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REVIEW OF LITERATURE
BILATERAL SYNCHRONUS MULTIFOCAL
WARTHIN'S TUMOR - A CASE REPORT & REVIEW
Shekhar Gogna1, Sanjeev Parshad2, RK Karwasra3, Dharampal Yadav4, Priya
Goyal5
1-Assistant professor, 2- Professor 3- Senior Professor, 4,5- Resident,
Department of surgery, PGIMS Rohtak, Haryana , India- 124001
ABSTRACT:
Warthin’s tumor is a benign parotid tumor with strong predilection for male sex and smoking. Bilateral
warthin’s tumors have been reported in literature however bilateral warthin’s tumor involving both
supertficial and deep lobe is rare condition. Surgery offers the only definitive cure. In this tricky situation
bilateral preservation of facial nerves and total removal of tumor are important points to consider. We
report a rare case of a 65 years male with bilateral mutifocal warthin’s tumor involving both superficial
and deep lobes of parotid gland. This report also highlights the importance of surgical decision to be taken
in such situation.
KEYWORDS: Warthin’s tumour
Cite this Article:S Gogna, Sanjeev P, RK Karwasra, Dharampal Y, Priya Goyal:Bilateral synchronus
multiofocal warthins tumor- A case report & Review, Journal of Head & Neck physicians and surgeons Vol
3 ,Issue 2, 2015 :Pg43-9
INTRODUCTION:
Warthin's tumor is the second most frequent benign tumor of the parotid gland representing 6 to
10% of all tumors of the salivary glands.1 it is also known by the name of adenolymphoma of the
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parotid gland or papillary cystadenoma lymphomatosum. It presents as an asymptomatic, slowly
growing mass in parotid fossa. It has predilction for male sex (M:F=10:1) affecting in 5th& 6th
decade of life and has propensity for smokers.2 Studies dedicated to its epidemiologic and
demography have shown increasing rise in females because of increased trend of smoking in
females.3,4 Warthins tumor is usually unilateral and metachronus, the incidence of bilaterality is
only 6%.5 The combination of bilateral and multifocal warthin’s tumor is even a rarity. In a
paper by Ibi et al frequency of bilateral multifocal warthin’s tumor was 0.9% . 6 The second
study by Lamelas et al reportd it to be about 3%. 3 Surgical resection is the only definitive cure.
The decision to operate becomes tricky when warthin’s tumor is synchronus and more so
involving both the superficial lobe as well as deep lobe. This case report brings forth the rare
association of synchronus multifocality in parotid tumor and aims to help surgeons to decide
upon the type and timing of surgical resection.
CASE REPORT:
We present a case of a gentleman of 65 years of age retired school teacher. Chief complaint was
that patient developed slowly growing non tender mass in right parotid region 2 years ago. This
was followed 1 year later with similar kind of complaint on left side. Medical history was
significant for smoking, he was a heavy smoker (about 20 cigarettes/day), non alcoholic and had
Diabetes milletus- II. On general physical examination the size of swelling in right parotid region
was 4*4 cm and 3*3 cm on left side, both were firm in consistency, smooth surface and had well
defined margins. Intra oral examination was normal. Bilateral facial nerve functions were
normal. Provisional diagnosis of bilateral warthin’s tumor was made. Investigations were
performed, FNAC from both the lesions showed warthins tumor (figure I). MRI was done which
showed T1 enhancing lesions in bilateral parotid gland involving both superficial as well as deep
lobes (figure II). Total conservative parotidectomy was done on right side followed by same
procedure on left side after 2 months(figure III, IV). We routinely cover the exposed facial nerve
with pedicled sternocleidomastoid flap to prevent frey’s syndrome figure (V)
Postoperatively
facial nerve functions were normal ( figure VI).
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Figure 1- , FNAC from both the lesions showed
warthins tumor
Figure 2- T1 enhancing lesions
in bilateral parotid gland involving both superficial as well as deep lobe.
Figure 3- Total conservative parotidectomy
was done on right side
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Figure 4- Total conservative parotidectomy
was done on left side after 2 months
Figure 5 – Covering of the exposed facial
nerve with pedicled sternocleidomastoid flap to prevent frey’s syndrome figure
Figure 6 - Facial nerve functions were
normal
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DISCUSSION:
Term “Warthin tumor”, was named after Aldred Warthin, the American pathologist.7 Warthin’s
tumor is grossly a cystic neoplasm with thin fibrous capsule. Cysts are filled with mucoid
substance and solid areas. Microscopically it has both epithelial and lymphoid component. Cystic
spaces are surrounded by double layer of oncocytic epithelium. Outer layer is composed of
columnar cells with finely granular cytoplasm with small papillary infoldings. Inner layer is
composed of small cuboidal epithelium. Lymphoid tissue forms the stroma with reactive
germinal centre formation.8 There are two most common theories about the histogenesis of this
tumor. The first theory asserts that Warthin’s tumor could be an atypical parotid adenoma in
which an inflammatory response causes extensive lymphocytic infiltration. The second theory
states that, there is entrapment of salivary ductal cells in embryonic lymphocytic-rich tissues.2
Radiological investigations are of paramount importance during management of this tumor.
Ultrasound feature of Warthin’s tumor are based on its echo structure, margins and vascularity.
CT utilizes structure, margins, number of lesions, pattern of enhancement, to differentiate
between various parotid lesions. The current role of MRI is to define facial nerve anatomy and
deep lobe leisons.9 No radiological study is superior over another. Surgeon should be able to
interpret the anatomy and characterstics of the lesion on his own as provided by radiological tests
to be able to be safely and completely resect it. Definitive diagnosis can only be made at
histopathological excision only. We routinely do not advocate FNAC of clinically benign parotid
lesions. There are few reasons for our philosophy, FNAC can be challenging in the case of
multiple lesions and there are sampling errors.9 Despite the fact some studies report excellent
sensitivity and specificity of FNAC, some cases have been reported where FNAC was thought
to be responsible for intralesional hemorrhage that switched a classical histology to the
metaplastic subtype.10 The treatment for bilateral tumors is the surgery, similar to that indicated
for solitary tumors, i.e., superficial parotidectomy for the superficial lobe, or total parotidectomy
with facial nerve preservation in case of deep lobe tumors. The important issue that arises is the
timing of surgery for bilateral tumors when both lobes are involved. In a paper by Nicolai et al,
they reported two cases and one of them had bilateral tumor which were removed in same
setting. In this study status of bilateral deep lobes is not mentioned10 Ascani reported a case of
bilateral warthin’s tumor simple enucleation was performed in same setting. In this case probably
deep lobe was not involved.11 In another case report by Kremp in 2008 staged procedure of
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bilateral resection was done with the gap of 2 years, as the other mass continued to enlarge,
however status of deep lobe is again not clear in this report.12 On searching the literature the
clear consensus on this kind of situation was missing. To fill this gap we strongly feel that such
reports should come from surgical centers. Staged procedure should better option if deep lobes
are also involved along with superficial lobes. Surgery in the same sitting can be done if bilateral
superficial lobes only are involved. Regular follow up is of paramount importance as there is
possible association of Warthin’s tumour with extra-salivary neoplasms.13
CONCLUSION:
Warthin’s tumor is a benign tumor of parotid gland. Bilateral multifocal warthin’s tumor
involving both superficial and deep lobes is a rare condiotion. Treatment guidelines are obiously
not clear. Staged total parotidectomy should be the preferable option keeping in mind the
preservation of facial nerves. Regular follow up is of paramount importance.
REFERENCES:
[1]. Ellis GL, Auclair PL, Gnepp DR. Surgical pathology of the salivary glands.
Philadelphia: W.B. Saunders; 1991,pp.165.
[2] Teymoortash A, Werner JA: Tissue that has lost its track: Warthin’s tumour.
Virchows Arch 2005, 446:585-588.
[3] Lamelas J, Terry JH Jr, Alfonso AE. Warthin’s tumor: multicentricity and increasing
incidence in women. Am J Surg. 1987;154:347-51.
[4] Monk JS, Church JS. Warthin’s tumor: a high incidence and no sex predominance in
central Pennsylvania. Arch Oto.1992 : 118(5):477-478.
[5] Maiorano E, Lo Muzio L, Favia G, et al. Warthin’s tumor: a study of 78 cases with
emphasis on bilaterality, multifocality and association with other malignancies. Oral
Oncol. 2002;38(1):35-40.
[6] Frazetta M, Cortese E, Matranga S, Renda F, Divita G. Warthin’s tumor of the parotid
gland. GChir 1997; 18: 101-105.
[7] WARTHIN A.S., Papillary cystadenoma lymphomatosum: a rare teratoid of the
parotid region, J Cancer Res, 1929,13:116–125.
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[8] Simpson RHW, Eveson JW. Warthin’s tumor. In: Barnes L, Eveson JW, Reichart P,
eds. World Health Organization Classification of Tumors: Pathology and Genetics of
Head and Neck Tumors. Lyon: IARC Press, 2005;263-265.
[9] Djekidel et al. Warthin’s Tumor Multimodality Imaging. Anatomical and
Scintigraphy Imaging Review, Including PET-CT and SPECT-CT. OMICS J Radiology
2013, 2:3.
[10] Nicolai et al. Bilateral and multifocal Warthin’s tumor of parotid gland: two case
reports and review of literature. Oral Implantol (Rome). 2014 Jan-Mar; 7(1): 25–31.
[11]Ascani, T. Pieramici1, C. Rubini1, M. Messi, P. Balercia. Synchronous bilateral
Warthin’s tumours of the parotid glands: a case reportACTA otorhinolaryngologica ita
lica 2010;30:310-312.
[12] Kremp AN, Nelson LB. Bilateral Warthin Tumors of the Parotid Gland Head Neck
Pathol. 2008 Sep; 2(3): 175–176
[13] Scasso CA, Papini M, Eligi C, et al. An unusual neck mass: theWarthin’s tumor. Acta
Otorhinolaryngol Belg 1998;52:55-7.
Acknowledgement- NIL
Conflict of Interest- None Declared
Ethical approval: This article does not contain any studies with human participants or animals
performed by any of the authors Informed consent: Informed consent was obtained from patient
to report this case for educational purpose .
Correspondence Addresses :
Shekhar Gogna
Phne- +919896379623
Assistant professor, department of surgery
PGIMS Rohtak, Haryana , India- 124001
Email- [email protected]
Res: 478-GF, Omaxe city, rohatk , India 124001
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