Bilateral Piriform sinus fistulas: A case study and review of management options

Abstract

Background:
Piriform sinus fistulas occur due to developmental abnormalities of the third and fourth branchial arches, and almost always occur unilaterally. They generally present as recurrent abscesses in the anterior-inferior neck, with concurrent thyroiditis. They have conventionally been managed with complete removal of the sinus tract, and thyroidectomy if required; however, endoscopic approaches have been increasingly favored. Herein we describe a case of bilateral piriform sinus fistulas, and present a review of the literature concerning their endoscopic management.

Case presentation:
Our patient was determined to have bilateral piriform sinus fistulas based on computer tomography, magnetic resonance imaging and microlaryngoscopy. We performed electrocauterization of the proximal fistula tracts, followed by injection of fibrin sealent. Our patient has not had a recurrence in the ten months since his procedure. There were no complications. Twenty-three articles describing an endoscopic approach to these fistulas were identified through PubMed, and a search through the references of related articles was completed.

Conclusion:
Of one hundred and ninety-five patient cases we reviewed, an endoscopic procedure success rate of 82% and complication rate of 5.6% was determined. Piriform sinus fistulas that occur bilaterally are a rare congenital abnormality of the neck. Endoscopic approaches are an acceptable alternative option to open procedures, with similar success and a lower rate of complications.

Full text

C A S E R E P O R T Open Access
Bilateral Piriform sinus fistulas: a case study
and review of management options
Deanna Lammers
1
, Ross Campbell
2
, Jorge Davila
2
and Johnna MacCormick
2*
Abstract
Background: Piriform sinus fistulas occur due to developmental abnormalities of the third and fourth branchial
arches, and almost always occur unilaterally. They generally present as recurrent abscesses in the anterior-inferior
neck, with concurrent thyroiditis. They have conventionally been managed with complete removal of the sinus
tract, and thyroidectomy if required; however, endoscopic approaches have been increasingly favored. Herein we
describe a case of bilateral piriform sinus fistulas, and present a review of the literature concerning their
endoscopic management.
Case presentation: Our patient was determined to have bilateral piriform sinus fistulas based on computer
tomography, magnetic resonance imaging and microlaryngoscopy. We performed electrocauterization of the
proximal fistula tracts, followed by injection of fibrin sealent. Our patient has not had a recurrence in the
ten months since his procedure. There were no complications.
Twenty-three articles describing an endoscopic approach to these fistulas were identified through PubMed,
and a search through the references of related articles was completed.
Conclusion: Of one hundred and ninety-five patient cases we reviewed, an endoscopic procedure success
rate of 82% and complication rate of 5.6% was determined. Piriform sinus fistulas that occur bilaterally are
a rare congenital abnormality of the neck. Endoscopic approaches are an acceptable alternative option to
open procedures, with similar success and a lower rate of complications.
Keywords: Piriform sinus fistula, Fourth Branchial fistula, Third Branchial fistula, Branchial arch abnormality,
Suppurative Thyroiditis, Endoscopic repair
Background
Third and fourth branchial apparatus anomalies, commonly
referred to as piriform sinus fistulas (PSFs), are sinus tracts
and fistulas that develop from the piriform sinus. They
occur more commonly on the left side, and typically
present in childhood with recurrent acute suppurative
thyroiditis and neck abscess often following an upper
respiratory tract infection [1–3]. Infants and neonates may
have respiratory distress, stridor, dysphagia and feeding dif-
ficulties due to tracheal compression from the abscess
[3,4]. Thyroid function is usually normal [1].
PSFs are uncommon and can be difficult to diagnose,
due in part to their non-specific presentations [1].
Furthermore, bilateral PSFs are extremely rare, with a
thorough literature search revealing only four other
patients described with this condition [5–7]. Herein, we
present a case of a patient with bilateral piriform sinus
fistulas, and review their management.
Case presentation
Our patient initially presented at 10 months of age with
rapid development of a mass in the left neck that was ten-
der, firm and erythematous. It was associated with fever as
well as dysphagia, and decreased oral intake. The patient
had no significant past medical history, nor family history.
A lateral neck x-ray suggested a retropharyngeal infectious
process. After 48 h of antibiotic treatment, there was
minimal clinical improvement; a computer tomography
(CT) scan of the neck demonstrated a 5.4 × 3.5 × 4.2 cm
left neck abscess extending to left parapharyngeal and
retropharyngeal spaces (Fig. 1). Following imaging, the
patient underwent incision and drainage. The wound
* Correspondence: jmaccormick@cheo.on.ca
2
Children’s Hospital of Eastern Ontario, 401 Smyth Road, Ottawa, ON K1H
8L1, Canada
Full list of author information is available at the end of the article
© The Author(s). 2018 Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0
International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and
reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to
the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver
(http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.
Lammers et al. Journal of Otolaryngology - Head and Neck Surgery (2018) 47:16
DOI 10.1186/s40463-018-0258-y

culture grew Streptococcus viridans and Hemophilus
parainfluenzae.
For 6 months after the surgery, the wound persisted to
drain intermittently, culminating in a submandibular
cellulitis. A CT neck and wound culture at that time
were unremarkable, and the patient improved after 5
days of IV antibiotics.
At 8 years of age, the patient again presented with a 4 day
history of sore throat, fever, drooling, and neck stiffness and
swelling; IV antibiotic therapy was initiated. A CT neck
confirmed a bilobed right-sided retropharyngeal abscess
measuring 2.3 × 2.9 × 3.3 cm and 2.0 × 2.1 × 2.8 cm in the
superior and anterior lobes, respectively. The thyroid gland
was noted to be intimately involved with this inflammatory
process (Fig. 2). The patient was brought to the operating
room for incision and drainage of the abscess. The wound
culture was positive for Streptococcus anginosis.
Shortly following the resolution of the infection, magnetic
resonance imaging (MRI) was performed identifying bilateral
communications between the piriform sinuses and thyroid
lobes (Fig. 3). Direct laryngoscopy confirmed the diagnosis
of bilateral piriform sinus fistula with the passage of 4 Fr
ureteric catheters though tracts originating in the piriform
sinuses, on both the left and right sides (Fig. 4).
After lengthy discussions and deliberation and review of
the relevant literature, the family opted for endoscopic
electrocauterization with fibrin sealant management (Fig. 5).
We favoured this approach as the PSFs were bilateral, and
therefore a total thyroidectomy may have been required for
definitive surgical management.
Cauterization and obliteration of the bilateral PSFs was
performed without complication. First, a flexible catheter
waspassedintothepiriformsinustoconfirmitslocation.
With the location confirmed, a Bugbee catheter was inserted
into the fistula and the edges were cauterized at a setting of
8 W. Once adequate cauterization was achieved, Tisseel was
injected into the fistula tract. This procedure was performed
in both the left and right PSFs. A follow-up 6 months with
direct laryngoscopy confirmed closure of both tracts. There
has been no recurrence 10 months following the procedure,
at the time of writing of this manuscript.
Fig. 1 Axial enhanced CT showing involvement of the thyroid gland
with surrounding multiloculated abscess
Fig. 2 Axial enhanced CT illustrating involvement of the abscess
with the right lobe of the thyroid gland
Fig. 3 Coronal MRI identifying bilateral tracts from the piriform sinus
to the thyroid gland
Lammers et al. Journal of Otolaryngology - Head and Neck Surgery (2018) 47:16 Page 2 of 6

Discussion and conclusions
A thorough literature search about the endoscopic
management for piriform sinus fistulas was performed.
To identify journal articles, a literature search using
PubMed was conducted using the filters English
language and years 1998-2017. The references in each
article were also reviewed to find additional papers.
Twenty-three articles were identified as relevant studies.
Table 1lists the year the study was published, number of
patients enrolled, patient characteristics (age, gender),
techniques used, success rate, amount of follow-up and
complication rate. A procedure was considered a success
if did not have to be repeated for either recurrence, or
incomplete fistula tract closure. For research groups that
had multiple publications using repeat patient cases,
only their last publication was included.
A PubMed search was also used to find cases of
bilateral piriform sinus fistulae. No filters were used.
Four cases were found from three publications.
From one hundred and ninety-five cases of piriform
sinus fistulas, based on 23 studies, we calculated an
endoscopic success rate average of 82% and complica-
tion rate of 5.6%. Post-operative follow-up times ranged
from 6 weeks to 18 years with an average of 35.5 months.
Patient ages ranged from newborns to 31 years, and
there was a gender distribution of 1.8 females to 1 male
(56 females, 31 males). However, the gender and ages of
the patients in the studies could not be identified for
55% of patient cases presented. All the studies were
published from 2003 to 2016.
PSFs are notoriously difficult to diagnose. While neck
abscesses are a common entity in pediatric otolaryngology
patients, PSFs are rare. With a repeated history of anterior
neck abscesses, or when imaging identifies thyroid
gland involvement, this diagnosis should be considered.
Confirmation is typically done with direct laryngoscopy,
which has a positive predictive value (PPV) of 90%, or
barium swallow (PPV 88%) [1,2]. Management of a
piriform sinus fistula involves antibiotics and incision
and drainage for acute infections, and observation,
followed by surgery or endoscopic techniques for long-
term management.
In the acute setting, the choice of antibiotic should reflect
typicaloralfloraaswellasStaphylococcus aureus [2].
Surgical drainage for source control is indicated where
there is abscess formation [1].
Considering long-term management, observation
may be considered, especially for asymptomatic PSFs;
however, 89-94% of patients will continue to have
recurrent infections [2,3]. Therefore, further treat-
ment is usually necessary.
Fig. 4 Left piriform sinus fistula seen in (a); cannulated in (b); Right piriform sinus fistula seen in (c); cannulated in (d)
Fig. 5 Right piriform sinus fistula following electrocauterization (left)
and fibrin sealant into the fistula tract (right)
Lammers et al. Journal of Otolaryngology - Head and Neck Surgery (2018) 47:16 Page 3 of 6

Surgical removal of the sinus tract, and the thyroid
gland if required, is typically curative, and has been
the standard of care for many years [1,9]. However,
excising the entire fistula tract can be very technically
challenging, especially if the patient has significant
scarring from repeated infections and prior surgical
drainages. Incomplete excision may lead to recurrence [1].
In addition, the recurrent laryngeal nerve is at risk for
injury leading to potential vocal cord paralysis. Salivary
fistulas, hemorrhage, wound infection, cicatrises, Horner
syndrome and injury to branches of the facial nerve are
other possible complications. Reported success rates range
from 85 to 100% [2–4,6] with a complication rate of
5-6% [2,3].
Table 1 Endoscopic repair patient demographics, techniques used, success rates, follow-up and complication rates
Author Year Number of Patients Patient Age Patient Gender Technique Success rate Duration of follow-up Complication rate
Shrime [29] 2003 1 1d F CC with silver nitrate 100% –100% (Transient
vocal cord palsy)
Cigliano [12] 2004 1 9 F FS repeated at short
term interval three times
100% 15 m 0%
Ahmed [14] 2008 3 3-9y 1 M
2F
Secondary EC following
failed surgical excision
100% 9-13 m 0%
Pereira [8] 2008 2 2-18y 2 M CC with silver nitrate 100% 2y 0%
Chen [13] 2009 9 3-16y 1 M
8F
EC +/−polyglactin sutures 78% 7 m-8y 0%
Miyauchi [30] 2009 12 14-31y 2 M
10F
CC with 30% TCA 83% 4-21 m 0%
Leboulanger [15] 2010 19 1d-18y –2EC
13 CO
2
Laser
4 Thulium laser
68% 6 m-5y 0%
Bajaj [16] 2011 3 <1y –EC 100% 6w 0%
Zhang [21] 2012 1 15y M CC 0% 5y 0%
Cha [9] 2013 44 –– 31 CC with 20-40% TCA
13 Secondary CC with
TCA following failed
surgical excision
77% 18 m-18y 0%
Park [20] 2013 2 13 m-5y 1 M
1F
CC with 30% TCA 100% 7-18 m 100% (Transient
vocal cord palsy)
Watson [17] 2013 5 2-12y 1 M
4F
1EC
2CO
2
Laser
2 CC with silver nitrate
100% 11-41 m 0%
Parida [19] 2014 3 11-12y 1 M
2F
2 CC with silver nitrate
1 Secondary CC with
silver nitrate following
failed surgical excision
100% 2-3y 0%
Sun [11] 2014 22 6 m-14y 7 M
15F
EC 91% 1 m-14y 0%
Wong [28] 2014 2 10-14y 1 M
1F
1EC
1 Secondary EC following
failed surgical excision
50% 4y 50% (mild hoarseness that
resolved within 2 weeks)
Hwang [24] 2015 13 1.5-15y 9 M
4F
CC with 20% TCA 54% 5.5y (median) 0%
Josephson [1] 2015 1 7y F CO2 laser with chromic
suture
100% 4y 0%
Kamide [18] 2015 1 20y F Electrocauterization 100% 1y 0%
Abbas [25] 2016 1 12y F Electrocauterization 100% 22 m 0%
Di Nardo [22] 2016 1 3y F Secondary Glubran 2
sealing following 4 failed
surgical excisions
100% 6y 0%
Huang [27] 2016 5 5-7y 3 M
2F
KTP laser assisted EC with FS 80% 7-36 m 0%
Matsuzaki [26] 2016 2 9-26y 1 M
1F
Endoscopic partial
resection with
polydioxanone suture
100% 1-2y 0%
Zhang [23] 2016 42 –– 11 EC
31 Coblation cauterization
88% 2-40 m 7% (temporary hoarseness)
EC electrocauterization, CC Chemocauterization, FS fibrin sealent, TCA trichloroacetic acid, Mmale, Ffemale, mmonth, yyears
Lammers et al. Journal of Otolaryngology - Head and Neck Surgery (2018) 47:16 Page 4 of 6

An alternative to surgery is endoscopic electrocautery
or chemocauterization, followed by fibrin sealing or
endoscopic suture ligation if necessary. This closes off
the proximal portion of the tract, preventing leakage of
pharyngeal contents into the sinus. Endoscopic tech-
niques have recently been shown to be safe and effective
[9,10], and can be performed as an outpatient
procedure, thereby reducing hospitalization and associ-
ated costs. The associated risks are lower compared to
an external technique, also with the advantage of lacking
a surgical incision and consequent scar [9,11]. However,
this is a newer procedure, and data regarding long-term
results is scarce. Recent studies suggest the success rate
is equivalent to, or slightly less than surgery, with
success rate average of 82% (Table 1)[1–3,8–29].
In summary, piriform sinus fistulas are uncommon
developmental anomalies in children. The presentation
of repeated anterior neck abscesses, particularly if the
thyroid gland is involved should prompt the clinician to
consider this entity, and trigger an appropriate work-up.
As the fistulas may occur bilaterally, a careful inspection
of both sides is required during laryngoscopy. Definitive
management has classically been performed through an
excision of the tract. Endoscopic electrocautery of the
tract may be an acceptable alternative, with comparable
success and a lower rate of complications.
Abbreviations
CT: Computer tomography; MRI: Magnetic resonance imaging; PPV: Positive
predictive value; PSF: Piriform sinus fistula
Acknowledgements
Not applicable.
Funding
This research did not receive any specific grant from funding agencies in the
public, commercial, or not-for-profit sectors.
Availability of data and materials
Data sharing is not applicable to this article as no datasets were generated
or analysed during the current study.
Authors’contributions
JM and RC performed patient diagnosis, investigations and treatments. DL
completed the literature review. All three analyzed and interpreted patient
data, and were major contributors in writing the manuscript. JD aided in
imaging selection for the manuscript. All authors read and approved the
final manuscript.
Ethics approval and consent to participate
Informed consent was obtained from the patient. The procedures were in
accordance with the ethical standards of the Children’s Hospital of Eastern
Ontario.
Consent for publication
Informed consent was obtained from the patient.
Competing interests
The authors have no potential competing interest with respect to the
research, authorship, and/or publication of this article.
Publisher’sNote
Springer Nature remains neutral with regard to jurisdictional claims in
published maps and institutional affiliations.
Author details
1
University of Ottawa Faculty of Medicine, Roger Guindon Hall, 451 Smyth
Rd., Ottawa, ON K1H 8M5, Canada.
2
Children’s Hospital of Eastern Ontario,
401 Smyth Road, Ottawa, ON K1H 8L1, Canada.
Received: 6 September 2017 Accepted: 29 January 2018
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