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The Journal of Laryngology and Otology

November 1998, Vol. 112, pp. 1092-1094

Mucocele mimicking a Warthin's tumour recurrence


D. REJALI, F.R.C.S., R. SIMO, F.R.C.S., M. SMALL, F.R.C.S.

Abstract
We report an unusual case of an extravasation mucocele complicating superficial parotidectomy. The tumour
excised was a Warthin's tumour. Three months following the primary surgery a cystic lesion appeared in the
parotid bed. It was initially thought to represent a recurrence. The area was re-explored and a mucocele excised.
The pathogenesis of mucoceles and the difficulties encountered when dealing with parotid tumour recurrence
are discussed.
Key words: Adenolymphoma; Parotid neoplasm; Post-operative complications

Introduction malignancy. Full blood count and urea and electrolytes


Warthin's tumour (adenolymphoma) is a benign mono- were normal. The liver function test showed an isolated
morphic adenoma (Seifert et ah, 1986). It is the second raised gamma GT. Chest radiograph was normal.
most frequently encountered tumour in salivary glands The patient subsequently underwent superficial paroti-
after pleomorphic adenoma. It represents 15 per cent of all dectomy. A 3 cm in diameter nodule was removed with the
epithelial salivary tumours. It typically occurs in the lower superficial parotid gland. Histopathological examination of
part of the parotid salivary glands of men in their sixth or the specimen revealed the presence of a completely
seventh decades. Bilateral tumours are observed in 10 per excised Warthin's tumour (Figure 1). Post-operatively the
cent of cases (Seifert et ah, 1986). Adequate surgical patient had neuropraxia of the marginal mandibular
resection is usually curative. Post-operative complications branch of the facial nerve. This had resolved one month
following superficial parotidectomy include facial nerve later.
palsy, Frey's syndrome, seroma/haematoma or wound On review at three months later the patient had
infections. Recurrence after adequate resection of this developed a 1 cm non-tender swelling in the inferior
tumour is reported to be between 0 and 12 per cent region of the right parotid bed (Figure 2). Fine needle
(Leverstein et ah, 1997). Surgery for recurrent tumours is aspiration cytology revealed cyst debris, inflammatory cells
difficult due to fibrosis and there is an increased risk of and macrophages.
damage to the facial nerve (Conley, 1988). Clinically it was felt that recurrence could not be
The occurrence of cystic lesions in the parotid region excluded and therefore she underwent re-exploration of
after local surgery has been reported. These include the parotid region. The deep lobe of the parotid gland and
retention cyst after rhytidectomy - face lift - (Habal, an inferiorly placed one cm nodule was removed. The
1978) and epidermoid cyst following otomastoid surgery histopathological examination of the specimen revealed a
(Shaheen et ah, 1975; Thompson and Bradley, 1991).
We describe an extravasation mucocele as a complica-
tion of Warthin's tumour excision.

Case report
A 66-year-old female was referred with a two-month
history of a painless swelling in the region of the angle of
the right mandible. Past medical history included hyster-
ectomy for papillary adenocarcinoma of uterus 20 years
prior to presentation. She had a history of heavy alcohol
intake. There are no other otolaryngological symptoms nor
other relevant past medical history. On examination a soft
non-tender 4 cm in diameter mass was noted in the tail of
the right parotid gland. There was no facial paralysis or
regional lymphadenopathy. There was no other abnor-
mality on otolaryngological or general examination. Fine
needle aspiration cytology revealed 2 ml of straw-coloured
fluid. The cytopathological examination of the fluid FIG. 1
showed cohesive epithelial cells, erythrocytes, macro- Histopathological section of the completely excised Warthin's
phages and amorphous debris. There was no evidence of tumour (H & E; X 50).

From the Department of Otolaryngology and Head and Neck Surgery, Royal Preston Hospital, Preston, UK.
Accepted for publication: 10 August 1998.

1092
CLINICAL RECORDS 1093

At nine months following re-exploration she remains


well with no evidence of recurrence.

Discussion
Primary parotid gland cysts in adults are rare, represent-
ing five per cent of all parotid tumours (Richardson et al,
1978; Pieterse and Seymour, 1981; Cohen et al, 1984).
These fall into three categories: epidermoid, branchial and
ductal (retention) cyst (Pieterse and Seymour, 1981).
Parotid cysts can occur in association with tumours. The
most frequent association occurs with Warthin's tumour
(Richardson et al, 1978).
Mucoceles are round circumcised lesions that contain
mucus. They are classified into retention and extravasation
mucoceles. The latter are common in younger patients and
occur most frequently in the minor salivary glands of the
lower lip, cheek, floor of mouth, tongue, palate and upper
lip (Seifert et al, 1986). The aetiology of these is traumatic.
The lesions can be produced experimentally by ligation or
division of a salivary duct (Bhaskar, 1975). The competing
factors that determine the formation of a mucocele are the
rate of mucus production and the speed of phagocytosis of
the extravasated mucus (Seifert et al, 1986). The muco-
celes previously reported in the parotid are of the retention
variety (ductal cyst). The pathogenic process being partial
obstruction of the duct due to kinks, microliths or
inspissated secretions (Seifert et al, 1986). There are no
reports of the extravasation variety as a primary occur-
rence in the parotid gland.
Well recognized complications of superficial parotidect-
omy include haematoma, facial paralysis, damage to
cutaneous sensory nerves,fistulaformation, seroma, Frey's
syndrome and trismus (Shaheen, 1997).
FIG. 2 Superficial parotidectomy is a commonly performed
Non-tender swelling in the inferior region of the right parotid surgical procedure. It is surprising that localized extra-
bed. vasation mucoceles have not been reported previously as a
complication of this procedure. The reasons for this may
be due to the fact that most of the saliva-producing
one cm extravasation mucocele (Figure 3). The deep lobe parenchyma of the gland is excised. The deep lobe
of the parotid gland showed features of atrophy and generates only small quantities of saliva which can usually
fibrosis. The patient had no facial weakness post-opera- be phagocytosed before noticeable accumulation (Seifert
tively. et al, 1986). This may also explain why there are reported
On review one month following re-exploration she had cases of mucoceles in the parotid region after rhytidectomy
developed a superficial wound infection secondary to a (Habal, 1978). The saliva-generating capacity of the
retained suture which was removed in clinic. She was parotid gland remains intact and inadvertent trauma to a
placed on oral antibiotics. On subsequent review the major salivary duct results in extravasation of more mucus
wound healed satisfactorily. than can be reabsorbed. An alternative destiny for any
mucus produced by any remaining functioning gland is
leakage from the wound as a salivary fistula or to form a
diffuse 'seroma' which usually resolves with conservative
management and/or repeated aspirations. In the above
patient these processes did not occur, and mucus accumu-
lated and formed an isolated collection resembling a
recurrence.
The recurrence of Warthin's tumour after adequate
excision is rare. In the study by Leverstein et al. (1997) the
reported recurrence was 0 per cent. The causes of
recurrence include inadequate excision due to capsular
dissection, gross spillage or multicentricity (Conley, 1988).
When encountered with a recurrence of a benign tumour
after adequate excision it is important to review the
original histology. On review up to three per cent of the
original histopathological diagnosis can be changed
(Conley, 1988). Conley in 1988 recommended pre-opera-
tive computed tomography (CT) scan and/or magnetic
FIG. 3 resonance imaging (MRI) when investigating recurrent
Histopathological section of the excised extravasation muco- tumours. This assesses the extent of the lesion allowing
cele (H & E; x 50). better surgical planning. In this case the skin overlying the
1094 D. REJALI, R. SIMO, M. SMALL

lesion was thin and the lesion superficial. It was felt that in Conley, J. (1988) Problems with reoperation of the parotid
this case further imaging would not alter the management gland and facial nerve. Otolaryngology - Head and Neck
strategy. Surgery 99: 480-488.
It is difficult to make a prospective diagnosis of parotid Habal, M. B. (1978) Parotid retention cyst as a complication of
cystic lesions. Clinical and radiological assessment is rhytidectomy. Case report. Plastic and Reconstructive
inaccurate. The position of the cysts - usually under the Surgery 61: 290-291.
dense parotid facia - makes physical examination unreli- Ishikawa, T., Yasui, R., Fujito, T. (1991) Large retention cyst
able. Radiology is at present unable to reliably differenti- of the accessory parotid gland associated with a muco-
ate between the secondary fibrotic and atrophic changes epidermoid tumour in the cyst wall. Journal of Oral and
which occur in the salivary tissues surrounding benign cysts Maxillofacial Surgery 49: 884-886.
and malignant tumours associated with cysts (Cohen et ah, Leverstein, H., Van Der Wai, J. E., Tiwari, R. M., Van Der
1984). Fine needle aspiration cytology cannot differentiate Waal, I., Snow, G. B. (1997) Results of the surgical
between cysts in isolation from those which are a part of a management and histopathological evaluation of 88 parotid
neoplasm (Ishikawa et ah, 1991). The safe option as in the Warthin's tumours. Clinical Otolaryngology 22: 500-503.
above case, is to re-explore. Pieterse, A. S., Seymour, A. E. (1981) Parotid cysts. An
analysis of 16 cases and suggested classification. Pathology
Re-exploration has a significantly higher morbidity. The 13: 225-234.
fibrosis and scarring make the dissection more difficult. Richardson, G. S., Clairmont, A. A., Erickson E. R. (1978)
The facial nerve is at increased risk because it can be Cystic lesions of the parotid gland. Plastic and Reconstruc-
difficult to differentiate and separate it from scar tissue. tive Surgery 61: 364-370.
The normal anatomy may also be distorted (Conley, 1988). Seifert, G., Miehlke, A., Haubrich, J., Chilla, R. (1986)
Ideally a surgeon experienced in parotid surgery should Diseases of the Salivary Gland. Georg 'Thieme Verlag,
perform the procedure with the assistance of facial nerve Thieme Inc. Stuttgart, pp 171-180.
monitoring. The patient should be aware of the increased Shaheen, N. A., Harboyan G. T., Nassif, R. I. (1975) Cysts of
risks prior to consent. the parotid gland. Review and report of two unusual cases.
In conclusion we feel that the extravasation mucocele Journal of Laryngology and Otology 89: 435^44.
should be included as a complication of superficial Shaheen, O. H. (1997) Benign salivary gland tumours. In
parotidectomy. These can be difficult to differentiate Scott-Brown's Otolaryngology. 6th Edition. (Kerr, A. G.,
from recurrence of a Warthin's tumour as the cytopatho- Hibbert, J., eds.), Butterworth Heinmann, Oxford, pp 20/17.
logical and radiographic features can be non-specific. Re- Thompson, A. C , Bradley, P. J. (1991) Iatrogenic epidermoid
exploration of the parotid bed is necessary but the higher cyst of the parotid region following ear surgery. Journal of
morbidity should be taken into consideration. Laryngology and Otology 105: 227-228.

References Address for correspondence:


Bhaskar, S. N., Bolden, T. E., Weimann, J. P. (1975) D. Rejali,
Pathogenesis of mucoceles. Journal of Dental Research 35: 30 Frances Avenue,
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