International Journal of Research in Medical Sciences
Gaikwad MD et al. Int J Res Med Sci. 2024 Jul;12(7):xxx-xxx
www.msjonline.org pISSN 2320-6071 | eISSN 2320-6012
DOI: https://dx.doi.org/10.18203/2320-6012.ijrms2024????
Case Report
Cervicofacial actinomycosis with chronic parotid sialadenitis-mimicking
malignant neoplasm of parotid gland: a rare case
M. D. Gaikwad*, Bibi Zainab, Amir Choudhary
Department of Surgery, JIIUS, IIMSR, Jalna, Maharashtra, India
Received: 15 May 2024
Revised: 31 May 2024
Accepted: 01 June 2024
*Correspondence:
Dr. M. D. Gaikwad,
E-mail: [email protected]
Copyright: © the author(s), publisher and licensee Medip Academy. This is an open-access article distributed under
the terms of the Creative Commons Attribution Non-Commercial License, which permits unrestricted non-commercial
use, distribution, and reproduction in any medium, provided the original work is properly cited.
ABSTRACT
Cervicofacial actinomycosis is an uncommon disease caused by Actinomycosis israelii. Chronic ‘sialadenitis’ of
parotid gland is an insidious inflammatory disorder and may form a fibrous mass. We report a case of 55 years old
female presenting with mass in right parotid region-on imaging-malignant neoplasm of parotid gland. But on
histopathology-actinomycosis and chronic parotid sialadenitis.
Keywords: Actinomycosis, Chronic parotid sialadenitis
INTRODUCTION CASE REPORT
Actinomycosis israeili is a gram-positive bacterium. They A 55-year-old female was admitted in the general surgery
are harmless but become virulent when there is breach in department with complains of a progressive painful
mucosa and presence of co pathogens. Cervicofacial swelling in the right parotid region with trismus and
actinomycosis still occurs occasionally and the lesion can restricted mouth opening of 4 weeks duration. Unable to
mimic many other diseases, including neoplasms of the eat and loss of weight. The patient did not give any
head and neck causing difficulty in diagnosis.1,2 history of trauma or tooth extraction. No history of fever.
She had consulted dental surgeon and received medicine
Classically actinomycosis manifests as a slow growing, with no relief. She belongs to poor socioeconomic class.
painless mass with multiple draining sinus tract on the History of chronic tobacco chewer.
skin or oral mucosa and occasionally presence of thick,
yellow exudate with characteristic sulphur granules. On local examination: Swelling in right parotid region of
face 4×4 cm, diffuse, firm to hard in consistency with
Chronic sialadenitis of the parotid gland is an uncommon irregular margins, fixity to overlying skin, no
clinical condition characterized by intermittent, often discoloration (Figure 1 and 2) local temperature normal,
painful parotid swelling and/or discharge of pus and softness on top of swelling, mobility restricted. Mouth
inflammatory changes in the affected gland.3,4 Chronic opening restricted allowing only 1 finger. On intraoral
sialadenitis of the parotid gland is an insidious examination few teeth missing. No caries. Skin
inflammatory disorder which tends to progress and excoriation after FNAC. No sinuses in the wound and no
maybe lead to the formation of a fibrous mass, chronic granules.
sialadenitis is a major disorder that can cause the salivary
hypofunction. Routine hematological examination was normal.
International Journal of Research in Medical Sciences | July 2024 | Vol 12 | Issue 7 Page 1
Gaikwad MD et al. Int J Res Med Sci. 2024 Jul;12(7):xxx-xxx
The mass was adherent to the parotid gland. Excised in
toto. The parotid gland was bulky and not looking like
malignant so superficial lobe of the parotid gland
removed; muscles not excised. 2-3 Lymph nodes
removed which was enlarged. Post operative period
uneventful. Discharged on tenth day. Post operative
patient has facial paralysis due to neuropraxia. Slowly
recovered.
Figure 1: Clinical image on presentation.
Figure 3: Post operative clearance of disease.
Figure 2: Clinical image lateral view.
Ultrasonography revealed hypoechoic lesion in right
parotid with cystic area and solid component within.
Enlarged lymph nodes in right submandibular region
neoplastic origin.
CECT face and neck-a large lobulated heterogeneously
enhancing mass lesion of size 5.4×3.3 cm in right parotid
space involving the right parotid gland superficial and Figure 4: Excised specimen.
deep lobe, masseter muscle, buccinator muscle and
portion of temporalis, multiple discrete cervical lymph
nodes level Ⅰ, Ⅱ, Ⅲ. These features are likely suggestive
of neoplastic pathology likely malignancy of right parotid
gland with metastatic type of lymphadenopathy.
FNAC from swelling suggestive of organizing
suppurative lesion.
Smear for staining and culture sensitivity (from
discharge). Gram stain-plenty of pus cells seen. No
organism seen. ZN stain-no acid-fast bacilli seen. Culture
sensitivity report sterile. X-ray chest was normal.
The patient undergone surgery-superficial parotidectomy
with mass excision (Figure 3-5). Ulcerated skin over the Figure 5: Post op image.
mass excised.
International Journal of Research in Medical Sciences | July 2024 | Vol 12 | Issue 7 Page 2
Gaikwad MD et al. Int J Res Med Sci. 2024 Jul;12(7):xxx-xxx
diseases, including neoplasms of the head and neck
causing difficulty in diagnosis.7
Actinomycotic infection is usually caused by tooth
extraction, odontogenic infection or trauma.8 The etiology
was unclear in our patient because she had no identifiable
portal of entry. However, actinomycosis in parotid region
involving masticator muscles, buccal and temporal and
vegetative foreign body in the mass with chronic parotid
sialadenitis has not been reported previously.
Chronic sialadenitis of the parotid gland usually
diagnosed as recurrent parotitis, non-obstructive parotitis,
obstructive parotitis, or benign lymphosialadenopathy. 9
Figure 6: Actinomycosis under 10×HE.
In actinomycosis, imaging techniques like USG and
CECT show inconsistent findings and are non-
contributing to positive diagnosis. Histopathology is the
gold standard for diagnosis.10 The disease is difficult to
diagnose as it commonly mimics like tuberculosis,
chronic granulomatous lesion and malignancy.4 Hence it
is considered as the most misdiagnosed disease, and is
listed as a rare disease by the office of rare disease (ORD)
of the national institute of health (NIH).
Management of cervicofacial actinomycosis includes
antibiotics and surgery.
In chronic sialadenitis of the parotid gland initial
management consists of antibiotics, analgesics and major
surgical procedures (superficial parotidectomy) in the
reported case.11,12
Figure 7: Actinomycosis under 40×HE.
In our case, clinically and on imaging it was not possible
Histopathology to differentiate two pathologies separately. On imaging-
USG and CECT diagnosed as malignant neoplasm of
Parotid: ducts and acini with moderate to severe parotid with secondaries in lymph nodes.
infiltration of lymphocytes. Areas of fibrosis,
hyperplastic lymph node. On histopathological examination we found, the mass
covered with skin showed-actinomycosis and fragments
Skin covered mass pseudo cystic structure, deep to this of vegetative foreign body surrounded by acute
fragment of vegetative foreign body surrounded by acute inflammatory reaction. Parotid gland shows infiltration of
inflammatory reaction, areas of fibrosis. Dark basophilic lymphocytes. Follicular hyperplastic lymph node
structures having eosinophilic sun ray appearance identified, which were inconsistent with the imaging
surrounded by strong neutrophilic aggregates- interpretation of neoplasm. Thus, a very rare case of
actinomycosis and parotid chronic sialadenitis. actinomycosis and vegetative foreign body in the parotid
region along with chronic sialadenitis of parotid gland.
DISCUSSION
CONCLUSION
Actinomycosis is a chronic, suppurative, granulomatous,
bacterial disease primarily caused by Actinomycosis Mass in parotid region can’t be diagnosed clinically and
israelii, a gram positive, non-acid fast, anaerobic or on imaging. Imaging may confuse as in this case.
microaerophilic filamentous branched bacterium, difficult Histopathology is the gold standard process after
to culture.5-7 The bacteria are normal inhabitant of oral resection. We think, the choice of treatment in such cases
cavity and found in caries teeth, tonsillar crepts, gingival is radical resection of the lesion and superficial
crevices.6 They are harmless but become virulent when parotidectomy, that to be decided on operation table.
there is breach in the mucosa and presence of co
pathogens. Cervicofacial actinomycosis still occurs Funding: No funding sources
occasionally and the lesion can mimic many other Conflict of interest: None declared
Ethical approval: Not required
International Journal of Research in Medical Sciences | July 2024 | Vol 12 | Issue 7 Page 3
Gaikwad MD et al. Int J Res Med Sci. 2024 Jul;12(7):xxx-xxx
REFERENCES 8. Omura D, Rai K, Nobuchika K, Fumio O.
Actinomycosis induced trismus and orbital
1. Chatterjee R, Shah N, Kundu S, Abdul Mahmud SK, involvement. Intern Med Adv Public.
Bhandari S. Cervicofacial Actinomycosis mimicking 2019;58(1):153-4.
osseous neoplas: a rare case. J Clin Diagn Res. 9. Wang S, Marchal F, Zou Z, Zhou J, QI S.
2015;9(7):ZD29-31. Classification and management of chronic
2. Ivanova C, Bachurska S, Popov K. Actinomycosis of sialadenitis of the parotid gland. J Oral
the parotid gland. Challenges in diagnosis and Rehabilitation. 2009;36(1);2-8.
treatment- case report. Int Bull Otorhinolaryngol. 10. Sittarai P, Srivanitchapoom C, Pattarasakulchai T,
2021;2:40-2. Lekawanavijit S. Actinomycosis presenting as a
3. Motamed M, Laugharne D, Bradley PJ. Management parotid tumor. Elsevier Ireland Ltd. 2011;39(2):241-
of chronic parotitis: a review. J Laryngol Otol. 3.
2003;117(7):521-6. 11. Amin MA, Bailey BM, Patel SR. Clinical and
4. Malloy KM, Rosen D, Rosen MR. Salivary gland radiological evidence to support superficial
diseases surgical and medical management: parotidectomy as the treatment of choice for chronic
Sialadenitis. New York: Thieme Medical Publishers, parotid sialadenitis: a retrospective study. Br J Oral
Inc. 2005. Maxillofac Surg. 2001;39(5):348-52.
5. Michael J, Belmont MD, Philomena M, Behar MD, 12. Moody AB, Avery CM, Waish S, Sneddon K,
Mark K, Wax MD. Atypical presentations of Langdon JD. Surgical management of chronic
actinomycosis. Head Neck. 1999;21(3):264-8. parotid disease. Br J Oral Maxillofac Surg.
6. Lancella A, Abbate G, Foscolo AM, Dosdegani R. 2000;38(6):620-22.
Two unusual presentations of cervicofacial
actinomycosis and review of literature. Acta
Otorhinolaryngeal Ital. 2008;28(2):89-93.
7. Volante M, Contucci AM, Fantoni M, Ricci, Galli J. Cite this article as: Gaikwad MD, Zainab B,
Cervicofacial Actinomycosis: still a differential Choudhary A. Cervicofacial actinomycosis with
diagnosis. Acta Otorhinolaryngol Ital. chronic parotid sialadenitis-mimicking malignant
2005;25(2):116-19. neoplasm of parotid gland: a rare case. Int J Res Med
Sci 2024;12:xxx-xx.
Provide at least three keywords.
International Journal of Research in Medical Sciences | July 2024 | Vol 12 | Issue 7 Page 4