Article

Voice Abnormalities at School Age in Children Born Extremely Preterm

MBChB, Neonatal Clinical Care Unit, King Edward Hospital, 374 Bagot Rd, Subiaco, Perth, Western Australia, 6008, Australia. .
PEDIATRICS (Impact Factor: 5.47). 03/2013; 131(3):e733-9. DOI: 10.1542/peds.2012-0817
Source: PubMed

ABSTRACT

Voice abnormality is a frequent finding in school age children born at <25 weeks' gestation in Western Australia. The objective of this study was to determine the frequency of voice abnormality, voice-related quality of life, and demographic and intubation factors in this population.
Survivors <25 weeks' gestational age in Western Australia born from 1996 to 2004 were included. Voice assessments (auditory perceptual assessment scale and Pediatric Voice Handicap Index) were carried out by speech pathologists. Intubation history was obtained by retrospective chart review.
Of 251 NICU admissions, 154 (61%) survived. Exclusions were based on severe disability (11) or distant residence (13). Of 70 assessed, 67 completed assessments, 4 (6%) were in the normal range and 39 (58%) showed moderate-severe hoarseness. Simultaneous modeling of demographic and intubation characteristics showed an increased odds of moderate-severe voice disorder for children who had more than 5 intubations (odds ratio 6.96, 95% confidence interval 2.07-23.40, P = .002) and for girls relative to boys (odds ratio 3.46, 95% confidence interval 1.12-10.62, P = .030). Tube size and duration of intubation were not significant in the multivariable model. Median scores of parent-reported voice quality of life on the Pediatric Voice Handicap Index were markedly different for preterm (22) and term (3) groups, P < .001.
Voice disorders in this population were much more frequent than expected. Further studies are required to assess voice across a broader range of gestational ages, and to investigate voice-protective strategies in infants requiring multiple episodes of intubation.

Full-text

Available from: Victoria Reynolds, Dec 22, 2015
Voice Abnormalities at School Age in Children Born
Extremely Preterm
WHATS KNOWN ON THIS SUBJECT: Isolated case reports of
abnormal voice after extremely preterm birth are well described;
however, there are no systematic studies of long-term voice
outcomes in children born preterm.
WHAT THIS STUDY ADDS: Signicant voice abnormalities were
found in more than half of tested chil dren born before 25 weeks
gestation. Multivariable analyses showed that the number of
intubations, not the duration of intubation, and female gender
were strongly associated with this adverse outcome.
abstract
BACKGROUND AND OBJECTIVES: Voice abnormality is a frequent nd-
ing in school age children born at ,25 weeks gestation in Western
Australia. The objective of this study was to determine the frequency
of voice abnormality, voice-related quality of life, and demographic
and intubation factors in this population.
METHODS: Survivors ,25 weeks gestational age in Western Australia
born from 1996 to 2004 were included. Voice assessments (auditory
perceptual assessment scale and Pediatric Voice Handicap Index)
were carried out by speech pathologists. Intubation history was
obtained by retrospective chart review.
RESULTS: Of 251 NICU admissions, 154 (61%) survived. Exclusions were
based on severe disability (11) or distant residence (13). Of 70 assessed,
67 completed assessments, 4 (6%) were in the normal range and 39
(58%) showed moderate-severe hoarseness. Simultaneous modeling
of demographic and intubation characteristics showed an increased
odds of moderate-severe voice disorder for children who had more
than 5 intubations (odds ratio 6.96, 95% condence interval 2.07
23.40, P = .002) and for girls relative to boys (odds ratio 3.46, 95%
condence interval 1.1210.62, P =.030).Tubesizeanddurationof
intubation were not signicant in the multivariable model. Median
scores of parent-reported voice quality of life on the Pediatric Voice
Handicap Index were markedly different for preterm (22) and term (3)
groups, P , .001.
CONCLUSIONS: Voice disorders in this population were much more
frequent than expected. Further studies are required to assess voice
across a broader range of gestational ages, and to investigate voice-
protective strategies in infants requiring multiple episodes of intu-
bation. Pediatrics 2013;131:e733e739
AUTHORS: Noel French, MBChB,
a
,
b
,
c
Rona Kelly, MBBS,
b
Shyan Vijayasekaran, FRACS,
d
Victoria Reynolds, BSc,
e
Jodi
Lipscombe, BSc,
e
Ali Buckland, BSc,
e
Jean Bailey, BSc,
e
Elizabeth Nathan, BSc,
f
and Suzanne Meldrum, PhD
g
a
Neonatal Clinical Care Unit, King Edward Hospital, Perth,
Western Australia;
b
State Child Development Centre, Health
Department of Western Australia, Perth, Western Australia;
c
Centre for Neonatal Research and Education, and
g
School of
Paediatrics and Child Health, University of Western Australia,
Perth, Western Australia; Departments of
d
Otolaryngology, and
e
Speech Pathology, Princess Margaret Hospital, Perth, Western
Australia; and
f
Biostatistics and Research Design Unit, Women
and Infants Research Foundation, Perth Western Australia
KEY WORDS
extremely preterm infants, long-term outcomes, endotracheal
tube, dysphonia, quality of life
ABBREVIATIONS
CIcondence interval
ELBWextremely low birth weight
ETTendotracheal tube
GRBASauditory perceptual assessment scale
ICCintraclass correlation coefcient
ORodds ratio
PDApatent ductus arteriosus
PVCpolyvinyl chloride
pVHIPediatric Voice Handicap Index
www.pediatrics.org/cgi/doi/10.1542/peds.2012-0817
doi:10.1542/peds.2012-0817
Accepted for publication Nov 19, 2012
Address correspondence to Noel French, MBChB, Neonatal
Clinical Care Unit, King Edward Hospital, 374 Bagot Rd, Subiaco,
Perth, Western Australia, 6008, Australia. E-mail: noel.
french@health.wa.gov.au
PEDIATRICS (ISSN Numbers: Print, 0031-4005; Online, 1098-4275).
Copyright © 2013 by the American Academy of Pediatrics
FINANCIAL DISCLOSURE: Dr Vijayasekaran is a consultant to
Ar throcare, which manufactures products used in ear, nose,
and throat surgery. These products were not used in relation to
this study. The other authors have indicated they have no
nancial relationships relevant to this article to disclose.
FUNDING: This study was supported by a grant from the Women
and Infants Research Foundation, Ethics Approval number 1828/EP.
PEDIATRICS Volume 131, Number 3, March 2013 e733
ARTICLE
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Page 1
Extreme preterm birth (,27 weeks
gestation) is associated with adverse
medical and social outcomes.
1
Morbidity
has been shown to be inversely related
to gestational age.
1
Long-term outcome
studies to school age and beyond have
identied a number of conditions more
prevalent in the surviving population of
extremely preterm infants.
25
Voice
quality has not been included in these
long-term outcomes and reported only
in isolated case reports.
6
Extremely pr eterm infants usually r equire
respir ato ry support, typically endotra-
cheal intubation, in the neonatal period
because of physiologic immaturity .
7
Dys-
phonia is a recognized complication of
endotracheal intubation. Laryngeal inju-
ries visualized postextubation in neonatal
subjects range fr om mild erythema,
edema, and granulation of the vocal folds
to arytenoid cartilage dislocation, sub-
glottic stenosis, laryngeal tears, and
avulsion of the vocal folds.
6,812
Surgical
ligation of a patent ductus arteriosus
(PDA) has been frequently associated
with left vocal fold paralysis in some se-
ries.
13
Several authors have identied
a relationship between intubation factors
and neonatal laryngeal injury .
9,10,12, 1419
It
remains unclear which factors, such as
length of intubation, frequency of reintu-
bation, and size of tube, are directly
linked to persistent laryngeal disorders
affecting voice quality.
In our long-term follow-up program, voice
abnormality has been a common nding,
particularly in children born at ,25
weeks gestation. Voice outcomes in
studies of children intubated in the neo-
natal period have so far been limited to
those undergoing laryngeal reconstruc-
tion for recognized airway complica-
tions
2022
or a fter surgical ligation of the
PDA,
23
and to extr emely low birth weight
(ELBW) infants at 12 months of corrected
age.
19
Mild voice abnormalities, such as
breathiness and roughness, ar e common
in young childrenbecause ofvocal overuse
in childhood.
24
Many of these cases will
self-resolve, but more severe abnormali-
ties require further investigation and in-
tervention. Voice assessments typically
involve auditory per ceptual measures,
quality-of-life assessments, and acoustic
analysis, although the latter is rarely
reported in children and has not been
validated in the pediatric population.
25,26
It is apparent that children as young as 5
are capable of reecting on the social,
emotional, and physical aspects of their
voice abnormalities, and consequent
negative emotional experiences have
been reported.
27
Among other things,
children with chronic dysphonia cite
concerns with participation limitation,
negative evaluation of their voice quality
by others in their social and academic
environments, and managing emotions,
such as frustration.
28
Therefore, dys-
phonia has signicant, negative effects
on the quality of life of affected children.
Little is known about the nature and
incidence of later voice disorders in
preterm children who were intubated
after birth. In this study, we aimed to
further explore demographic and in-
tubation factors associated with voice
abnormality in this population at school
age and to assess the voice-related
quality of life of these children.
METHODS
P atients were recruited from the neonatal
follow-up progr am of the single tertiary
neonatal service, which provides neonatal
intensive care to all extremely preterm
infants born in Western Austr alia, across2
sites at King Edward Memorial Hospital
and at Princess Margaret Hospital for
Children. Cases selected were ,25-week
survivors between 6 and 15 years of age,
born fr om 1996 to 2004, inclusive. Age
criteria were selected to optimize com-
pliance with voice assessments, which
require d reading a sample of connected
speech and producing prolonged vowel
sounds. Data from the neonatal follow-up
progr am were used to exclude cases in
which known disability was likely to pre-
vent adequate assessment, or in which
the family lived more than 200 km from
the study center.
Neonatal clinical variables were avail-
able from the NICU database, and in-
cluded demographic variables, clinical
morbidities, duration and type of re-
spiratory support, and duration of oxy-
gen therapy. In addition, chart review
was undertaken to determine additional
information relating to each intubation/
reintubation episode, such as type of
endotrachealtube(ETT),size ofETT, route
(oral or nasal), and body weight at the
time of each intubation episode. A ratio
of ETT size relative to body weight at
the time of intubation/reintubation was
created by dividing ETT size in milli-
meters by body weight in kilograms on
the day of the procedure. The maximum
ETT ratio of all intubation episodes was
recorded for each infant.
Throughout the study period, uncuffed
siliconized PVC (Portex, internal diam-
eter 2.03.5 mm) or uncuffed Ivory PVC
(Portex, internal diameter 2.53.5 mm)
ETTs were used. (Smiths Medical Aus-
tralia Pty Ltd, Brisbane, Australia)
Eight percent of all intubation episodes
were with Ivory tubes. Before 2000,
ETT placement was predominantly
nasotracheal after initial orotracheal
placement for labor ward resuscitation,
and after 2000, placement was pre-
dominantly orotracheal for all episodes.
Elective versus emergency reintubation
or degre e of difculty of intubation were
insufciently documented in the chart to
allow analysis of these variables. Surgi-
cal ligation of a PDA is rarely carried out
in extremely preterm infants in Western
Australia, but was recorded where this
occurred. Premedication befor e intu-
bation was not used in this population.
Voice Assessments
1. The GRBAS
29
is a widely used audi-
tory perceptual assessment scale
that determines ratings for each
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of 5 voice characteristics: G (grade
or severity of hoarseness), R
(roughness: abnormal variations in
pitch and loudness), B (breathiness:
occurs when there is excess air
heard in the voice), A (asthenicity:
weakness of the voice), and S
(strain: excess muscle tension used
to produce voice). Each parameter
is scored by using a 4-point ordinal
scale from 0 to 3 (0, normal; 1, mild;
2, moderate; and 3, severe), and
the mean of the values summed
as the mean GRBAS. The G severity
(hoarseness, 03 as above)
30
was
used as the primary outcome mea-
sure.
2. The Pediatric Voice Handicap Index
(pVHI) is a quality-of-life question-
naire completed by parents and
measures the impact of a voice dis-
order.
31
The pVHI is scored on a 5-
point Likert scale presented in 3
subscales each of 7 to 9 questions:
(1) functional (eg, At home we have
difculty hearing my child.), (2)
physical (eg, My child uses a great
deal of effort to speak.), and (3)
emotional (eg, My child is frus-
trated with his/her voice prob-
lem ). The maximum score
attainable from these questions is
92, whereas normophonic control
populations typically have total
scores #2.
A term-born comparison group of 40
children was also recruited and they
underwent the same measures. These
children were recruited with parental
consent from an orthopedic clinic of the
childrens hospital, and were all born at
term with no history of previous in-
tubation or recent respiratory illness.
Although not matched with the preterm
group, they were of similar age and
gender distribution.
The GRBAS was administered by 2
speech pathologists with postgraduate
experience in clinical voice assessment.
Inter- and intrarater reliability were
calculated on a random sample of 10%
of the raw voice samples.
Statistical Method
Continuous data were summarized by
using median, interquartile range, and
range, and categorical data were sum-
marized by using frequency distri-
but