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Original article

Diagnostic accuracy of lateral neck radiography


in ruling out supraglottitis: a prospective
observational study
Takashi Fujiwara,1 Hiroshi Okamoto,2 Yasuhiro Ohnishi,3 Toshio Fukuoka,4
Kazuyuki Ichimaru5
1
Department of Otolaryngology ABSTRACT
Head and Neck Surgery, Ehime Objective To assess the diagnostic accuracy of lateral Key messages
University, Toon City, Ehime,
Japan neck radiographs (LNR) for acute supraglottitis in adults.
2
Department of Emergency Design A single centre prospective observational study.
What this paper adds
Medicine, Kurashiki Central Setting Emergency department at Kurashiki Central
Hospital, Kurashiki City, ▸ Observational studies have suggested that
Hospital, Japan.
Okayama, Japan lateral neck radiographs can be used to rule
3 Participants Adult patients who underwent LNR to
Department of Radiology, out supraglottitis, but the diagnostic accuracy
Kurashiki Central Hospital, detect supraglottitis.
of the test in prior studies may be affected by
Kurashiki City, Okayama, Japan Main outcome measures Presence of supraglottitis,
4 spectrum bias.
Department of Emergency based on nasopharyngeal laryngoscopy or a follow-up
Medicine, Kurashiki Central ▸ In this prospective study, lateral neck
telephone call, 7–30 days after the visit.
Hospital, Kurashiki City, radiographs had a PPV of 58.6% and NPV of
Okayama, Japan
Results 140 patients had LNR during the study period.
94.5%, with a negative LR of 0.22.
5
Department of Otolaryngology 35 patients were excluded from further analysis because
Radiographs could not rule out all cases of
Head and Neck Surgery, of lack of consent. Of the 105 eligible patients,
Kokura Memorial Hospital, supraglottitis in this prospective study and
21 patients (20%) were given the diagnosis of
Kitakyushu City, Fukuoka, appears to miss milder cases.
supraglottitis: 17 of 29 with a radiographic abnormality,
Japan
and 4 of 76 patients without a radiographic abnormality.
Correspondence to Three of the four cases where LNR was negative was
T Fujiwara, Department of grade 1, and all cases of grade 3 or higher had
Otolaryngology Head and Neck settings.3 4 Abnormal findings on LNR in patients
abnormal LNR. Sensitivity and specificity (95% CI) of
Surgery, Ehime University, with supraglottitis are thickening of the epiglottis,
LNR for supraglottitis were 81.0% (64.2 to 97.7) and
Shitsukawa, Toon City, Ehime called the ‘thumb sign’,5 6 and a poorly defined val-
791-0295, Japan; 85.7% (78.2 to 93.2), respectively. The positive
lecula air pocket, termed the ‘vallecula sign’.7
t.fujiwarabi@gmail.com predictive value of LNR was 58.6% (40.7 to 76.5) and
Previous studies reported high sensitivity (83–
the negative predictive value was 94.7% (89.7 to 99.8).
Received 19 October 2013 100%) and specificity (87–100%) of LNR for
The positive likelihood ratio of LNR was 5.67 (3.27 to
Revised 21 February 2014 supraglottitis.6–8 However, these retrospective
Accepted 28 March 2014 9.82) and the negative likelihood ratio was 0.22 (0.10
studies calculated diagnostic accuracy using
Published Online First to 0.51).
20 August 2014 inappropriate spectrum patients. The diagnostic
Conclusions LNR showed only moderate sensitivity
value of LNR was calculated using patients with
and specificity for supraglottitis and would miss some
supraglottitis diagnosed by laryngoscopy. Generally,
cases of supraglottitis if the pre-test probability is high.
LNR are used to screen for, and laryngoscopy is
LNR was very sensitive for grade 3 or higher supraglottitis,
not always performed in patients who have normal
but would miss milder cases.
LNR.6 7 Previous studies did not sufficiently
Trial registration UMIN000011928.
address those patients who had radiographs but did
not have laryngoscopies. Also, the specificity of
LNR was calculated using patients with trauma and
INTRODUCTION
foreign bodies as the control group,7 although
Supraglottitis is an inflammation of the epiglottis
LNR of lingual tonsillitis patients, who present
and surrounding soft tissues, and can cause sudden
with similar symptoms to supraglottitis, may also
life threatening airway obstruction which may
have abnormal radiographs.9 Spectrum bias is asso-
require emergency airway management.1 The most
ciated with a risk of overestimating the test accur-
common symptom of supraglottitis is a sore throat;
acy.10 Therefore, we conducted a prospective
however, this is a very common symptom in the
validation study to determine the diagnostic accur-
emergency setting, resulting mostly from self-
acy of LNR in the diagnosis of supraglottitis in
resolving diseases, such as the common cold.
adults.
Ruling out supraglottitis is thus challenging for the
emergency physician. Direct visualisation of the
epiglottis by nasopharyngeal laryngoscopy or indir- METHODS
ect laryngoscopy is the gold standard for a diagno- Study population and protocol
sis of supraglottitis; however, these tests require We performed a single centre, non-interventional,
To cite: Fujiwara T, special training and expertise.2 Soft tissue lateral prospective study in the emergency department of
Okamoto H, Ohnishi Y, et al. neck radiographs (LNR) are a common procedure Kurashiki Central Hospital, from 16 April 2011 to
Emerg Med J 2015;32: for screening for supraglottitis because they are 31 March 2013. The study was conducted and
348–352. easily obtained and available in most clinical reported in accordance with Standards for
348 Fujiwara T, et al. Emerg Med J 2015;32:348–352. doi:10.1136/emermed-2013-203340
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Original article

Reporting of Diagnostic Accuracy principles.11 We prospectively otolaryngologists (TaF and KI) classified the findings according
recruited consecutive adult patients (>15 years old) whom had to Katori’s laryngoscopic grading system: grade 1: the patient’s
undergone LNR to evaluate supraglottitis. LNR were performed epiglottis is slightly swollen with the entire length of the vocal
if patients had a sore throat, odynophagia, drooling, voice folds visible by scope; grade 2: the patient’s epiglottis is moder-
change or dyspnoea. The emergency department physicians ately swollen with more than half of the posterior vocal folds
decided whether the patients needed LNR, and trained radio- visible by scope; and grade 3: the patient’s epiglottis is severely
logical technologists performed the LNR. Nasopharyngeal laryn- swollen with less than half of the posterior vocal folds visible by
goscopy was performed in patients with radiographic scope (figure 1).14 Emergency department physicians decided
abnormalities, as well as in those without abnormalities but in whether to prescribe antibiotics before or after the laryngoscopy
whom the emergency physician still suspected supraglottitis. All or if the patient did not undergo the procedure. For those who
enrolled patients were followedup by telephone interview after did not undergo nasopharyngeal laryngoscopy during the visit,
7–30 days. Planned exclusions were patients who did not provide we conducted a follow-up telephone interview after 7 days
consent or who were not followed up by telephone interview. because supraglottitis generally worsens within 7 days. If we did
For each enrolled patient, data on patient characteristics, not reach participants at this time, we called repeatedly in an
symptoms and the presence of supraglottitis were collected from attempt to reach them within 30 days of the visit. If the patient’s
the medical records. The institutional review board of Kurashiki symptoms had resolved, we defined the outcome as negative for
Central Hospital approved the study. supraglottitis. If the patient visited the emergency department
In contrast with therapeutic or interventional studies, formal repeatedly, we defined the outcome as positive or negative based
sample size calculations based on power assumptions for diag- on the diagnosis at the subsequent emergency department visits.
nostic modelling cohort studies do not exist. As we intended to We also asked whether patients were prescribed antibiotics from
roughly calculate the sensitivity and specificity of LNR in the other hospitals or physicians.
clinical setting, we defined the sample size as 18 patients with
supraglottitis; this number is similar to the numbers in previous
Statistical analysis
studies.5–7 The incidence of supraglottitis in adults is 1–3 per
Using the laryngoscopic diagnosis of supraglottitis or the tele-
100 000/year.12 13 Kurashiki Central Hospital is an urban hos-
phone interview as the standard, we calculated the test
pital that serves 800 000 people in the western area of Okayama
characteristics (sensitivity, specificity, predictive value and likeli-
prefecture, and there are approximately 70 000 visits to the hos-
hood ratios) of LNR with 95% CI using the normal approxima-
pital emergency department annually. At Kurashiki Central
tion. All statistical analyses were performed using SPSS software
Hospital, 10–15 patients with supraglottitis were hospitalised
V.19.0 (SPSS Inc, Chicago, Illinois, USA). The first author (TaF)
annually from 2000 to 2010, and about 20% were diagnosed
had full access to all study data and analyses.
without LNR. We set a 2 year study duration to enroll a suffi-
cient number of patients with disease.
RESULTS
From 16 April 2011 to 31 March 2013, 140 LNR were per-
Index and reference standard formed to rule out supraglottitis, and 105 LNR met the inclu-
The index test was the LNR. A trained radiological technologist sion and exclusion criteria of the study. Thirty-five patients
performed all LNR. One emergency physician (HO) and one (25%) were excluded because they did not give consent. No
radiologist (YO) independently interpreted each LNR. HO and patient had respiratory obstruction precipitated by LNR.
YO were blind to the patient’s history, symptoms and laryngo- Nasopharyngeal laryngoscopy was performed in 71 (67.6%)
scopic results. Disagreement was resolved by discussion, and the patients, 34 (32.4%) patients were followed-up by telephone
presence of a ‘thumb sign’ and ‘vallecula sign’ were recorded. interview. (figure 2). The characteristics and symptoms of the
Interobserver differences between HO and YO were analysed 105 enrolled patients are shown in table 1.
using k statistics, including 95% CI. Twenty-nine patients had positive LNR. Interobserver agree-
The reference standard was the findings of nasopharyngeal ment for the thumb sign was substantial, at 92.1% (12.1%
laryngoscopy or follow-up telephone interview after 7 days. agreement on positive ratings and 80.0% agreement on negative
The laryngoscopies were performed in the emergency depart- ratings), with a κ value of 0.71 (95% CI 0.55 to 0.87).
ment or otolaryngology department by otolaryngologists. Two Interobserver agreement for the vallecula sign was substantial, at

Figure 1 Katori’s scope grading of supraglottitis. (A) Grade 1: the patient’s epiglottis is slightly swelled and the entire length of the vocal folds
can be seen with the scope. (B) Grade 2: the patient’s epiglottis is moderately swelled and more than half of the posterior vocal folds can be seen
with the scope. (C) Grade 3: the patient’s epiglottis is severely swelled and only less than half of the posterior vocal folds can be seen with the
scope.
Fujiwara T, et al. Emerg Med J 2015;32:348–352. doi:10.1136/emermed-2013-203340 349
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Original article

Figure 2 Details of the patients who underwent lateral neck radiographs.

92.9% agreement (13.6% agreement on positive ratings and department physicians in four cases before the subsequent laryn-
79.3% agreement on negative ratings), with a κ value of 0.75 goscopy, while six patients who did not undergo laryngoscopy
(95% CI 0.60 to 0.90). Of the 29 patients with an abnormal were treated with antibiotics.
radiograph, 26 underwent LNR and 3 were discharged by the A radiographic abnormality was seen in 57.1% of the radio-
emergency physician. Of the 76 patients with a normal radio- graphs with grade 1 supraglottitis. Four patients (19%) with
graph, 45 underwent LNR and the rest were discharged. Among supraglottitis had false negative LNR; 3 of these were grade 1.
patients who had normal LNR and were discharged with phone All patients with grade 3 supraglottitis, which is an indication
follow-up, 71.0% had a sore throat and 41.9% had odynopha- for artificial airway management,15 had an abnormality on x-ray
gia, while among those who had a normal x-ray and laryngos- (positive thumb or vallecula sign).; 87.5% (7/8) of grade 2
copy, 100% had a sore throat and 68.9% had odynophagia. In supraglottitis patients had detectable radiographic abnormalities.
sum, 71 patients underwent laryngoscopy, and supraglottitis was (table 2). In all patients who were followed-up by telephone
diagnosed in 21 (grade 1=7, grade 2=8, grade 3=6) (table 2), interview, symptoms had resolved.
for a total incidence of 20% (figure 2). Supraglottitis was diag- The test characteristics of the LNR using the final interpret-
nosed in 17 of the 26 (65%) patients who had abnormal x-rays, ation agreed on by both the emergency physician and radiologist
and four of the patients (13%) who had normal xrays. No are displayed in Table 3. The LR positive of the LNR was 5.67
patients who were discharged had supraglottitis. (95% CI 3.27, 9.82) and the LR negative was 0.22 (95% CI
Half of the laryngoscopies were performed within 1 h of the 0.10, 0.51). The sensitivity of the emergency physician's inter-
LNR. The median time interval from LNR to laryngoscopy was pretation was slightly higher than the radiologist's 81% (62.2,
1 h (mean±SD, 3.6±5.2). Laryngoscopies were delayed in 97.8) vs. 66.7% (95% CI 46.5, 86.8), but specificity was similar
patients who had normal radiographs and were less likely to (84.5% vs. 85.7%).
have supraglottitis and in those who underwent radiographs on Six of the patients with negative LNR who did not undergo
the weekend. All of the laryngoscopies were performed within larygnoscopy were treated with antibiotics. The diagnoses of
24 h of the LNR. Antibiotics were prescribed by emergency these patients were acute pharyngitis (n=4), tonsillitis (n=1)
and intestinal perforation (n=1). We could not be certain if
these six patients had supraglottitis. If we re-analysed the results
Table 1 Demographics and clinical symptoms of enrolled patients without these six patients, specificity decreased from 85.7% to
84.6 (76.6 to 92.6)%.
Enrolled Laryngoscope Telephone
patients follow-up interview
(n=105) (n=71) follow-up (n=34) DISCUSSION
In this prospective study, LNR had relatively low sensitivity and
Age (years) 42.8±18.1 43.8±17.6 40.7±19.1
specificity for supraglottitis in adults. Previous studies reported
Males (n (%)) 56 (53.3) 43 (60.6) 13 (38.2)
a sensitivity and specificity of the classic thumb sign of 83–
Symptoms (n (%))
100% and 87–100%, respectively.6 8 In 1997, Ducic et al7
Sore throat 95 (90.5) 70 (98.6) 25 (73.5)
reported a new radiographic sign, vallecula sign, which had
Odynophagia 69 (65.7) 53 (74.6) 16 (47.1)
98.2% sensitivity and 99.5% specificity in their report. Based
Dyspnoea 13 (12.4) 5 (7.0) 8 (23.5)
on these findings, some authors suggested that the LNR might
Voice change 8 (7.6) 8 (11.3) 0 (0.0)
obviate the use of routine laryngoscopy. However, these retro-
Fever (37.5°C) 40 (38.1) 26 (36.6) 14 (41.2)
spective studies were at high risk of overestimating the diagnos-
Enrolled patients (n=105) included patients followed by laryngoscopy (n=71) and tic value, and no validation study of the vallecula sign has been
those followed by telephone (n=34).
reported.
350 Fujiwara T, et al. Emerg Med J 2015;32:348–352. doi:10.1136/emermed-2013-203340
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Original article

emergency department population. As previous studies calculat-


Table 2 Results and outcomes of lateral neck radiographs
ing the diagnostic accuracy were limited by spectrum bias,6 7 the
Supraglottitis true sensitivity and specificity were not clear. The incidence of
Grade 1 Grade 2 Grade 3 No supraglottitis Total supraglottitis in this study was similar to that in previous
reports,12 13 so our results can be generalised to other clinical
Radiographic abnormality settings. Kurashiki Central Hospital serves 800 000 people in
Positive 4 7 6 12 29 the western area of Okayama prefecture, where few hospitals
Negative 3 1 0 72 76 have more than one otolaryngologist to manage supraglottitis.
Thumb sign Thus a patient with suspected supraglottitis at other hospitals in
Positive 2 7 5 5 19 the western Okayama prefecture would be transferred to
Negative 5 1 1 79 86 Kurashiki Central Hospital. In our study, the incidence of supra-
Vallecula sign glottitis was 20.0%. The pre-test probability might change
Positive 4 7 4 10 25 according to the clinical setting and the physician’s decision.
Negative 3 1 2 74 80 The negative/positive predictive value of this study will be
Total 7 8 6 84 105 affected by the incidence in the population. This study included
Radiographic abnormality—positive means thumb sign positive or vallecula sign only adults, and it might not be possible to generalise our
positive. results to paediatric supraglottitis which may have a more
serious course.
The study had some limitations. Firstly, not all patients under-
The LNR is often used to rule out a diagnosis of supraglotti- went laryngoscopy to verify the diagnosis. It is difficult to have
tis; hence a low negative likelihood ratio and high negative pre- an otolaryngologist available at all times to perform laryngos-
dictive value are needed. In this study, the negative likelihood copy because they tend to have other commitments. If a patient
ratio was 0.22, which generates small changes in probability, is suspected of having supraglottitis, because of the risk of
and the negative predictive value was 94.7%, which indicated airway obstruction, laryngoscopy takes priority over the otolar-
that 1 in 20 patients would be missed by LNR. As ruling out yngologist’s other work, while in patients who are suspected of
supraglottitis based on negative LNR findings might result in having the common cold, laryngoscopy is not prioritised. It is
misdiagnosis, for patients with probable supraglottitis, nasopha- improper to make the latter patients wait for laryngoscopy and
ryngeal laryngoscopy should be considered to make a definite also ethically improper to subject a patient suspected of having
diagnosis. However, compared with paediatric supraglottitis, a common cold to unnecessary invasive laryngoscopy.
adult supraglottitis is less severe, with a lower risk of airway Therefore, we defined the reference standard as laryngoscopy or
obstruction, especially for grade 1 supraglottitis. Thus, if the telephone follow-up. In this study, only 57% (4/7) of grade 1
role of LNR is to detect only severe supraglottitis, then perhaps supraglottitis patients had a radiological abnormality, and 8.9%
these x-rays have sufficient diagnostic accuracy. (4/45) of patients with normal radiographs and laryngoscopy
In this study, one emergency physician and one radiologist had supraglottitis. Patients who had normal radiographs without
interpreted the LNR independently, and we evaluated interob- laryngoscopy were followed-up by telephone interview because
server disagreement. The thumb and vallecula signs were in the emergency department physician estimated that they had a
agreement about 92% of the time, and the kappa value sug- low probability of supraglottitis. It is possible that a maximum
gested moderate interobserver agreement. The radiologist is an of 8.9% of patients followed-up by telephone had supraglottitis.
expert in the interpretation of radiographs, including LNR. 35 patients were excluded because they did not consent to
However, in Japan, emergency physicians almost always inter- participate in the study. Most of the excluded patients had
pret LNR. This is because of the increased need for interpret- normal radiographs and a lower rate of odynophagia (65.7% in
ation of CT, MRI and positron emission tomography images by the enrolled patients vs 42.9% in the excluded patients), and
radiologists, whose time might otherwise be spent interpreting were younger (42.8±18.1 years for the enrolled patients vs 36.1
LNR. The sensitivity of the LNR read only by an emergency ±16.5 for the excluded patients). Patients at low risk of epiglot-
physician was higher than the radiologist’s, but specificity was titis were excluded from the study, which might decrease the
similar. validity of the study. In all of the patients who were followed-up
by telephone interview, symptoms had resolved, and none was
Strengths and limitations diagnosed with supraglottitis. Therefore, it is unlikely there
To our knowledge, this is the first prospective validation study were many missed patients with supraglottitis and hence missed
of LNR for detection of adult supraglottitis in a general cases should have had little, if any, effect on our results.

Table 3 Lateral neck radiograph test characteristics


Radiographic abnormality
(thumb or vallecula sign) Thumb sign Vallecula sign

Sensitivity (%) 81.0 (64.2 to 97.8) 66.7 (46.5 to 86.8) 71.4 (52.1 to 90.8)
Specificity (%) 85.7 (78.2 to 93.2) 94.0 (89.0 to 99.1) 88.1 (81.2 to 95.0)
Positive predictive value (%) 58.6 (40.7 to 76.5) 73.7 (53.9 to 93.5) 60.0 (40.8 to 79.2)
Negative predictive value (%) 94.7 (89.7 to 99.8) 91.9 (86.1 to 97.6) 92.5 (86.7 to 98.3)
Positive likelihood ratio 5.67 (3.27 to 9.82) 11.2 (4.74 to 26.5) 6.00 (3.21 to 11.20)
Negative likelihood ratio 0.22 (0.10 to 0.51) 0.35 (0.20 to 0.63) 0.32 (0.17 to 0.62)
Values are median (95% CIs).

Fujiwara T, et al. Emerg Med J 2015;32:348–352. doi:10.1136/emermed-2013-203340 351


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Original article

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352 Fujiwara T, et al. Emerg Med J 2015;32:348–352. doi:10.1136/emermed-2013-203340


Downloaded from http://emj.bmj.com/ on November 17, 2015 - Published by group.bmj.com

Diagnostic accuracy of lateral neck


radiography in ruling out supraglottitis: a
prospective observational study
Takashi Fujiwara, Hiroshi Okamoto, Yasuhiro Ohnishi, Toshio Fukuoka
and Kazuyuki Ichimaru

Emerg Med J 2015 32: 348-352 originally published online August 20,
2014
doi: 10.1136/emermed-2013-203340

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