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DOI No.: 10.21176/ ojolhns.0974-5262.2015.09.

SOLITARY PAROTID MASS PRESENTING AS SARCOIDOSIS :


UNUSUAL PRESENTATION
*V. V. Rokade, **N. A. Pathak, ***S. V. Nemade
ABSTRACT :
Objective: To demonstrate an unusual case
of sarcoidosis in which the patient presented with
solitary parotid mass and no other manifestations
of the systemic disease.

INTRODUCTION:
Jonathen Hutchinson first reported sarcoidosis in
1869. Several causative agents have been implicated
in its aetiology which includes infective agents like
mycobacterium and no infective agents like exposure

Case report: A 12years old girl child presented with

to beryllium dust. Detection of mass of parotid gland is

history of swelling in right parotid region since three years.

generally considered an indication for superficial

On local examination there was firm smooth non tender

parotidectomy. In most cases pathology will identify a

swelling palpable in right parotid region. FNAC was


suggestive of chronic granulomatous disease. Sr Calcium,
ACE enzymes, Serology, ANCA was normal. Monteux test
was negative. ECG was normal. Superficial parotidectomy
was done. Histopathology features were suggestive of

euplastic process. We describe a case in which patient


presented with discrete solitary parotid mass that was
turned out to be sarcoidosis on histopathology.

CASE REPORT:

sarcoidosis.

A 12years old girl child presented in ENT OPD


Conclusion: We believe that this case is of interest
as descriptions of such presentations of sarcoidosis as
a solitary discrete mass in parotid gland and absence
of systemic involvement are not prevalent in the
literature. As all the investigations were normal, only
histopathology report after superficial parotidectomy
guided us to reach up to final diagnosis of sarcoidosis .

with history of swelling in right parotid region since

Keywords : Parotid Sarcoidosis, Non


caseating granulomatous disease.

opening was normal. There were no signs of facial

Corresponding Address:

of

size with or without eating. General practitioner treated

her with antibiotics with no improvement.


On local examination there was firm smooth non
tender swelling palpable in right parotid region. Duct
paralysis. General examination revealed no evidence
peripheral

lymphadinopathy

&

hepatosplenomegaly. CVS, RS and


examination were within normal limits.

no

CNS

A-2, 303 Sun Empire, Sun City Road


Vadgaon Bk., Pune-51
Ph.: 9922160881
E-mail: vidyarokade@hotmail.com

On diagnostic work up her haemogram and ESR


was normal. FNAC showed plenty of epithiloid cell
granulomas with multinucleated giant cells; features
suggestive of chronic granulomatous disease.

*Assistant Professor of ENT, **Assistant Proffesor ENT Dept., ***Assistant Proffesor ENT Dept., Smt Kashibai Navale
Medical College and General Hospital Ambegaon Narhe, Pune

33

Vol.-9, Issue-I, Jan-June - 2015

Dr.Vidya V.Rokade.

three years with no history of fever or fluctuations in

DOI No.: 10.21176/ ojolhns.0974-5262.2015.09.1

To rule out various granulomatous diseases various for AFB. All the features were suggestive of

investigations carried out. Biochemical investigations: sarcoidosis (Fig II).


Blood Sugar, KFT, LFT, Sr Calcium, ACE enzymes,
Serology, ANCA were normal. Monteux test was
negative. ECG was normal. Radiological investigations:
Xray

chest, USG abdomen revealed no

abnormality.USG patotid with colour Doppler showed


enlarged right parotid gland with multiple lymph nodes
in it.
CT& MRI

(plain & contrast) demonstrated

enlargement of right parotid gland with homogenous


enhancement with involvement of superficial lobe of
gland with multiple lymph nodes in it (FIG I).

Fig.-2: H & E Stain400X

Postoperative period was uneventful. Patient


is under close follow up for last one year without
any medical treatment. Her x ray chest remained
normal and had not manifested any symptoms of
systemic sarcoidosis.
Fig.-2: Showing homogenous enhancement of superficial
labe of parord in Right side in C.T.

Inspite of long list of investigations we could not


arrive at the final diagnosis. Hence, to obtain tissue

Vol.-9, Issue-I, Jan-June - 2015

diagnosis

superficial

parotidectomy

was

done.

Intraoperatively we found firm mass in superficial lobe


of parotid gland with two to three enlarged lymph nodes

DISSCUSION:
Despite extensive studies, no agent has been
identified as the cause of sarcoidosis. Sarcoidosis
is currently considerd as a chronic inflammatory
disease, distinguished by hyperimmune reactions
to an unspecified agent at the lesion sites.

granulomas surrounded by lymphocytes & Langerhans

Sarcoidosis affects lungs in 90% of cases


followed by lymph nodes & spleen. Salivary
glands are rarely involved.

type of giant cells.Also seen were Schwamanns bodies

Parotid gland involvement occurs in 0.5to 15%

in giant cells & asteroid bodies as well as Hamazaki

patients of systemic sarcoidosis. However, in 6% of


patients it is confined to parotid gland only& may

. Deep lobe was normal on palpation . Histopathogy


showed

numerous non caseating epitheloid cell

Wegenbreng

inclusions

with

no

malignancy. Z N staining was negative


34

evidence

of

DOI No.: 10.21176/ ojolhns.0974-5262.2015.09.1

manifest as uni or bilateral painless swelling.


Parotid enlargement is a more frequent finding in
children diagnosed with early onset sarcoidosis.
Parotid gland involvement in sarcoidosis may
manifest itself in a variety of clinical patterns. The most
common pattern is represented by bilateral diffuse
parotid

gland.

second

pattern

is

and those with the progressive disease, although the


dosage and the duration of the treatment are not well
defined and no consensus exists as to whether
treatment should be given to asymptomatic patients
and patients with only mild disease. Patients are
usually treated for at least six months initially.

characterized by asymptomatic parotid lesions. Third

Methotrexate. Azathioprine, Cyclophosphamide

pattern is in the form of Herefords syndrome. A Clinical

& recently Infliximab has been tried with varying

pattern of sarcoidosis characterized by isolated parotid

success rate for the patients refractory to steroids.

enlargement with absence of general symptoms of


sarcoidosis would be extremely uncommon.

However,

spontaneous

regression

of

10

this

disease has been reported. Early diagnosis and

In cases of solitary parotid sarcoidosis preoperative

treatment can slow up or stop the progression to

clinical diagnosis is difficult as there is no specific lab test

organ destruction. Research is still on going

as such. However FNAC can be helpful. Depending upon

regarding better diagnostics & treatment modalities.

the conclusion of FNAC we have to rule out various

SUMMARY:

infective and granulomatous conditions. Markers of


activity for sarcoidosis include raised serum ACE levels,

We believe that this case is of interest as

abnormal calcium metabolism, positive Kveim Siltzbach

descriptions

skin test & radioactive gallium scanning (Gallium-67

sarcoidosis as a solitary discrete mass in

Citrate)

4,5

. However, ACE levels are typically elevated in

80% of children

parotid

6,7

gland

such
and

presentations

absence

of

of

systemic

involvement are not prevalent in the literature.

and 60% of adults with late onset

sarcoidosis. The ACE test has proven to be less sensitive

of

As all the investigations were

normal, only

in patients diagnosed with early onset sarcoidois. Many

histopathology

other conditions are associated with elevated ACE levels

parotidectomy guided us to reach up to final

like milliary TB, Leprosy and Gauchers disease. It is

serologic testing and special tissue staining techniques.


The combination of an elevated ACE levels and positive
gallium-67 scan is 85% specific for sarcoidosis.

superficial

diagnosis of sarcoidosis

important to rule out various infectious and granulomatous


diseases on the basis of history and physical examination,

report after

So we believe this makes the case an unusual


presentation.
Acknowledgement: We would like to express our

appreciation to Dr

S.D.Deshmukh

Professor,

- 2015

enlarged

the cornerstone of treatment for symptomatic patients

A definitive diagnosis of sarcoidosis is best

and accurate diagnosis.

achieved by integrating clinical data with presence of

DISCLOSURES

noncaseating granulomas. The main goal of the

(a) Competing interests/Interests of Conflict- None

granuloma formation that ultimately causes end stage


organ destruction by hyaline fibrosis. Steroids remains

(b) Sponsorships - None

Vol.9,

treatment is to minimize or prevent inflammation &

Issue- JanJune
I,

Department of Pathology SKNMC, Pune for prompt

(c) Funding - None


35

DOI No.: 10.21176/ ojolhns.0974-5262.2015.09.1

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