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DEVELOPMENTAL MEDICINE & CHILD NEUROLOGY SYSTEMATIC REVIEW

Diagnostic accuracy of the clinical feeding evaluation


in detecting aspiration in children: a systematic review
IRENE CALVO1,2 | AIFRIC CONWAY1 | FILIPA HENRIQUES1 | MARGARET WALSHE1

1 Department of Clinical Speech and Language Studies, Trinity College, Dublin, Ireland. 2 Department of Neurorehabilitation Sciences, Casa Cura
Policlinico, Milano, Italy.

Correspondence to Irene Calvo at Department of Neurorehabilitation Sciences, Casa Cura Policlinico, Via Giuseppe Dezza 48, 20144 Milano, Italy. E-

mail: calvoi@tcd.ie. This article is commented on by Sellers on page 530 of this issue.

The aim of this systematic review is to determine the diagnostic accuracy of clinical feeding
PUBLICATION DATA been reported across several different paediatric clinical
Accepted for publication 7th December 2015.
conditions.3,4 OPA represents a considerable threat to
Published online 9th February 2016.
respiratory and general health status in children with severe
medical conditions10 and significantly impacts the quality of
ABBREVIATIONS
CFE Clinical feeding examination FEES Fibre-optic endoscopic evalua tion life of children and caregivers.11 The main con sequences of
of swallowing OPA include malnutrition, dehydration, fail ure to thrive,
OPA Oropharyngeal aspiration SLT Speech and language therapist SROC progressive lung disease, bronchiectasis, respiratory failure,
Summary receiver operating characteristic chronic lung disease, recurrent wheez ing, intermittent stridor,
VFSS Videofluoroscopic swallowing study atelectasis, recurrent pneumonia or respiratory infections,
evaluation (CFE) compared to instrumental assessments in detecting aspiration pneumonia, and even tually death. 11,12 Therefore,
oropharyngeal aspira tion (OPA) in children. This is important to support early recognition of OPA is essential in providing prompt and
clinical decision-making and to provide safe, cost-effective, higher quality appropriate management of hydration and nutrition, avoiding
care. All published and unpublished studies in all lan guages assessing the the exacerbation of respiratory consequences, and
diagnostic accuracy of CFE compared to videofluoroscopic swallowing decreasing the morbidity and mortality associated with
study (VFSS) and/or fibre-optic endoscopic examination of swallowing
aspiration.10
(FEES) in detecting OPA in paediatric populations were sought. Databases
were searched from inception to April 2015. Grey literature, citations, and Children with oropharyngeal dysphagia at risk of aspira tion
references were also searched. Two independent review ers extracted and are usually referred for a clinical feeding evaluation (CFE). 13
analysed data. Accuracy estimates were calculated. Research reports were A thorough CFE usually involves recording of medical history,
translated into English as required. Six studies examining the diagnostic an oral motor evaluation of the swallowing structures at rest,
accuracy of CFE using VFSS and/or FEES were eligible for inclusion. and feeding trials.14,15 A CFE is adminis tered by a trained
Sample sizes, populations studied, and CFE characteristics varied widely. therapist, usually a speech and language therapist (SLT) or
The overall methodological quality of the studies, assessed with QUADAS-
an occupational therapist. Clinicians use the occurrences of
2, was considered ‘low’. Results suggested that CFEs trialling liquid
consisten cies might provide better accuracy estimates than CFEs trialling different clinical markers12,13,16–18 and especially the presence
solids exclusively. This sys tematic review highlights the critical lack of of a cough during or after oral tri als as guides to detect OPA.
evidence on the accuracy of CFE in detecting OPA in children. Larger well- Some CFEs may include the use of cervical auscultation 19
designed primary diagnostic test accuracy studies in this area are needed and monitoring by pulse oximetry.20 However, OPA can
to inform dysphagia assessment in paediatrics. sometimes occur without the manifestation of a cough or
other overt sign of aspira tion (change in vocal quality,
breathing patterns, etc.). This condition is defined as silent
aspiration.21–23 Because of the lack of overt signs, silent
Advancements in neonatal care have resulted in an increase
in the incidence of feeding and swallowing disorders in the aspiration may not be recognized during clinical assessment
and thus recommendations for oral feeding may put the child
paediatric population.1–3 Oropharyngeal aspiration (OPA), at risk of developing more severe medical and respiratory
defined as the passage of secretions, food, liquid, or saliva consequences.
below the true vocal cords into the respiratory airways, 4,5 is There is no universally accepted standard CFE. 24 A wide
one of the most harmful consequences of oropharyngeal
range of CFEs are used in clinical practice, and many are
dysphagia. The exact epidemiology of OPA across different
neither validated nor established to be reliable. Therefore, it
medical conditions is unknown;3 its presence is signifi cantly is difficult to know which CFEs are best for detecting OPA. A
associated with medical diagnoses of developmental delay detailed CFE provides direct recommendations for the
and cerebral palsy.6–9 Nonetheless, occurrences of OPA have management of patients and informs about the need

© 2016 Mac Keith Press DOI: 10.1111/dmcn.13058 541


for further instrumental assessment of swallowing safety.24 detecting the presence of OPA and silent aspiration in the
Performing objective instrumental assessments is consid paediatric population.11 Both evaluations provide good
ered to be the best practice in diagnosing the presence of agreement rates in detecting aspiration events in both adults
aspiration.25, 26 The videofluoroscopic swallowing study and children.27–29 However, despite the growing number of
(VFSS) and fibre-optic endoscopic evaluation of swallow ing requests for VFSS and FEES for paediatric patients, they
(FEES) are considered the most reliable assessments for may not always be readily available in all clinical settings.
Furthermore, instrumental assessments are by their nature Allied Health Source, ProQuest Dissertations and Theses
brief and may not detect all occurrences of OPA Global) from inception to April 2015. Text words were
episodes.10,11,30 They require the cooperation of the child, combined and database subject headings were used that
and their administration can invoke fear and anxiety in the were synonymous with the target condition, index test, and
child, thus they may not reflect the child’s typical feeding and reference standard tests. Appendix SI (published online)
swallowing ability.31 In addition, they require specialist provides a complete audit trail of the search strate gies. The
healthcare practitioners and specific machines, and can conference proceedings from the last 5 years of the
therefore be highly expensive procedures. More over, the European Society for Swallowing Disorders, the Dysphagia
invasiveness and the risks associated with their Research Society, and the UK Swallowing Research Group
administration limit their use in children with critical clini cal were manually searched. Authors of potentially relevant
conditions for safety reasons. abstracts were contacted to obtain fur ther data. Two
On the contrary, CFE is not considered accurate for scientific journals related to the area of dysphagia and CFEs,
detecting OPA, particularly in view of the high prevalence of Dysphagia and International Journal of Speech-Language
silent aspiration reported in children.6,9,32,33 However, it is a Pathology, were manually searched for relevant articles.
common procedure that is readily available and gener ally Citation alerts were set for the search strategies in each
feasible. It is low cost and can be performed across short electronic database, where possible. In PubMed the ‘Related
time periods. In addition, it is the assessment that most Articles’ function was used to further searching. Citations of
closely resembles normal mealtimes, it can be per formed in the selected studies were sought using Scopus, Web of
a variety of clinical settings, and it is widely used in clinical Science, and Google Scholar. The reference lists of the
decision-making. A recent study by Cocks and Ferreira 34 included studies were checked for other potentially relevant
revealed that UK SLTs rely more on clinical than instrumental studies.
assessments when making decisions about oral versus non-
oral feeding. Inclusion/exclusion criteria
Aiming for an early diagnosis of OPA, in order to avoid All published and unpublished studies in all languages were
serious consequences to the child’s health, means there is a included if they reported the accuracy of CFE compared to
need for an accessible, valid, and reliable diagnostic tool to VFSS and/or FEES in detecting OPA in children. Index tests
be used in the paediatric population. Therefore, some studies were defined as all CFEs conducted to detect aspira tion in
have attempted to investigate the sensitivity and specificity of paediatric populations. These tests were typically
CFE for detecting OPA in children. 13,16 However, the administered by an SLT or occupational therapist testing
evidence is conflicting and not sufficiently clear to make a swallowing function using different fluid and solid consis
strong impact on clinical practice. System atic reviews have tencies according to the development of the child’s feeding
been published looking at the diagnostic accuracy of CFE in abilities. These CFEs included assessment of the presence
detecting OPA in adult populations. 35– 41 In these studies, or absence of OPA events for each participant. Reference
FEES and VFSS were used as the crite rion or reference standard tests were VFSS and FEES. Populations were
standard tests. However, given the diver sity of populations children presenting with a history of swallowing problems.
and CFEs, these reviews cannot be used to infer conclusions Papers were included if participants were aged 18 years or
about paediatric populations. Despite the growing interest in less and presented with oropharyngeal dysphagia as a pri
this area of research, to date no systematic reviews have mary cause of OPA. There were no restrictions based on the
been completed that assess the diagnostic accuracy of diagnosis of underlying causes of oropharyngeal dys phagia.
paediatric CFE in detecting OPA. The aim of this systematic Any clinical and care setting was considered,
review is to examine the diag nostic accuracy of CFE because paediatric dysphagia is a condition routinely diag
compared to instrumental assess ments for the detection of nosed and managed in various clinical settings from acute
OPA in children. The objective is to address the lack of care hospital wards to outpatient services or community
evidence on the diagnostic accu racy of CFEs and provide clinics. The personnel conducting the CFEs had to be pro
clinicians with evidence to fessionals involved in dysphagia management. Studies with
participants of mixed ages were excluded whenever it was
542 Developmental Medicine & Child Neurology 2016, 58: 541–553 not possible to extract the data about the relevant age group
of interest. Studies with participants who had only secondary
What this paper adds
aspiration were excluded, because this kind of aspiration is
• Reviews the evidence on diagnostic accuracy of paediatric clinical
feeding examinations (CFEs) for detecting aspiration. related to dysfunction of the oesophageal phase and the
• Suggests using liquids rather than solids to enhance CFE sensitivity. • presence of gastroesophageal reflux.
Identifies a critical lack of evidence on accuracy of paediatric CFEs. •
Directs further research on diagnostic accuracy of CFE using different Data extraction and quality assessment
consis tencies. One reviewer (IC) screened titles and abstracts. Full texts of
potentially eligible abstracts were screened by two inde
inform clinical decision-making. In this systematic review the
pendent reviewers (IC and AC for the studies written in
target condition being assessed is OPA, the index test is
English; IC and FH for the studies written in Portuguese and
CFE, the reference standard tests are VFSS and/or FEES,
Spanish). Disagreements were resolved by a third author
and the population of interest is paediatric patients.
(MW). Data were extracted by two independent authors (IC,
MW) using a specific data extraction form and completing a
METHOD 292 contingency table. True-positive, true-negative, false-
Search strategy positive, and false-negative results for index and reference
A systematic search was conducted in 10 electronic data standards were recorded on this 292 table for each included
bases (PubMed, EMBASE, CINAHL, PsycINFO, Scopus, study. The methodological quality of the included studies was
Web of Science, LILACS, SciELO, ProQuest Nursing and assessed by two independent reviewers (IC, MW) using the
QUADAS-2 tool.42 Four domains were rated as presenting the CFE.46,48 Three studies presented different data sets for
‘high’, ‘unclear’, or ‘low’ risk of bias and ‘high’, ‘unclear’, or different consistencies trialled in the CFE. DeMatteo et al. 16
‘low’ concerns regard ing clinical applicability. Disagreements differentiated between liquids and semi-solid foods, while
were resolved by discussion. Foroni et al.48 reported results using liquids and pur ees
separately. In addition, Beer et al. 50 provided sepa rate data
Data analysis for saliva, thin liquids, and pur ee trials. There were no
The type of data in this review was binary or dichotomous, specific definitions of the terminology used to refer to the
where the presence or absence of OPA was reported as different consistencies in any of the studies.
present or absent, or as positive or negative. The analysis of Overall, the methodological quality of the studies was
the data was carried out using RevMan software (Ver sion considered to be ‘low’. Four studies were considered at high
5.3. Copenhagen: The Nordic Cochrane Centre, The risk of bias; five out of six studies raised high con cerns
Cochrane Collaboration; 2014). Data were analysed and regarding clinical applicability. An assessment of the
presented separately for each reference standard test as rec methodological quality of the included studies is pre sented
ommended by Naaktgeboren et al. 43 In the case of studies graphically in Figures 2 and 3. The study by Sui ter et al. 47
providing separate data for different consistencies trialled in was judged to have a high risk of bias for the index test
CFE and instrumental evaluations, the data sets refer ring to domain, as the SLT who administered the CFE had
liquid and food consistencies were considered and analysed previously administered the reference standard test, and thus
independently for each study. The following esti mates of was not blinded to its results. Three studies were judged at
diagnostic test accuracy were calculated: sensitiv ity, high risk of bias for flow and timing. The domain of flow and
specificity, positive predictive value, negative predictive timing refers to the methods of administration of both the
value, and likelihood ratios. Estimates of sensitivity and index and the reference stan dard tests. It considers whether
specificity for each primary study were displayed in cou pled all patients received the same reference standard, and
forest plots and summary receiver operating charac teristic whether they were all
(SROC) plots.44 A meta-analysis was planned to summarize
sensitivity and specificity estimates of CFE in detecting OPA. Review 543
The summary statistic chosen was the average operating
point, which is applicable to the dichoto mous nature of OPA,
obtained using the bivariate model. 45 We also planned to
investigate the sources of heterogene ity using a meta-
regression approach applied to the bivari ate model. The
following covariates were individuated:
different age groups; different clinical diagnosis; the use of
other tools during CFE.

RESULTS
The electronic search identified 8785 records. Five addi tional
records were retrieved through manual searching. In total, 40
potentially eligible studies were assessed for inclu sion.
Thirty-four records were excluded; Appendix SII (published
online) lists the excluded studies and reasons for exclusion.
Six studies met the inclusion criteria for this review. 16,46–50
The process of inclusion is shown in Figure 1.
All the included studies were published in the period
between 2003 and 2014 in peer-reviewed scientific jour nals.
Four were prospective studies,16,46,47,50 one was a ret
rospective study,48 and in one study the method of data
collection was not reported (Table I).49 The sampling method
was consecutive in two studies16, 50 and unclear in the rest of
the studies. All studies were carried out in ter tiary care
settings and university hospitals. Three studies were set in
Brazil,46,48,49 one in Canada,16 one in Ger many,50 and one in
the USA.47 Overall, the sample size of the included studies
was small, ranging from 4 to 93 chil dren.46,48 The population
sample varied within the studies, but all samples comprised
participants with different neu rological disabilities. Definitions
of OPA were provided in all but one study. 48 The CFEs used
in the studies were dif ferent (Table I), and none were
validated or standardized tools. CFEs were administered by
SLTs in three stud ies46,47,50 and by one SLT and
occupational therapists in one study;16 two studies did not
provide clear information about the administrators.48,49 All but
two CFEs49,50 were administered using a standard protocol.
Two CFEs reported the use of cervical auscultation to support
database search

(n=8785)

Records after duplicates


removed

(n=6730)

Titles screened for eligibility Excluded based on title (n=6730) (n=6479)


Records identified through hand searching:

Conference proceedings (n=1)

Potentially relevant articles from references of included reports (n=2)


Abstracts screened for eligibility Potentially relevant articles Excluded based on full text (n=31) Full

(n=251) (n=40) text not retrievable (n=3)

Excluded based on abstract

(n=211)
Included studies

(n=6)

Figure 1: Flow diagram showing the process of selection of records and studies for the review.

included in the analysis. It also looks at the time interval 544 Developmental Medicine & Child Neurology 2016, 58: 541–553
between carrying out the two examinations. In this review, received swallowing therapy between the clinical and the
considering the episodic nature of OPA events and the instrumental evaluation.
influence that health and clinical conditions have on its Applicability concerns were raised in the patient selec tion
occurrence, a time interval of less than 24 hours was domain for four studies, as their sample population might not
generally judged to be appropriate. In the study by Araujo et be representative of patients in clinical practice or did not
al. 46 the time interval varied between 7 days and 20 days. exactly match the review population. In two studies 16,46 the
This was considered too long, especially because there was sample population represented a rather small age range. In
not enough information on the nature and severity of the one study48 the sample population was relatively small and
clinical conditions of the sample popu lation. Moreover, two included only patients with a rare genetic syndrome that is
patients were excluded from the final analysis without fairly unusual in common clinical practice. In one study 49 all
16
explanation. DeMatteo et al. pre sented a more acceptable participants presented with a diagnosis of severe cerebral
time interval (the VFSS was administered on the same day or palsy, and therefore the sample population already had a
within 48 hours of the CFE); however, not all participants higher risk of OPA. The index test used by Suiter et al. 47 (3oz
50
were included in the analysis. Beer et al. administered the water swallow test) raised applicability concerns because it
FEES from 1 day to 5 days after the CFE, and not all was not believed to be completely adequate for the
participants were included in the analysis; moreover, children population studied. Consecu tive drinking is a skill that may
might have not be completely devel oped by young children and may put
children at higher
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546 Developmental Medicine & Child Neurology 2016, 58: 541–553


Patient selection 50% 75% 100% 0% 25% 50%
Index test
Reference standard
Flow and timing

0% 25%
75% 100%

Risk of bias Applicability concerns

High Unclear Low

Figure 2: Risk of bias and applicability concerns graph. The review authors’ judgements about each domain are presented as percentages of
the included studies.

Risk of bias Applicability concern

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it w it

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Araújo et al. (2014) ? ++ ++ – –

+ + ???? ++++++
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––

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DeMatteo et al. (2005) solids

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+++++

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+
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High Unclear Low

Figure 3: Risk of bias and applicability concerns summary. The review authors’ judgements about each domain are shown for each included study.

risk of aspiration and penetration. 51,52 In addition, the


amounts and consistencies of food used in the index test
differed from those trialled in the reference standard test.
One study49 raised concerns in the reference standard
domain because the presence of OPA was recorded only
when the inhaled material was not spontaneously expelled
from the airways.
Four studies compared CFEs with VFSS.16,46,48,49 Esti
mates of the accuracy of CFE compared to VFSS are pre
sented in Table II. The prevalence of OPA in the study
samples based on VFSS findings ranged from 18% to 84%;
however, the characteristics of the sample population and the
number of patients enrolled varied widely between the
primary studies. Sensitivity estimates were highly vari able,
and ranged from 0.17 (95% confidence interval [CI] 0.05–
0.37) to 0.93 (95% CI 0.76–0.99). Specificity esti mates
ranged from 0.00 (95% CI, 0.00–0.52) to 1.00 (95% CI 0.16–
1.00). As a general trend, estimates of sensitivity

Review 547
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t

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n

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specificity
t

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ranged
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l

t
(95% CI
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s
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(95% CI
s

0.04–0.78)
and from
m

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N

were
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when 0.67) to
estimates 0.14 (95%
of CI 0.02–
specificity 0.43)
were low, respectivel
y. Com was systematic widely reported by values al.48 and When been cohort of in these
plete considered review within the Furkim et more Beer et trialling reported in 300 studies. In
estimates inappropria investigate included al.49 is high consistent al.50 both liquids the children. the study
of te because d the studies. and might with found that
exclusively literature Therefore, by Furkim
accuracy of the diagnostic The be current
the preva
, CFEs that liquids aspiration et al.,49
accuracy prevalence 9 showed are of liquids
are heterogene explained literature. lence rate which
reported in ity of study of CFE in of OPA in by the DeMatteo of OPA consis generally may be included
Table III. designs, detecting this review characteris et al.16 was almost
tently aspirated more both liquids
Sensi tivity participant OPA in varied tics of their found the the same higher more than easily and food of
and children widely from sample sensitivity food of detected smooth pur
s, index difference with liquids
specificity tests, and 18% to population, values thicker by CFE. ee
in OPA and pur
estimates target included 84%, as (range consistenc Bolus consistenc
prevalence ees. 6,7,13,51,54–control is
six studies possibly as dysphagia 1.00– y. y, sensi
are condition comparing a result of to be The food 16,47,48 56
displayed characteris is more consistent consistenc 0.92) This is more diffi tivity
CFE to the prevalent ,50 cult and estimates
in a tics, and VFSS or differences with the y used in sup ported
in children aspiration might have
coupled methodolo FEES. literature the CFE compared by Weir et
in sample with severe 9 may occur been
forest plot gical claiming influenced to CFEs al. who
Sample sizes and cere bral more influenced
(Fig. 6) quality of popula that liquids not only using food found that
clinical pop palsy.53 In readily by the high
and an the are more the of thicker thin liquids
tions, CFE ulations before prevalence
SROC plot included characteris investigate contrast, frequently prevalence consistenci were more swallow
other aspirated rate, but es (range overtly of
(Fig. 7). studies. tics, and d in the ing liquids.
studies 16,46 than
also 0.93– aspiration
Pooling of estimates primary aspirated This might
,47,50 solids. 9,54
measures 0.17). 16,46,4 in their
the studies DISCUSSI of studies. than other explain
of 8–50 sample
in a meta- ON accuracy The value reported However, It has consistenci higher population,
analysis This varied of 84% prevalence Foroni et accuracy. frequently es in a sensitivity as

548 Developmental Medicine & Child Neurology 2016, 58: 541–553


Study TP FP FN TN Sensitivity (95% CI) Sensitivity (95% CI) Specificity (95% CI)
Specificity (95% CI)
Araújo et al. (2014) 4 2 20 65 0.17 (0.05, 0.37) 0.97 (0.90, 1.00)
DeMatteo et al. (2005) 22 19 16 0.46 (0.29, 0.63)
liquids 2 0.92 (0.73, 0.99)
17 2 0.33 (0.04, 0.78)
4 DeMatteo et al. (2005) solids 0.65 (0.44, 0.83)
9
2 1 0011 1.00 (0.16, 1.00) 0.50 (0.01, 1.00 (0.16, 1.00) 0.50 (0.01,
Foroni et al. (2010) liquids Foroni et al. (2010) purée 21 0.99) 0.99)
Furkim et al. (2003) 0 0.00 (0.00, 0.52) 0 0.2 0.4 0.6 0.8 1 0 0.2
25 5 2 0.93 (0.76, 0.99) 0.4 0.6 0.8 1

Figure 4: Coupled forest plot of the estimates of sensitivity and specificity in videofluoroscopic swallowing studies. TP, true positive; FP, false
positive; FN, false negative; TN, true negative.

is

1
3
5

1
0.9

0.8

0.7

2 0.6

0.5

4
0.4

0.3
6
0.2

0.1

0
10
0.9 0.8 0.7 0.6 0.5 0.4 0.3 0.2 0.1
Specificity

Figure 5: Summary receiver operating characteristic plot of accuracy of clinical feeding examination compared to videofluoroscopic swallowing
study. 1. Furkim et al. (2003). 2. Foroni et al. (2010) pur ee. 3. Foroni et al. (2010) liquids. 4. DeMatteo et al. (2005) solids. 6. Ara ujo et al. (2014).
○, Study estimate.

Table III: Estimates of accuracy of clinical feeding examination compared to fibre-optic endoscopic evaluation of swallowing

Prevalence

Study n Sens (95% CI) Spec (95% CI) (0.19–1.00) 1.16 (0.94–1.44) 0.00 (0.00–NaN) 22 0.33 (0.04–0.78) 0.44 (0.20–
(95% CI) PPV (95% CI) NPV (95% CI) PLR (95% CI) NLR (95% CI)

Beer et al. (2014) 0.70) 0.27 (0.11–0.50) 0.18 (0.03–0.52) 0.63 (0.31–0.87) 0.59 (0.17–1.98) 1.52
liquids
Beer et al. (2014)
puree (0.73–3.14) 56 1.00 (0.75–1.00) 0.51 (0.35–0.67) 0.23 (0.13–0.36) 0.38 (0.22–
Suiter
et al.
(2009) 0.56) 1.00 (0.81–1.00) 2.04 (1.50–2.78) 0.00 (0.00–NaN)
21 1.00 (0.59–1.00) 0.14 (0.02–0.43) 0.33 (0.15–0.56) 0.36 (0.17–0.61) 1.00

Sens, sensitivity; CI, confidence interval; Spec, specificity; PPV, positive predictive value; NPV, negative predictive value; PLR, positive
likelihood ratio; NLR, negative likelihood ratio.

Review 549
Study TP FP FN TN Sensitivity (95% CI) Specificity (95% CI) Sensitivity (95% CI) Specificity (95% CI)
Beer et al. (2014) liquids 7 2 4
12 9
2 1.00 (0.59, 1.00) 0.14 (0.02, 0.43) 0 Beer et al.
13
Suiter et al. (2009)
21 0 0.2 0.4 0.6 0.8 1 0 0.2 0.4 0.6 0.8 1
22 1.00 (0.75, 1.00) 0.51 (0.35, 0.67)
0
Figure 6: Coupled forest plot of sensitivity and specificity estimates in fibre-optic endoscopic evaluation of swallowing studies. TP, true
positive; FP, false positive; FN, false negative; TN, true negative.

is

S
0.9
Suiter et al. (2009) Beer et al.
1
0.8

0.7

0.6

0.5

0.4

0.3

0.2
Beer et al. (2014) purée
0.1

0 0.7 0.6 0.5 Specificity 0.4 0.3 0.2 0.1 0


1 0.9 0.8 Study estimate

Figure 7: Summary reciever operating characteristic plot of accuracy of clinical feeding examination compared to fibre-optic endoscopic evaluation of
swallowing.

higher disease prevalence and higher disease severity are the included CFEs. However, the influence of disease
associated with higher sensitivity values.57 prevalence on a test’s sensitivity and specificity is difficult to
Conversely, higher levels of specificity occurred in CFEs determine, and there is no specific correlation between
trialling food of solid or pur ee consistency 16,46,48–50 com disease prevalence and accu racy estimates. 58 Beer et al.50
pared to the ones using liquids. Specificity values with food of reported the same prevalence rates for liquid and solid trials
thicker consistency ranged between 0.44 and 0.97. DeMatteo using CFE, and found con
et al.16 explained the poor performance of CFE trialling solids
550 Developmental Medicine & Child Neurology 2016, 58: 541–553
by relating it to the low prevalence of OPA events with solids:
their prevalence rate for OPA with solids was the lowest of all siderably higher sensitivity estimates for liquids and higher
specificity estimates for solids. aspirate,9 further research should focus on the influence of
Even if a test does not produce acceptable values for both silent aspiration in diagnosing OPA during clinical
sensitivity and specificity, it could still be used in clinical examination. Furthermore, there is still a lack of evidence
practice as a guide to rule in or rule out the presence of the concerning the accuracy of cervical auscultation in supporting
disease when either of the two values is very high. The CFE the diagnosis of aspiration.
used by Araujo et al. 46 provided a specificity value of 0.97
and might be considered a good test to rule in OPA events.
Its positive likelihood ratio of 5.58 confirms the moderate REFERENCES
increase in the likelihood of OPA with a positive result in the To conclude, we have a number of recommendations
CFE. Conversely, three CFEs16,47,50 in this review might be based on the findings of this review. Firstly, researchers and
informative in ruling out the occurrence of OPA events. The clinicians must be consistent in the terminology used in
CFEs used by DeMatteo et al.,16 Beer et al.,50 and Suiter et reporting clinical assessments.37 Most of the difficulties in the
al.47 pro vided remarkable sensitivity and negative predictive interpretation of the studies were caused by incon sistency in
value the definitions of OPA, silent aspiration, and laryngeal
estimates with liquid trials; therefore they may be proposed penetration and in the different terms used for solid and liquid
as valid tests in which a negative CFE result can exclude the consistencies.61 Moreover, discrepancies still exist in the
presence of OPA. Nonetheless, it must be remembered that definition of what constitutes ‘screening’ and ‘diagnostic’ tests
predictive values are dependent on the prevalence of the for dysphagia.62 Secondly, CFEs vary widely in clinical
disease in the sample population and change accord ingly in
practice,24 and universally accepted stan dard protocols for
different settings and populations.59,60 Therefore, the ability of VFSS and FEES to be used with chil dren are not yet
a CFE to rule in or rule out OPA cannot be reliably applied to
available.55 Therefore there is a need to establish consistent
every clinical situation.59,60 and reliable standards of practice for carrying out CFEs and
To our knowledge, this is the first systematic review to instrumental evaluations.63 Deter mining the inter- and
investigate the diagnostic accuracy of CFE in detecting OPA intrarater reliability of tests currently utilized in clinical practice
in the paediatric population. It highlights a critical lack of is fundamental for research and clinical practice. Reaching
evidence concerning the accuracy of CFE when diagnosing consensus on the components of the CFE would be a first
OPA, with only six studies included. Because of the foreseen step in the development and test ing of a consistent and
limited research in the area of paediatric dysphagia, wide reliable assessment protocol.
inclusion criteria were set for this review, resulting in low Finally, it is essential that large and well-designed pri mary
overall methodological quality and con siderable diagnostic accuracy studies are conducted in order to
heterogeneity of the included studies.57 As a result, one estimate the accuracy of CFE in detecting OPA. Accurate
acknowledged limitation of this review was the impossibility of methodological rigour is rare in diagnostic accuracy stud ies,
carrying out a meta-analysis. Moreover, the small sample and adherence to STARD guidelines64,65 is paramount,
sizes and heterogeneity of the sample populations pose a because better reporting will translate into easier searches
threat to the applicability of the review findings. and more available evidence.66
The preliminary findings of this review may indicate that
tests using liquids provide better accuracy estimates. How
ACKNOWLEDGEMENTS
ever, larger primary studies are needed to support this finding
The authors have stated that they had no interests that might be
and investigate the diagnostic accuracy of tests using
perceived as posing a conflict or bias.
different consistencies. Moreover, as accuracy changes
according to prevalence rates, studies focusing on specific
paediatric populations or clinical diagnoses might offer SUPPORTING INFORMATION
findings more relevant and directly applicable in clin ical The following additional material may be found online:
practice. In addition, as children are more likely to silently Appendix SI: Electronic databases search strategy.
Appendix SII: List of excluded studies.

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