Spontaneous closure of branchial sinus of the pyriform fossa

Abstract

Management of third and fourth branchial cleft anomalies are similar. These anomalies should be suspected in a child with recurrent low-anterior neck abscess. Investigations in the form of cross-sectional studies and examination of the pharynx under anaesthesia will facilitate diagnosis and resolution of abscess. Spontaneous closure of the pyriform sinus can occur following conservative management with antibiotic treatment and abscess drainage. This emphasise the role of second-look prior to implementing endoscopic cauterisation or surgery.

Full text

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LiuZ, etal. BMJ Case Rep 2018. doi:10.1136/bcr-2017-223691
CASE REPORT
Spontaneous closure of branchial sinus of the
pyriformfossa
Zhaobo Liu,1 Mohd Afiq Mohd Slim,2 Conor Jackson,1 Keith Trimble1
Findings that shed new light on the possible pathogenesis of a disease or an adverse effect
To cite: LiuZ,
Mohd SlimMA, JacksonC,
etal. BMJ Case Rep
Published Online First:
[please include Day Month
Year]. doi:10.1136/bcr-2017-
223691
1Royal Belfast Hospital for Sick
Children, Belfast, UK
2NHS Greater Glasgow and
Clyde, Glasgow, Glasgow, UK
Correspondence to
Dr Zhaobo Liu,
paulliuzb@ doctors. org. uk
Accepted 3 April 2018
SUMMARY
Management of third and fourth branchial cleft anomalies
are similar. These anomalies should be suspected in a child
with recurrent low-anterior neck abscess. Investigations
in the form of cross-sectional studies and examination of
the pharynx under anaesthesia will facilitate diagnosis and
resolution of abscess. Spontaneous closure of the pyriform
sinus can occur following conservative management with
antibiotic treatment and abscess drainage. This emphasise
the role of second-look prior to implementing endoscopic
cauterisation or surgery.
BACKGROUND
Branchial cleft anomalies are an important differential
diagnosis for paediatric recurrent anterior and lateral
neck abscesses. They account for approximate 17%
of all paediatric neck masses.1 Third or fourth bran-
chial cleft anomalies are a rare occurrence.2 3 They are
associated with the classical finding of pyriform sinus
fistula, which can be appreciated from cross-sectional
studies or by direct visualisation of the opening at pyri-
form sinus during pharyngoscopy.4 5 Image modalities
used to diagnose this condition include ultrasonog-
raphy, CT with or without fistulography and MRI.2 6
Third and Fourth cleft anomalies can only be differ-
entiated definitively from each other by their relation-
ship with the Superior Laryngeal Nerve (SLN) during
neck dissection.5 However, their management remains
similar.2
Currently, there is limited evidence reporting spon-
taneous closure of these anomalies.7 8 The physiology
and timeframe of closure are also not well under-
stood. Here, we describe a third or fourth branchial
cleft anomaly, which was diagnosed based on the CT
(figure 1), pharyngoscopy (figure 2) and clinical find-
ings which underwent spontaneous resolution from a
16-month-old girl who presented with low-anterior
neck mass and restricted neck movement.
CASE PRESENTATION
A 16-month-old girl presented to the Accident and
Emergency department when her parents noticed
she has a restricted neck movement and reduced
oral intake, following a 1 week history of upper
respiratory tract infection. There was no associated
swallowing difficulty, choking episodes or noisy
breathing. The baby had no other significant past
medical history and had achieved all her develop-
mental milestones.
On examination, she had a firm indurated
low-anterior neck mass left to the midline, in the
region of her thyroid gland with no signs of cellu-
litis or external fistula opening. She was admitted
and treated with intravenous co-amoxiclav.
INVESTIGATIONS
Ultrasound of her neck showed a cystic collec-
tion anterior to her thyroid gland. Contrast-en-
hanced CT Neck showed a fluid collection with
surrounding soft tissue enhancement anterior to
the left thyroid gland lobe, consistent with abscess
formation (figure 1). No other abnormal contrast
enhancement of the soft tissue was noted at the
level of the thoracic inlet. Thus further thoracic
window assessment was not performed.
DIFFERENTIAL DIAGNOSIS
The main differential diagnoses for this patient
include third or fourth branchial sinus or bran-
chial cleft fistulae. It was unlikely for this case to
be a branchial cleft fistula as a fistula tract was not
visible on cross sectional imaging and there was an
absence of an external fistula opening on examina-
tion. Only a branchial sinus opening was visualised
on pharyngoscopy.
Both third and fourth branchial cleft anoma-
lies can present as pyriform sinus fistulae, more
commonly on the left side.2 5 8 While third branchial
cleft fistula tends to originate from the upper lateral
wall of the pyriform sinus, the fourth branchial cleft
fistula tends to originate from the caudal end of the
pyriform sinus.5 6 They can also be distinguished by
the location of their fistula tract. The third bran-
chial cleft fistula runs superficial to both superior
and recurrent laryngeal nerves while the fourth
branchial cleft fistula runs deep to the superior but
superficial to the recurrent laryngeal nerve.6 9 Both
can present with recurrent neck abscesses.8 While
cysts from the third branchial cleft frequently arise
posterior to the sternocleidomastoid muscle in
the posterior triangle, the fourth branchial cleft
anomaly tends to present with left-sided neck
abscess and acute suppurative thyroiditis, as the
track inclines to end at the upper part of the left
thyroid.5 8 In this case, it was difficult to distinguish
between third and fourth branchial sinus based on
clinical findings. However, this does not have clin-
ical significance, as the management of both third
and fourth branchial anomalies is the same.
TREATMENT
Subsequently, the patient underwent inci-
sion and drainage of the neck abscess with

2LiuZ, etal. BMJ Case Rep 2018. doi:10.1136/bcr-2017-223691
Findings that shed new light on the possible pathogenesis of a disease or an adverse effect
pharyngolaryngoscopy under general anaesthesia. During visual-
isation of the pharynx, an opening was noted at the left pyriform
sinus (figure 2). A third or fourth branchial cleft anomaly was
diagnosed based on the physical examination, pharyngolaryn-
goscopy and imaging characteristics.
OUTCOME AND FOLLOW-UP
The patient was discharged on a weeklong course of oral
co-amoxiclav and was re-admitted for planned endoscopic
cautery of pyriform sinus 4 weeks later. Before the surgery, the
risks and benefits of endoscopic cauterisation and open-neck
surgery including left hemithyroidectomy were discussed with
the parents. On the day of the definitive procedure, the pyri-
form sinus was visualised again, but the opening at the pyriform
sinus had scarred and closed over (figure 3). Thus no further
interventions were required. The patient also had her 6 month
follow-up. During this period, she did not have any readmis-
sions, signs or symptoms that suggested the re-accumulation
of her neck abscess. She was subsequently discharged from the
specialty follow-up clinic.
DISCUSSION
Third and fourth branchial cleft anomalies occur following an
incomplete involution of the branchial apparatus.1 This can
result in the formation of cyst, sinus or fistula.1 Traditional treat-
ments for congenital third or fourth branchial cleft anomalies
are similar. These include treating the acute infective phase with
Figure 1 CT Neck with contrast.
Figure 2 Initial pharyngolaryngoscopy view of the left pyriform
sinus.*Red Circle highlighting the opening of branchial sinus.
Figure 3 Follow-up pharyngolaryngoscopy view of the spontaneously
closed left pyriform sinus probed with a 5-Ch ureteric catheter.

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LiuZ, etal. BMJ Case Rep 2018. doi:10.1136/bcr-2017-223691
Findings that shed new light on the possible pathogenesis of a disease or an adverse effect
antibiotics and incision and drainage of the abscess, followed
by open-neck surgery involving removal of the track or endo-
scopic cauterisation.2 One systematic review on the management
of third branchial cleft anomalies demonstrated that failure
rate for endoscopic cauterization is comparable to traditional
open-neck surgery (15% and 18% respectively; median age of
diagnosis: 10-year-old).8 However, A case series of 20 patients
with fourth branchial cleft anomalies (median age of diagnosis:
4-year-old) noted that there is a higher rate of failure of endo-
scopic treatment in neonatal presentations as these lesions are
naturally secreting and re-opening of the tract can occur.2 They
recommended open-neck surgery for neonates with large cyst
or early recurrence following incision of the cyst, while endo-
scopic approach was recommended for older children due to
their lower relapse rate.2 It is the author’s opinion that endo-
scopic cauterisation should be first line in managing branchial
cleft anomalies for older children if sinus were to persist during
second-look as was our practice in this case study.
Spontaneous resolution of pyriform sinus fistula has previ-
ously been reported in children aged between 3 to 11.5 years
old.7 A systematic review of third branchial cleft anomalies
reported 10 cases of resolution following conservative treatment
across all age groups with the majority of these cases belonging
to those aged four and above.8 This case report provide addi-
tional evidence that spontaneous resolution of branchial cleft
sinus occurs in an infant of 16 months’ old.
It is possible that cases of spontaneous resolution might
be higher than the number reported in the literature as bran-
chial cleft anomalies could often be misdiagnosed in children
presenting with a single or recurrent neck abscesses. Therefore,
we advocate high vigilance for branchial cleft anomalies in
children presenting with recurrent low-neck abscesses, especially
for those anterior to the thyroid gland or presenting with suppu-
rative thyroiditis. Cross-sectional studies and examination under
anaesthesia should be performed in this group of patients to
clinch the diagnosis. Acute phase management will involve anti-
biotic treatment followed by incision and drainage of the abscess
as incision and drainage as a single treatment modality alone is
associated with high recurrence rate.8 We also propose that a
planned second-look should be considered in all cases. Endo-
scopic cauterization should only be considered if sinus were to
persist at that stage. Out-patient follow-up should be offered for
monitoring if the definitive intervention is reserved during the
second look, at least in 6 months’ time.
Acknowledgements The authors would like to thank Dr Nicholas Clarke,
paediatric radiologist, for assisting with obtaining the images for this case report.
Contributors ZL: Manuscript preparation and editing. MAMS: Manuscript
preparation and editing. CJ: Manuscript editing and operating surgeon. KT:
Manuscript editing, operating surgeon and supervisor.
Funding The authors have not declared a specific grant for this research from any
funding agency in the public, commercial or not-for-profit sectors.
Competing interests None declared.
Patient consent Next of kin consent obtained.
Provenance and peer review Not commissioned; externally peer reviewed.
© BMJ Publishing Group Ltd (unless otherwise stated in the text of the article)
2018. All rights reserved. No commercial use is permitted unless otherwise expressly
granted.
REFERENCES
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3 Ford GR, Balakrishnan A, Evans JN, et al. Branchial cleft and pouch anomalies. J
Laryngol Otol 1992;106:137–43.
4 Kubota M, Suita S, Kamimura T, et al. Surgical strategy for the treatment of pyriform
sinus fistula. J Pediatr Surg 1997;32:34–7.
5 Adams A, Mankad K, Offiah C, et al. Branchial cleft anomalies: a pictorial review
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treated by endoscopic Glubran 2 sealing: A rare case and literature review. SAGE Open
Med Case Rep 2016;4:2050313X1667215.
7 Wasniewska M, Vigone MC, Cappa M, et al. Acute suppurative thyroiditis in childhood:
spontaneous closure of sinus pyriform fistula may occur even very early. J Pediatr
Endocrinol Metab 2007;20:75–7.
8 Nicoucar K, Giger R, Pope HG, et al. Management of congenital fourth branchial arch
anomalies: a review and analysis of published cases. J Pediatr Surg 2009;44:1432–9.
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Learning points
►High index of suspicion for branchial cleft anomalies in
children presenting with recurrent abscess of low anterior
thyroid region.
►Diagnosis of branchial cleft anomalies should be made with
cross-sectional imaging and pharyngoscopy under general
anaesthesia.
►Antibiotics and abscess drainage should precede definitive
management third/fourth branchial cleft anomalies.
►A planned second-look with and without endoscopic
cauterization should be considered in older children
prior to definitive management of branchial cleft
anomalies.
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