Otolaryngology Case Reports 9 (2018) 26–28

Contents lists available at ScienceDirect

Otolaryngology Case Reports

journal homepage: www.elsevier.com/locate/xocr

An unusual case of tuberculous parotitis T

a b c b,∗
Dominique Bohorquez , Jason G. Newman , John Stern , Karthik Rajasekaran
a
Perelman School of Medicine at the University of Pennsylvania, Philadelphia, PA, USA
b
Department of Otorhinolaryngology: Head and Neck Surgery, University of Pennsylvania, PA, USA
c
Department of Infectious Diseases, University of Pennsylvania, PA, USA

A B S T R A C T

Background: Tuberculosis (TB) of the parotid gland is exceedingly rare, even in

areas of high disease burden. We report a case of isolated tuberculosis (TB) of the parotid gland.
Methods: 25-year-old Puerto Rican female, who was born and raised in the United States, presented with a 3-month of history of recurrent left parotid gland swelling
and pain. She had three attempts of fine needle aspirations (FNA), which were non-diagnostic. She was treated for a 3-month duration of antibiotics without much
success. She did not have any constitutional symptoms, personal history of immunosuppression or prior exposure to TB. On CT and MRI, there appeared to be a fairly
well-circumscribed mass in the deep lobe of left parotid. Because various FNA were unsuccessful in identifying the etiology of the mass, the decision was made to
perform a deep lobe parotidectomy.
Results: Histopathology revealed a large necrotizing granuloma with fibrous wall and numerous histiocytes. Based on these findings, she had a PPD and quantiferon
gold test, both of which were positive. A chest X-ray was negative, thus demonstrating that she had latent TB.
Conclusion: We believe this is one of the first reported cases of tuberculous parotitis in the United States. Clinicians should be aware of this rare entity and may
consider ordering a PPD test in those patients who have failed treatment or yielding a diagnosis.

Introduction to two different emergency rooms, received another course of aug-

mentin and a course of doxycycline. She ultimately was referred to our
Although extrapulmonary involvement of tuberculosis (TB) ac- institution for further care.
counts for 20% of overall active TB cases [1], parotid involvement is Upon presentation, she described 8 out of 10 pain in the left pre-
exceedingly rare with only a limited number of reported cases, even in auricular region that radiated to her mouth and jaw, limiting her ability
endemic regions. Tuberculous parotitis has been described in the lit- to eat. She did not have any fevers, night sweats, unintentional weight
erature as a slow-growing painless mass, often indistinguishable from loss or chills. She denied any history of trauma, oral drainage, or er-
malignancy [2]. This report describes an unusual presentation of tu- ythema over the region. She also denied any history of recent travel,
berculous parotitis in a non-endemic region in a patient without evi- immunosuppression, or previous exposure to TB. On examination, there
dence of primary disease. was minimal pre-auricular edema with exquisite tenderness over the
left parotid region without overlying skin changes. Drainage was not
Case report expressed from the parotid duct and there were no palpable cervical
lymph nodes. Her facial nerve function was intact, and the remainder of
A 25-year-old Puerto Rican female presented with a 3-month history her exam was normal.
of a recurrent and painful left parotid mass. She was born and raised in An MRI scan was ordered, which revealed a 2.3 × 2.1 cm periph-
the United States, and never traveled outside the country, except for a erally enhancing collection located in the left parotid gland (Fig. 2).
2-month period to Puerto Rico at the age of 13. When she initially Diagnostic considerations based on these features were not suggestive
presented to the emergency room, she was in 8 out of 10 pain. She had for pleomorphic adenoma or malignancy but rather an infectious or
no leukocytosis on her CBC. A CT scan was performed, and it was inflammatory process. FNA revealed necrotic cellular debris admixed
concerning for an abscess (Fig. 1). Given the location, an IR-guided FNA with histiocytes, lymphocytes, and neutrophils, suggestive of an in-
was performed, and no purulence was noted. She was preemptively flammatory process. Despite these findings, she had no leukocytosis or
started on unasyn and transitioned to augmentin based on her physical elevated inflammatory markers during any of her admissions. She also
exam and CT findings. Over the course of the next 3 months, she went had a negative rheumatological work-up.

∗
Corresponding author. Department of Otorhinolaryngology: Head and Neck Surgery, Penn Medicine, 800 Walnut Street, 18th Floor, Philadelphia, PA, 19104,
USA.
E-mail address: karthik.rajasekaran@uphs.upenn.edu (K. Rajasekaran).

https://doi.org/10.1016/j.xocr.2018.07.003
Received 25 June 2018; Accepted 16 July 2018
Available online 25 July 2018
2468-5488/ © 2018 Elsevier Inc. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/BY-NC-ND/4.0/).
D. Bohorquez et al. Otolaryngology Case Reports 9 (2018) 26–28

Fig. 1. CT Neck demonstrating a lesion in the left parotid concerning for an

abscess. Fig. 4. Gross appearance of the excised lesion.

granuloma with fibrous wall and numerous histiocytes. The tumor was
positive for the histiocyte marker CD68 but negative for fungal or AFB
stains. Postoperatively, the patient had a positive PPD and quantiferon
gold assay, suggesting previous exposure to TB. The patient had a ne-
gative chest x-ray and therefore was diagnosed with active extra-
pulmonary disease. She was treated with 2 months of rifampin, iso-
niazid, pyrazinamide, and ethambutol and continued on rifampin and
isoniazid for 4 additional months.

Discussion

TB is a necrotizing granulomatous disease caused by Mycobacterium

tuberculosis. This bacterium primarily affects the lungs but has the po-
tential to spread to extrapulmonary sites, more commonly the liver,
spleen, bone marrow, but rarely to salivary glands. Dixit et al. proposed
that the antibacterial properties of the proteolytic enzymes found
Fig. 2. Axial STIR MRI of the neck demonstrating the lesion in the left parotid within the gland and its continuous salivary flow prevent the multi-
gland. plication of mycobacteria, thereby reducing the likelihood of infection
in this area [1]. Although the exact pathogenesis of tuberculous par-
otitis remains unclear, the literature postulates that hematogenous
spread from a distant pulmonary site, lymphatic spread of infection
from a previously infected lymph node, autoinoculation of the parotid
gland by infected sputum, or spread from the adjacent oral cavity are
possible mechanisms [3,4].
The clinical presentation of tuberculous parotitis is variable, with
most reports describing it as a slowly-growing painless parotid mass
with only 25% of patients presenting with concomitant pulmonary TB
[2–4]. Like most reported cases, our patient did not have any signs or
symptoms of TB outside the parotid gland but did have some unusual
elements to her presentation. Firstly, she presented with latent TB in a
non-endemic region. Tuberculous parotitis is already exceedingly rare,
even in endemic areas. The year 2017 had the record lowest incidence
rate of TB in the United States (2.8 cases per 100,000 population)5,
further making this an unusual presentation given she had no recent
travel history or known TB exposure. The patient also reported debili-
tating pain from the affected site, refractory to trials of attempted
drainage, antibiotics, or pain medication. Reported cases describe tu-
Fig. 3. Intraoperative pictures before removal of the lesion.
berculous parotitis as a painless phenomenon [2–5].

For definitive diagnosis, a left total parotidectomy with facial nerve

preservation was performed. Intraoperatively it was clear that this was Conclusion
a deep lobe parotid lesion, which required meticulous dissection to
excise (Fig. 3). A 2.0 cm yellow, solid, well-circumscribed tumor was Although this is an exceedingly rare presentation of a parotid lesion,
removed (Fig. 4). No cultures were sent on the specimen because of this case highlights the importance of considering other rarer causes of
high suspicion for tumor. infection when standard treatment measures fail. A PPD is a relatively
Histopathological examination revealed a large necrotizing simple test that perhaps should be considered in cases of refractory
medical therapy.

27
D. Bohorquez et al. Otolaryngology Case Reports 9 (2018) 26–28

Appendix A. Supplementary data of Health and Human Services, CDC; 2017.

[2] Dangore-Khasbage S, Bhowate R, Degwekar S, Bhake A, Lohe V. Tuberculosis of
parotid gland: a rare clinical entity. Ped Dent 2015;37:70–4.
Supplementary data related to this article can be found at https:// [3] Dixit R, Shah V, Dixit K, Solanki RN, Kotadia J. Tuberculous abscess of parotid gland.
doi.org/10.1016/j.xocr.2018.07.003. J Indian Acad Clin Med 2005;6:161–3.
[4] Gupta V, Patankar K, Shinde A, Bhosale C, Tamhane A. Tuberculosis of the parotid
gland. Case Rep Radiol 2012;2012:1–3.
References [5] Lee IK, Liu JW. Tuberculous parotitis: case report and literature review. Ann Otol
Rhinol Laryngol 2005;114:547–51.
[1] CDC. Reported tuberculosis in the United States, 2017. Atlanta, GA: US Department

28