest Abnormal vestibular function was found in 28/32 (87.5%) ears tested through VEMP.
Asymmetrical vestibular response found in 4/30 (13.3%) cases through VEMP testing.
No vestibular response found in 12/30 (40%) children through VEMPtesting.
8/9
Licameli et al. (13) USA Cohort Group 1: 42 children with unilateral CI
Group 2: 19 children pre andpost-CI
Group 1: Mean age: 9 y.o
Age range: 5–22 y.o
Group 2: Mean age: 8 y.o
Age range: 2–23y.o
Patients in Group 1 being considered for a second CI on the contralateral side. None reported VOR
Computerized dynamic posturography,VEMP
60% of all patients had abnormal finding(s) in at least one laboratory test.
Abnormal ipsilateral VOR response observed in 22/42 (52%) of Group 1.
Abnormal findings on Computerized dynamic posturography testing found on 15/38 (39%) of Group 1, which indicate peripheral vestibular weakness and/or sensory organization deficit.
Reduced or absent VEMP responses found on 12/15 (80%) of Group 1.
Pre-CI, 2/19 (10%) of group 2 patients did not present any VEMP response.
Post-CI, 16/19 (84%) of group 2 patients indicated disappearances or reduction of VEMP responses (elevation in VEMP thresholds and/or decrease in VEMPamplitudes).
7/9
Zhou et al. (19) USA Cohort, retrospective Group 1: 23 children with bilateral SNHL
Group 2: 12 healthy children
Group 1: Age range: 2–16 y.o.
Group 2: Age range: 4–18y.o
SNHL: Moderate, severe, profound.
SNHL etiology: bialletic GJB2 mutation, congenital cytomegalovirus infection, bacterial meningitis, cogan syndrome
None specified VEMP Abnormal saccular function found in 21/23 (91.3%) through VEMP testing. 3/9
Jacot et al. (25) France Cohort, Prospective & retrospective Children with SNHL, 89 of which participated after CI procedure First examination:
Mean age: 51 mon
Age range: 7 mon−16.5 y.o
Second examination: Mean age of 52.8 mon
Age range: 7 mon−12y.o
SNHL—to be implanted with CI
Unilateral CI
None reported Bi-caloric test
Earth vertical axis rotation
Off vertical axis rotation
VEMP
Abnormal bilateral vestibular function found in 112/224 (50%), 45/224 (20%) showed complete areflexia, 50/224 (22.5%) showed partial asymmetrical hypo-excitability, and 17/224 (7.5%) showed partial symmetrical hypo-excitability.
Changes of vestibular function post-CI found in 51/71 (71%), from which 7/70 (10%) acquired ipsilateral areflexia
Long-term follow up reports partial recovery of vestibular responses is observed in 18.5% of the cases,post-CI
7/9
Cushing et al. (22) Canada Cross sectional Children with unilateral CIs Mean age: 3–19.3 y.o Severe to profound SNHL with unilateral cochlear implants None reported Caloric test
Rotatory chair test
VEMP
BOT-2
Abnormal horizontal canal function found in: 16/32 (50%) through caloric testing, and 14/37 (38%) through rotatory chair testing.
Absent saccular function bilaterally in 5/26 (19%) and unilaterally in 5/26 (19%) through VEMP.
Mean BOT-2 scores for children with SNHL and CI were significantly poorer than the norm.
10/10
Shinjo et al. (49) Japan Cross sectional Children with SNHL Mean age: 54.2 mon
Age range: 31–97 mon (2.5–8y.o)
Conditions included: Severe SNHL, fitted with hearing aids, congenital profound SNHL, progressive hearing impairment, LVA
Etiology: infection, meningitis, congenital auditory nerve disease, common cavity malformation in the inner ear, cochlear nervemalformation
None specified Ice water caloric test
Damped-rotational chair test
VEMP
Abnormal responses in at least 1 test found in 85% of children
Asymmetrical canal responses found in 7/20 (35%) cases, hypo-reactions found in 2/20 (10%) cases, and absence of response observed in 8/20 (40%) cases, all through caloric testing.
Decreased uni-directional canal response observed in 1/20 (5%) cases, decreased bidirectional response observed in 2/20 (10%) cases, and absence of response observed in 3/20 (15%) cases, all through rotational chair testing.
Asymmetrical saccular response found in 6/20 (30%) cases, and absence of bilateral response found in 4/20 (20%) cases, all through VEMP testing.
6/9
Jin et al. (50) Japan Cross sectional Group 1: 12 children who underwent CI surgery
Group 2: 9 healthychildren
Group 1: Mean age: 3.8y.o Age range: 2–7 y.o
Group 2: Age range: 8 mon−10y.o
Cochlear implantation (CI) None VEMP
Calorictest
Semicircular canal hypofunction found through ice water caloric testing in 6/10 (60%) of cases, and areflexia on 4/10 (40%), post implantation.Saccular function reduction observed in 7/12 (58.3%) of patients, through VEMP testing, post-implantation. 9/10
Bouccara et al. (51) France Cohort, Prospective Children Group 1: 240 childrenpost-CI Group 2: 28 children assessedpre-CI Mean age: 7.5 y.o
Age range: 2–15y.o
Idiopathic, genetic, or drug-related hearing loss None reported VNG 9/268 children (3%) present with abnormalities as per the VNG assessment at some point after the implantation 5/9
Lisboa et al. (15) Brazil Cohort Children with SNHL Age range: 10–14 y.o Severity of disease ranged from profound /severe bilateral to unilateral hearing loss None reported Ocular and labyrinthic tests
Caloric Test
RotatoryTest
Alterations on caloric testing found in 25/26 (96.1%) patients, from which:
Unilateral hyporreflexia was found in 4/26 (15.3%) patients
Bilateral hyporreflexia was found in 20/26 (76.9%) patients
Directional preference of asymmetrical nystagmus was found in 1/26 (3.8%)patients
7/9
Rine et al. (20) USA Cross sectional Children with SNHL Age range: 26–83 mon (2–6.9 y.o) Profound bilateral hearing loss Developmental delay PDMS, SCPNT Hypoactive vestibular function found in 20/39 (51.3%) cases through SCPNT.
Hyperactive vestibular function found in 18/39 (46.2%) cases through SCPNT.
Children with moderate to profound sensorineural hearing loss have a delay in gross motor development which is progressive and related to vestibular hypofunction.
7/10
Horak et al. (52) USA Case-control Group 1: 54 normal developing children Group 2: 30 children with bilateral hearing impairment Group 3: 15 children with learning disabilities Age range: 7–12 y.o.
Mean age: 9.2y.o
Bilateral hearing loss acquired within the first two years of life, congenital, post-meningitis, unknown etiology None reported Horizontal VOR
Sensory organization for postural orientation test
BOT for MotorProficiency
Abnormal VOR observed in 20/30 (67%) patients.
Hearing-impaired children with vestibular loss scored at the 29th percentile in motor proficiency because of a mean balance score only half thenormal.
7/9
Potter and Silverman (53) USA Case Control Children with SNHL Mean age: 6.1 y.o
Age range: 5–8.11y.o.
Hearing loss in the better ear ranged from 55 to 120 dB.
Average hearing loss: 100.5dB.
None reported SCPNT
Standing Balance subtests (eyes open and closed) of the Southern California Sensory Integrationtests
Abnormal (hypoactive) vestibular response found in 20/34 (58.8%) of cases through rotatory test (scores compared to norms). 15/34 (44.1%) showed no response to vestibular stimulation.
With eyes open, 44.1% of the deaf children had abnormal standing balance. With eyes closed, 35.3% had abnormalbalance.
4/9
Rosenblut et al. (54) USA Case Control
Group 1: 107 children with SNHL
Group 2: 57 aphasic children (not relevant)
Group 3: 16 healthy children
Age range: 3–13 y.o SNHL resulting from: Meningitis family history, maternal rubella, complications during pregnancy, or congenital brain abnormality Possible congenital brain abnormality Nystagmus assessment through modification of the test originated by Fitzgerald and Hallpike Depressed vestibular function found in 25/107 (23.4%) cases, and absent response reported in 27/107 (25.2%) according to nystagmus assessment. 5/9

vHIT, video head impulse test; cVEMP, cervical vestibular evoked myogenic potential; oVEMP, ocular vestibular evoked myogenic potential; SOT, sensory organization test; MCT, motor control test; ADT, adaptation test; ABR, auditory brainstem response; BOT-2, Bruininks-oseretsky test (second edition); LVA, large vestibular aqueduct; VNG, Videonystagmography; PDMS, peabody developmental motor scale; SCPNT, Southern California post-rotatory nystagmus test; VOR, vestibulo-ocular reflex; y.o, years old; NOS score, Newcastle-OttawaScale.