Article 3
Article 3
Article 3
A R T I C L E I N F O A B S T R A C T
Keywords: Objectives: Age of cochlear implantation (CI) is an important predictor of language development in those with
Delayed cochlear implantation congenital sensorineural hearing loss. Despite universal newborn hearing screening initiatives and the known
Early implantation benefits of early CI, a subset of congenitally deaf children continue to be evaluated for cochlear implants later in
Universal newborn hearing screening
childhood. This study aims to identify the barriers to early cochlear implantation in these children.
Barriers to care
Barriers to cochlear implantation
Methods: A retrospective review was conducted for all pediatric cochlear implants aged 3 years or older per
formed at a single academic institution between 2013 and 2017. Children implanted before the age three, those
with a prior unilateral cochlear implant, and those with progressive or sudden hearing loss were excluded.
Variables included newborn hearing screen results, age at hearing loss diagnosis, time of initiation and docu
mented benefit of hearing aids, age of implantation, pre/post-implantation evaluation scores, and reason for
delayed referral for cochlear implantation.
Results: Thirty-one patients were identified meeting these inclusion criteria. Twenty-one children were subject to
UNBS in the U.S. Fourteen of those children failed their newborn hearing screening. Average age at implantation
was 6.2 years. Four reasons were identified for increased age at cochlear implantation. Two categories represent
delays related to (1) Amplification continually prescribed even though the range of hearing loss and speech
development assessment suggests CI may have been more appropriate well before referral (N = 13) (2) Patients
were not subject to newborn hearing screening and/or timely diagnosis of their hearing loss (N = 8). In other
cases, patients were appropriately fit with hearing aids until evidence that they derived limited benefit and then
referred for CI (N = 8). Lastly, in a few cases, records were indeterminate with regards to the timing and
appropriate diagnosis of their hearing loss (N = 2).
Conclusion: Understanding the reasons for delayed cochlear implantation in congenitally deaf children might
allow the development of targeted interventions to improve outcomes. Specifically, those children who were not
referred before age 3 despite use of amplification with limited benefit offer one potential target population for
earlier CI.
* Corresponding author. Division of Otology, Neurotology and Skull Base Surgery Department of Otolaryngology – Head and Neck Surgery, New York University
Grossman School of Medicine, 530 First Avenue, Suite 7Q, New York City, NY, 10016, USA.
E-mail address: [email protected] (D.R. Friedmann).
https://doi.org/10.1016/j.ijporl.2022.111086
Received 29 September 2021; Received in revised form 1 February 2022; Accepted 15 February 2022
Available online 22 February 2022
0165-5876/© 2022 Elsevier B.V. All rights reserved.
S.A. Gordon et al. International Journal of Pediatric Otorhinolaryngology 155 (2022) 111086
2. Methods
3. Results
3.1. Delayed referral (GROUP 2)
Thirty-one children implanted after the age of 3 met our inclusion
criteria and were included for analysis (see Fig. 1). The individual pa Based on individual chart abstraction and discussion with the
tient demographics and outcomes are described in Table 1. Fourteen of treating audiologists, the children with delayed CI referral despite
the twenty-one children subject to UNBS reportedly failed their newborn limited demonstrable benefit from amplification was inferred to occur
hearing screening. Overall, 6 children were found to have cochlear related to three underlying factors—1) those related to family dynamics;
malformations—3 with Mondini malformation and an enlarged vestib 2) structural/socioeconomic; and 3) inadequate guidance from prior
ular aqueduct and 3 with cochlear nerve dysplasia. Nine children providers.
received simultaneous bilateral implantation and another five under
went bilateral implantation in a sequential manner. For those obtaining 3.2. Patient/family factors
a single implant, there were 9 children with right-sided CI and 8
receiving a left. Twenty-two of the thirty-one implants were Cochlear In this cohort, the majority (7/13) of delays in cochlear implantation
brand, 8 Advanced Bionic, and 1 Med-El. Mean age at first hearing aid were a result of family hesitancy. For example, Patients 3 and 17 grew
was 2.8 years old (range 4 months–6 years). Overall, the mean age at up in conservative Middle Eastern and East Asian families. Parents
cochlear implantation in this cohort was 6.2 years old (range 3.1–13.5 initially denied their child’s diagnosis, contributing to the delayed im
years). plantation. Specifically, Patient 3 failed her newborn hearing screening
Individual chart abstraction was performed to determine what fac and the family waited nearly two years before following up. She has
tors impacted on the age at implantation. In doing so, 4 themes were three older siblings with hearing loss that were similarly delayed in
identified: treatment. The family reported they were worried about the cultural
stigma associated with a diagnosis of hearing loss and ultimately
required multiple referrals for cochlear implantation.
2
Table 1
1 4.5 5.0 BIL none 3 Severe to Profound Loss BIL mild HL, moved out of
state
2 4.5 6.2 BIL none 3 ESPT unable to complete past IT-MAIS 25/40
training
3 2 13.5 L then R fail 2 PBK words: CNC words:
R HA- 24%, L HA- 8%, B/L- 20% R CI-66%, L CI-60%, B/l-80%
HINT C SENT (noise) AZ BIO (quiet)
R HA- 36%, L HA- 0%, B/L- 34% R CI-94%, L CI-86%, B/l-98%
AZ BIO(noise)
R CI-81%, L CI-68%, B/l-79%
4 2.5 3.7 R none 3 GASP words: PBK-1/2 list:
R HA- 0%, L HA- 83% R CI - 48%, L HA- 56%,
MLNT words: B/l-74%
R HA: DNT, L HA- 76%, B/l- 87%
5 0.5 4.9 R fail 2 LNT words: LNT words: HINT C Sentences (quiet):
R HA-0% Bilateral- 84% R CI-80% Bilateral- R CI-94% Bilateral- 100%
84% HINT C Sentences (noise):
R CI-93% Bilateral- 94%
6 5.5 6.1 BIL EVA, Cochlear N unknown 3 could not perform the training ESPT: Cat 3 8/17
Deficiency subtest correct
7 6 6.1 R then L pass 4 LNT words: LNT words: CNC words:
(2017) Bilateral: 56% CI-64% Bilateral: 84% R CI-96%, L CI-88%, B/l-96%
HINT sent (quiet)
R CI-90%, L CI-96%, B/l-100%
HINT sent (noise)
3
A similar reluctance was seen from the family of Patient 17, who had
Legend: BIL= Bilateral; EVA = Enlarged Vestibular Aqueduct; CSF= Cerebrospinal Fluid; R = Right; L = Left; HA= Hearing Aid; CI= Cochlear Implant; CAT= Category; IT-MAIS = The Infant-Toddler Meaningful Auditory
Integration Scale; GASP = Glendonald Auditory Screening Procedure; PBK= Phonetically Balanced Kindergarten Test; ESPT = The Early Speech Perception Test; LNT = Lexical Neighborhood Test; MLNT = Multisyllabic
a failed newborn hearing screening and then a subsequent ABR at 4
months indicating a profound loss in the left ear and a moderate loss in
AZ-Bio (noise)
AZ-Bio (quiet)
AZ-Bio (quiet)
Denial of a child’s diagnosis represents another explanatory theme
seen with the families of Patients 20, 21, and 29. Patient 20 failed his
2 years
Age appropriate speech testing determined by audiologist at time of preoperative and post-operative assessment. If blank, testing was not collected/performed or lost to follow up.
educational support services including speech and language therapy,
and assistive listening technology. Despite numerous early CI referrals,
he was not implanted until 8 years of age, because of the family’s belief
Post Operative Speech Testingc
noticed they were not developing speech and language. Both patients
were immediately fit with bilateral amplification and referred for CI.
Post Operative Speech
B/L- 79%
B/L- 98%
B/L- 92%
for hearing loss, they were unable to process the reality of the diagnosis
Cata Preoperative Speech Testingc
HINT C (noise)
HINT C (quiet)
HINT C (quiet)
cities with heavy involvement and support by social services. His follow
up is limited because he left the New York region immediately after
Newborn
implantation.
Screen
pass
A similar dynamic was seen with Patient 23 who failed her newborn
fail
hearing screening in the left ear, but passed in the right. She was born
and had all of her care at the public city hospital affiliated with our
institution. Her parents reported they had no concerns about her hearing
and didn’t follow up with ENT or audiology after her newborn screening
Anatomyb
Abnormal
until she started Pre-K at age 4. At this point, she was formally diagnosed
with profound hearing loss in the left ear and moderate hearing loss in
the right ear and fit with a hearing aid only in the right ear. In this re
view, she was categorized as delayed management because of the sig
Laterality
R, then L
treating providers delayed referral of the child for CI long after they met
11.1
12.0
Table 1 (continued )
criteria. For instance, Child 9 was diagnosed with profound loss on the
right and moderate hearing loss on the left from an ABR at age 3 and on
Age Aided
ear is doing well even with a “threatened contralateral ear.” Per chart
#
5
S.A. Gordon et al. International Journal of Pediatric Otorhinolaryngology 155 (2022) 111086
change or identifiable event that resulted in the CI referral. Similar investigation into barriers to access and utilization to pedi
A similar delay was seen for child 15. He was born full-term and atric hearing healthcare have been designed to examine the impact of
passed a newborn hearing screening. Hearing loss was ultimately diag broader social disparities on the care of children with hearing loss
nosed between the ages of 2–3 years after two surgeries for myr [38–47]. For instance, Bush et al. have identified rural zip code and
ingotomy and tympanostomy tubes. He attained expressive language at distance from cochlear implant centers as significant barriers to hearing
age 2.5 years but had continued difficulty with speech, resulting in a healthcare [38–41]. There has been conflicting data regarding insurance
formal diagnosis of hearing loss at age 3 when he was fit with amplifi status and barriers to hearing rehabilitation with a group from Case
cation bilaterally. His provider did not refer him for over 1.5 years for CI Western showing that socioeconomic status and Medicaid insurance did
despite meeting criteria. not impact age of cochlear implantation, however, they did note that
Child 13 and 25 had a provider/system failure that contributed to children with Medicaid insurance missed significantly more
their delayed implantation. Both children failed their newborn hearing post-operative follow up visits [42]. In contrast, a recent study analyzing
screening, had subsequent ABR testing suggestive of bilateral profound delayed cochlear implantation in the state of Florida found children with
hearing loss, and were found to have congenital inner ear anomalies on Medicaid were significantly more likely to be implanted after the age of
follow up CT scan. Patient 13 had marked bilateral cochlea dysplasia, 2 compared to children with private insurance [44]. Moreover, this
narrowed internal auditory canals, and absent semi-circular canals. This group determined that children who identified as either Black or His
child was not implanted until nearly four years old because she was panic were significantly more likely to have delayed implantation
initially denied coverage for implantation by her insurance carrier. compared to their White counterparts [44].
Child 25’s CT scan demonstrating bilateral hypoplastic cochlear and the The current study takes a more in-depth examination into delayed
family was incorrectly told by the provider that the child was not a cochlear implantation and identified four main categories of patients
candidate for CI because of his anatomy. who were implanted after the age of three years, excluding those with
progressive or sudden hearing loss as mentioned above. The children
3.3.1. Newborn hearing screening not performed (GROUP 3) who had amplification continually prescribed even though loss range
An overview of those patients not subject to infant hearing screening was on the range for CI; and those patients who arrived in the US later in
because they were born outside the U.S. is described in Table 2. Five out life seeking medical care—not subject to infant screening and timely
of eight of these patients are children that had the potential to be helped diagnosis, offer opportunities for intervention. In this cohort, the ma
sooner after moving to the US and implanted earlier, had they been jority (7/13) children had delays in care as a result of family/internal
properly referred. The remaining three patients were appropriately factors. Cultural norms, family stigma, and parental acceptance of
diagnosed and referred for evaluation of implant candidacy in a timely diagnosis had major impacts in how these children and families inter
manner and were implanted within one year of arrival to the U.S. acted with the health care system. Providers must incorporate this
knowledge and develop the necessary cultural competency to under
4. Discussion stand the rationale for family resistance to implantation or denial of
diagnosis in order to help properly navigate these at-risk children
In order to develop proper language skills, children require access to through the medical system.
acoustic-phonetic cues [32–36]. However, children born with severe to Similarly, for those not subject to infant screening because they were
profound hearing loss lack access to this information, which can limit born outside the U.S., we lack a safety net to ensure these children are
their communication skills and hinder their development of spoken accurately diagnosed and appropriately managed in a timely fashion. In
language [37]. Cochlear implants, when performed within the critical or many cases, such children remain in the United States, and in addition to
“sensitive period” for neuroplasticity—typically before age 3—can the personal consequences on their development, there are important
potentially mitigate many of these adverse consequences. While the downstream consequences on their educational and vocational oppor
need for early cochlear implantation in children with prelingual hearing tunities and potential economic burden on society [48]. While the
loss has been well established, many children are continually implanted introduction of newborn hearing screening programs has helped to
after this “sensitive period.” This study attempts to understand and accelerate detection of children with profound bilateral SNHL, as this
categorize barriers to early cochlear implantation in order to identify review demonstrates, early treatment is hampered by the complex de
targeted solutions to mitigate delayed implantation in the future. livery of hearing health care.
Table 2
Timeline of hearing healthcare interaction among children born outside the US and not subject to universal newborn screening (category 3): From birth to cochlear
implantation.
Patient # Native County Hearing Loss Newborn screen Age at Age at Diagnosis Age at Hearing Aid Age at
Arrival to US Implantation
6
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