Journal of Otology & Rhinology

Davidi et al., J Otol Rhinol 2015, 4:2
http://dx.doi.org/10.4172/2324-8785.1000218
Case Report
A SCITECHNOL JOURNAL
4th Branchial Cleft Cyst Anomaly:
Case Presentation
Erez S Davidi1,2, Meir Warman1,2*, Yonatan Lahav1,2, Doron
Schindel1,2 and Doron Halperin1,2
1Department
Journal of Otology &
Rhinology
resected in conjunction with the central part of the hyoid bone as
instructed in Sistrunk operation, nonetheless a continuous infective
tract was seen leading to the left thyroid gland which was felt stiff to
palpation. The Intra-operative decision was not to extend the surgery
further to the thyroid gland to avoid any injury to recurrent laryngeal
nerve as well as lack of parent’s consent.
of Otolaryngology Head & Neck Surgery, Kaplan Medical Center,
Rehovot
2Hebrew
University-Hadassah Medical School, Jerusalem, Israel
*Corresponding
author: Meir Warman, Department of Otolaryngology, Head and
Neck surgery, Kaplan Medical Center, Pasternak St., P.O.B 1, Rehovot 76100,
Israel, E-mail: MeirWa@clalit.org.il
Rec date: Sep 10, 2014 Acc date: Dec 18, 2014 Pub date: Mar 13, 2015
Abstract
The human branchial apparatus is composed of six paired
mesodermal arches, separated by endodermal and ectodermal
invaginations known as pouches and clefts, respectively.
Error in obliteration of a pouch or groove may create a sinus,
fistula or a cyst.
Approximately 95% of congenital anomalies of the branchial
apparatus involve the second arch, pouch or cleft, while the
remaining mostly arise from the first and third arches.
Fourth branchial involvement is extremely rare with only few
cases published in the English literature.
In this report we will emphasize the working dilemma in
establishing the correct diagnosis of fourth branchial cleft cyst.
Diagnostic tools and appropriate treatment will be addressed
as well.
Figure 1: Magnetic Resonance Imaging (MRI): Axial T1 sequence
with Gadolinium enhancement demonstrating hyper intense
process involving the central neck (white arrow) which cannot be
separated from the left lobe of the thyroid gland (black asterisk).
Keywords: Branchial cleft; Cyst; Fourth branchial cleft cyst;
Hemithyroidectomy
Case Presentation
A two and a half year old female child presented with fever, pain
and swelling of the left lower neck of one week duration. Her past
medical history is significant only for Asthma which is occasionally
treated by bronchodilators.
Physical examination revealed a 2×1.5 cm, tender mass involving
the mid neck and thyroid region with overlying skin inflammation.
The swelling subsided under intravenous treatment with Amoxicillin
Clavulanate for two weeks, however recurred once the child was
discharged. Thyroid function tests were non disturbed. Ultrasound
revealed a cystic mass in the central neck with left lateral extension.
The left thyroid gland was diffusely enlarged.
Differential diagnosis of infected thyroglossal duct cyst (TGDC),
thyroiditis, and thyroid abscess were considered. Ruling out thyroiditis
as the probable cause due to normal thyroid function tests, an infected
TGDC was the reasonable diagnosis and Sistrunk operation was
advised. During surgery a midline infected soft tissue mass was
Figure 2: Hematoxylin and eosin X4: Demonstrate fibrous capsule
(thick arrow) covering large lymphatic aggregates (arrowhead)
separated from normal looking thyroid follicles (thin arrow).
The post operative period was uneventful and the child was
discharged.
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Citation:
Davidi ES, Warman M, Lahav Y, Schindel D, Halperin D (2015) 4th Branchial Cleft Cyst Anomaly: Case Presentation. J Otol Rhinol 4:2.
doi:http://dx.doi.org/10.4172/2324-8785.1000218
Pathologic examination did not find cystic component inside the
infected tissue that could support the diagnosis of TGDC. The clinical
course of the child did not resolved and was suggestive of a persistent
left cervical inflammatory process.
Magnetic Resonance Imaging demonstrated (Figure 1) persistent
inflammatory process in the left side of the neck, approximate to the
trachea. It was unclear whether it involves the thyroid gland or not.
The diagnosis of 4th branchial cleft cyst was then suspected and the
child underwent left hemi-thyroidectomy a month later that was
uneventful. The final pathologic specimen (Figure 2) revealed cystic
structure next to normal looking thyroid follicles.
Diagnostic means with radio opaque swallowing test, contrast
enhanced CT or MRI should be followed to assert the diagnosis before
unilateral hemithyroidectomy is being considered.
References
1.
2.
One year after the thyroidectomy the patient is well with no signs of
inflammation or infection.
3.
Discussion
4.
Fourth branchial cleft anomaly is a rare entity, reported to be only
1-4% of all branchial cleft anomalies [1-3]. Third and Fourth pouches
are intimately related and are considered to be close in the differential
diagnosis, both originate from the pyriform sinus, the third from the
base and the fourth from the apex [4]. They share a similar anatomic
course. While the third ascends along the carotid sheath, over the
superior laryngeal nerve and piercing the thyrohyoid membrane [5-6],
the fourth passes under the superior laryngeal nerve and over the
recurrent laryngeal nerve and hypoglossal nerve. In the thorax it
travels around the aortic arch on the left and around the subclavian
artery on the right, entering the larynx at the cricothyroid joint [7].
Clinically, fourth branchial cleft anomaly presents as a recurrent
deep neck infection, abscesses or a draining pit [8].
Both third and fourth anomalies can cause recurrent thyroidits,
usually on the left side. Considering the large vascular and lymphatic
supply of the thyroid gland and its relative resistance to infection,
recurrent thyroiditis or thyroid abscess are an unusual and must raise
a suspicion to branchial anomaly [9].
The diagnosis of fourth branchial anomaly is challenging. In our
patient, Ultrasonography and Magnetic Resonance Imaging revealed
local inflammatory process with no clear involvement of thyroid gland
or any tract of infection accordance with previous publications
reporting this entity.
Other options for demonstrating the fistula course include X-ray
and Computed Tomography (CT) guided fistulography or by a more
direct mean of laryngo-pharyngoscopy.
The classic and definite treatment is complete surgical excision by
unilateral hemithyroidectomy with an 8% chance of recurrence
compared with 89% recurrence rate when only simple drainage is
being used [10-12]. Complications, especially recurrent laryngeal
nerve paralysis are higher in younger patients (less than 8 years of age).
Endoscopic procedures report successful outcome in obliterating
the fistulous opening at the piriform sinus using laser, electro
coagulation or chemical means [13-19].
Conclusion
Fourth branchial cleft anomaly must be suspected in any case with
recurrent left sided thyroiditis or lateral neck abscess.
Volume 4 • Issue 2 • 1000218
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Citation:
Davidi ES, Warman M, Lahav Y, Schindel D, Halperin D (2015) 4th Branchial Cleft Cyst Anomaly: Case Presentation. J Otol Rhinol 4:2.
doi:http://dx.doi.org/10.4172/2324-8785.1000218
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