Papillary Carcinoma in Branchial Cleft Cyst

ABSTRACT

Start with Branchial cyst I guess. Occurrence of Papillary carcinomas in a thyroid gland
is common and in Branchial cyst without a primary in a thyroid gland is rare. Here we
report a case of a 38-years old thyroid male with a papillary carcinoma thyroid
originating from left Branchial cleft cyst without any traces of tumor in a thyroid gland.

INTRODUCTION

Branchial cleft anomalies are a developmental disorder of the neck and present as
lateral neck masses. About 95% of the Branchial cleft anomalies get their origin from
the second Branchial arch [?]. The etiology of Branchial cysts is still unclear. However,
several theories have been proposed for the understanding of their origin. Earlier
theories give us the understanding of Branchial cysts being the congenital malformation,
whereas recent theories propose an “inclusion theory” [?]. “Inclusion theory “gives us
the idea for BCC formation i.e. epithelium from upper aero digestive tract or glandular
tissue enters a cervical lymph node via lymphatic’s and stimulates degeneration into a
lateral cervical cyst [?]. In our case, there was no evidence of ectopic thyroid in BCC
unless the histopathology and immunohistochemistry staining revealed the occurrence
of papillary carcinoma of thyroid.

Squamous cell carcinoma of Branchial cleft cyst is rare but papillary carcinoma arising
from them is extremely rare. Here we report a papillary thyroid carcinoma incidentally
found in a patient with left brachial cleft cyst without any evidence of papillary carcinoma
in the thyroid gland.
CASE PRESENTATION

A 38-year-old male presented with a 03 years history of a painless left sided neck
swelling for three years. without any cervical lymphadenopathy. The swelling was mildly
progressive without any associated symptoms i.e. fever, respiratory distress,
tenderness or painful neck movements. Physical examination revealed about a 6cm six-
centimeters mass in his left neck without any punctum or a cutaneous fistula. The skin
overlying the mass was not erythematous or having any overlying changes. The mass
firm and non-reducible and it was mobile in a vertical plane. There was no associated
cervical (or axillary?) lymphadenopathy.

To evaluate an ultrasound (USG) neck was ordered which revealed a well-defined

cystic area with echogenic foci (and debris) measuring 5.6 x 3.3 cm on the left
anterolateral aspect of the neck deep to the muscles. His blood work was carried out
which showed a normal full blood count, erythrocyte sedimentation rate (ESR), liver
function, renal function and thyroid function test.

REASON FOR SURGERY? ADD SOME DETAILS ABOUT THE SURGERY, THE
APPROACH ETC. Surgical excision of the lateral neck mass was sent for
histopathology which revealed papillary cell carcinoma in left Branchial cyst (Figure 1).

The histopathology report revealed cyst wall of fibrous tissue with flattened epithelial
lining infiltrated by a malignant neoplasm along with papillary structures with fibro
vascular cores and colloid. The lining epithelial cells were columnar with nuclear
overlapping. Nuclear grooves and inclusions were also seen, some psammoma bodies
were also present in the cyst wall. Immunohistochemistry staining of Branchial cyst
through CK-19 and TTF-1 (thyroid transcription factor -1) was carried out which was
inconclusive for CK-19 but positive on TTF-1. The role of immunohisto-staining will be
explained later in case discussion.

DISCUSSION
A Branchial cleft cyst commonly presents as a solitary, painless mass in the neck of a
child or a young adult [?]. Branchial anomalies can sometimes present bilaterally [?].
Branchial cleft cyst is a developmental disorder of 2 nd Branchial arch and anomalies in
this region are more common (reference).

Ectopic thyroids are rarely found in the region of sublingual, submandibular, intra-
tracheal, mediastinum, esophagus, lung, heart, and aorta and even in the abdomen [?].
The most frequent site is the hypoglossal remnants or Branchial cleft cyst [?]. But in our
case this fact was not supportive. The presence of a thyroid carcinoma in a lateral neck
cyst could be considered as a result of a metastatic spread, but in our case, there was
no thyroid primary landing us with a quagmire of the diagnosis.

Imaging and FNAC don’t play a role in a diagnosis unless an excisional biopsy is carried
out [?]. Total thyroidectomy is strongly recommended in such cases along with
consideration of a selective neck dissection [?].

In immunohistochemistry staining TTF-1 (thyroid transcription factor, also called thyroid

specific enhancer binding protein) provides useful information on the functional activities
and/or differentiation of thyroid tumors, moreover about the neoplastic and hyperplastic
nature of the thyroid tissue [?]. In our case it was positive; however the drawback of
TTF-1 is that it cannot distinguish between primary and metastatic tumors of BCC.
(Reference). Another immunohistochemistry stain CK-19 (cytokeratin-19) which is a
very useful tool for the diagnosis of papillary carcinoma [?]. The immune reactivity for
CK-19 is not specific for papillary carcinoma thyroid, although the extent and intensity of
staining are considerably greater in the

CONCLUSION
Papillary carcinoma, however a rare tumor but its occurrence in Branchial cleft cyst
makes it a challenge in regards of a diagnosis as well as the treatment for the clinical
physicians and surgeons. Imaging and FNAC don’t play a role in a diagnosis unless an
excisional biopsy is carried out. Total thyroidectomy is strongly recommended in such
cases along with consideration of a selective neck dissection. (Reference)
REFERENCES

1.

Therefore, on the basis of histopathology report, the patient was diagnosed with
metastatic papillary thyroid carcinoma in Branchial cyst without any primary in the
thyroid gland. Thyroid scan in the patient gave the evidence of functioning tissue in the
region of both lobes of thyroid. To correlate with the thyroid scan, thyroid profile and
USG neck was carried out post operatively which was unremarkable showing no thyroid
nodule or cervical lymph adenopathy.