The Iranian Journal of

Otorhinolaryngology
Vol.17, No.41,Autumn-2005

1384

Recurrence of Lacrimal Gland Pleomorphic Adenoma

(Two case report with review of literature)
1

Etezad Razavi M.MD,2Saber MoghaddamA.MD, 3KargozarA.MD,

4
Sharifi N.MD,5Yazdani A.MD
Associated Professor of Ophthalmology,4Assistant Professor of Pathology,
Assistant Professor of Ophthalmology-Mashhad University of Medical Sciences

1,2,3

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Abstract

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Introduction: In cases with lacrimal gland mixed tumor preoperative delicate clinical and radiological
diagnosis lead to proper surgical approach. Incomplete resection of lacrimal gland mixed tumor may
be complicated by severe tumor recurrence with the risk of malignant transformation.
In this case report, we present 39 years old man with history of transcranial excision of lacrimal gland
tumor. Six months before admission progressive proptosis and inferomedial displacement of right
globe (approximately 3 cm) has been developed. Soft tissue hypertrophy of eyelids and corneal
leukoma developed due to the chronic progressive course of proptosis with chronic corneal exposure.
The other case also was a 38-year-old man with significant proptosis and history of two time lacrimal
gland tumor excision, one from transcranial approach.
The surgical procedure was performed in both cases through anterolateral orbitotomy without bone
removal and the whole lesions were removed with pseudo capsule.
The vision of the first patient improved from hand motion preoperatively to one meter finger count
after surgery, and there was no recurrence in both cases 6 months postoperatively.
In general, regarding risk of malignant degeneration and recurrence after incomplete excision or
incisional biopsy of lacrimal gland mixed tumor, it is strongly recommended to perform complete
excision with psudocapsule in the first surgery.
Key words: Pleomorphic adenoma, Lacrimal gland, Tumor recurrence, Benign Mixed Tumor

Introduction

linically, the large majority of lacrimal

gland masses represent as idiopathic
inflammatory disease (dacryadenitis), which
usually responds to anti-inflammatory
medication and does not require surgical
intervention and biopsy.
Of those lacrimal gland tumefactions which
do not present with inflammatory signs and
symptoms, approximately half will represent
lymphoproliferative disorders, and the other
half are epithelial neoplasm's.

Etezad Razavi M.MD

Address: Mashhad University of Medical Sciences.
Khatamolanbia Eye Hospital
Acceptation date: 81/10/22 Confirmation date: 82/6/16

Page 9-14

Computed tomography scanning is very

helpful in evaluating lesions in the lacrimal
gland region.
CT contour analysis can be used to
differentiate inflammatory conditions and
lymphoid proliferations from frank larimal
gland neoplasm's. Inflammatory and
lymphoid proliferations within the lacrimal
gland tend to cause it to expand diffusely,
making it appear elongated, whereas
neoplasm's appearing as isolated globular
masses, tend to displace and indent
the globe (1).

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The Iranian Journal of Otorhinolaryngology

No.41, Autumn-2005
Examination of ocular motility showed
significant limitation of ductions of the right
eye in upward and outward rotation.
In computerized tomography, a large
multilobulated lesion with involvement of
superolateral region of right orbit can be
seen. The tumor diameter was approximately
4 cm. defect of orbital roof bone noted on
CT and heterogeneous radiodensity with
area of calcification are visible. The globe
displaced inferiorly about 3cm that
presented on CT and there is no significant
sign of any bone erosion. (Fig 2 a&b)

Fig. 2A: Axial View

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Approximately 50% of epithelial tumors are

benign
mixed
tumors
(pleomorphic
adenoma), and about 50% are carcinomas.
In this report, we introduce recurrence of
two cases of benign mixed tumor after
incomplete resection by neurosurgical
subfrontal craniotomy approach. Both of
them operated with lateral orbitotomy
approach and complete resection performed
with no sign of recurrence after six months.
Case report
The first case was a 39-years-old man who
presented to our clinic with a huge
superlateral orbital mass with rapid
progression during the last sixth months.
The patient had a history of lacrimal gland
tumor resection from the orbital roof
(by Transcranial approach), ten years ago in
the neurosurgery setting.
Recently the mass progressively enlarged
with displacement of globe inward and
downward.In external eye examination, a
large nodular mass appeared in the
superolateral aspect of the right orbit (in the
lacrimal gland region) displacing the globe
nasally and inferiorly, accompanied by axial
proptosis.Significant protrusion of the right
eye with lagophthalmos and incomplete
closure of eyelids led to corneal ulcer and
corneal opacity (leukoma) (Fig 1a).

Fig.2B: Coronal View

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Fig 2: Computerized Tomographic Scan of the

first case

Regarding to longavity and chronic course

with progressive growth of the tumor after
primary neurosurgical removal, and also the
lesion was painless and CT scan shows no
bone erosion with multilobulated pattern, we
highly suspected to recurrence of
plaomorphic adenoma.
So, complete resection of the tumor was
done with psuedocapsule performed through
the anterolateral orbitotomy incision.
The lesion extended from behined and
superior of globe to the orbital apex. After
removal the size of the tumor was 3x4x4 cm
with multilobulated pattern on the surface.

A: preoperative
Fig.1: Photographs of the first case with
recurrence of pleomorphic adenoma of lacrimal
gland

Visual acuity of the right eye was perception

of hand movement and for the left eye was
20/20. Significant enlargement of the right
upper lid and lower lid was related to the
chronic and prolonged course of the disorder
(Localized Gigantism).
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Recurrence of Lacrimal Gland Pleomorphic Adenoma

Etezad Razavi M, and

(Fig 3: a & b).

pleomorphic

Fig.4:Light
microscopy
of
adenoma (H&E stain, mag 100x)

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In serial section there was no neural or

vascular invasion or mitosis and necrosis
related to malignancy(Fig5).

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Fig.3:Intraoperative view of huge lacrimal gland

tumor.

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After complete tumor excision the visual

acuity improved from hand motion to one
meter counting finger. The eyelids laxity and
lateral canthus displacement reconstructed
with another surgery (Fig 1b).

B: postoperative

The entire lesion was sent for

histopathologic exam, and the report was
compatible with pleomorphic adenoma of
the lacrimal gland. Light microscopic exam
with hematoxilineosin stain revealed
epithelial elements with round monomorphus nucleus without mitosis mesenchymal
elements with hyalinzed mixoid stroma with
cartilageneous and osteoid differentiation
and degenerated bone particles were also
reported (Fig 4).
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Fig.5: Light microscopy of

adenoma (H&E stain, mag 400x)

pleomorphic

All of the histopathologic findings corresponded to recurrence of pleomorphic adenoma

of the lacrimal gland after incomplete tumor
removal.
The second case was a 38-years-old man
that came to us with complaints of proptosis
and infra displacement of the left eye from a
few months prior to his visit. He had a
history of two times surgery for lacrimal
gland tumor, one from the transcranial
approach. Magnetic resonance imaging
revealed a large mass extending superior and
behind the globe up to orbital apex.
Our surgical approach was the same as the
first case and after complete removal
pathologic exam confirmed recurrence of
pleomorphic adenoma of lacrimal gland.
The patient has no sign of recurrence after
six months.

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The Iranian Journal of Otorhinolaryngology

No.41, Autumn-2005
epithelial lacrimal gland tumors, primary
intervention should consist of complete
removal of the tumor without a preliminary
biopsy (2, 3,4).
Incisional biopsy of the lesion and violation
of the capsule of the pelomorphic adenoma
increased recurrence rate and risk of
malignant degeneration (5).
Regarding anatomical location of lacrimal
gland in superolateral region of the orbit
beneath the superior orbital rim, in spite of
extension of large tumors to orbital apex,
preferred surgical approach is though
anterolateral
orbitotomies.
When
a
pleomorphic adenoma is suspected on the
basis
of
clinical
and
radiological
appearances,the tumor with a rim of
surrounding normal tissues should be
removed without prior biopsy. This ensures
excision of any nodules extending outside
the main tumor; recurrence following
incomplete removal takes the form of
multiple scattered nodules (5,6,7,8).
Although in our cases the surfaces of lesions
are also multilobulated, histologically the
tumor is biphasic, with epithelial and
stromal elements.The epithelial portion is
composed of small ductules with an inner
cuboidal to columnar layer and an outer
spindle-shaped layer that often contains
clear cells. Squamous metaplasia may be
present.The outer layers show a gradual
transition to mesenchymal tissues, which
may display mioxoid, cartilaginous, bony or
adipose features (9).
Ultra structural examination has shown that
ductular cells in pleomorphic adenoma have
the characteristic of ductular cells of the
normal, and that the stromal cells retain
epithelial features in the from of
tonifilaments and desmosomes, although
occasional cells show myoepithelial features
with this filaments and dense bodies
(7,8,9,10).
In our two cases also, surgery was
performed through the lateral orbitotomy
with complete resection of tumor.

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Discussion
Patients with lacrimal gland benign mixed
tumor present with a progressive painless
downward and inward displacement of the
globe with axial proptosis. Symptoms are
usually present for more than 12 months.
A firm lobular mass may be palpated near
the superior lateral orbital rim, and orbital
imaging often reveals enlargement or
expansion of the lacrimal fossa. On CT Scan
the lesion appears well circumscribed but
may have a slightly nodular configuration.
Microscopic examination shows varied
cellular structure consisting primarily of
proliferations of benign epithelial cells and a
steroma composed of spindle-shaped cells
with occasional cartilaginous, mucinous, or
even osteoid degeneration or metaplasia.
This variability accounts for the mixed
tumor characterization of this lesion.
A pseudocapsule circumscribes the lesion,
but microscopic nodular extensions into the
pseudocapsule account for the tendency of
the lesion to recur if an appropriate margin
of surrounding orbital tissue is not also
removed at the time of excision.
Treatment should consist of complete
removal of the tumor with its pseudocapsule
and a surrounding margin of orbital tissue
without a preliminary biopsy.
If the capsule of the pleomorphic adenoma is
incised for direct biopsy, there is a 32% rate
of recurrence, and these recurrences have a
significant risk of malignant degeneration.
Analysis of the clinical presentation of the
lesion and its CT contour helps the surgeon
determine if the lesion is likely to be a
benign mixed tumor, in which case lateral
orbitotomy
is required for complete
excision as the initial approach.There are
confidential research results that show
incomplete resection of epithelial lacrimal
gland tumors and incisional biopsy of these
lesions is hazardous for patients (1).
If there are sufficient clinical and
radiological criteria leading to encapsulated

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Recurrence of Lacrimal Gland Pleomorphic Adenoma

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Otorhinolaryngol Relat Spec 2003 SepOct;65(5):295-9.

8- Tsunoda S, Yabuno T. Pleomorphic
adenoma of the lacrimal gland manifesting
as exophthalmos in adolescence case
report.Neurol Med Chir (Tokyo) 1994
Dec;34(12):814-6.
9- Auran J, Jakobiec FA, Kerbs W. Benign
mixed tumor of the palpebral lobe of the
lacrimal gland. Ophthalmology 1988;95:90 .
10- Dardick I, Van nastrand A WP. Jeans
MTD et al. Pleomorphic adenoma. 1:
ultrastructural organization of epithelial
regions. 2: ultra structural organization of
stromalregions. Hum pathol 1983;14: 780.
11- Henderson j. Orbital tumors. PhiladelphIa :WB saunders, 1973. P. 402-442.
12-Jones IS. Surgical Consideration in the
management of lacrimal gland tumors.Clin
plast surg 1978;5:561.
13-Wright JE. Surgical exploration of the
orbit in Stewart WB (ed):Ophtalmic plastic
Reconstructive
surgery.
Sanfransisco:
American Academy of Ophthalmology
;1984.
14- Sadick H, Riedel F. Benign mixed tumor
of the lacrimal gland.Clinical diagnosis and
surgical management. ORL J Otorhinolaryn
gol Relat Spec 2003 Sep-Oct;65(5):295-9.
15- Fichter N, Schittkowski M, Guthoff
RF. Diseases of the lacrimal gland
Ophthalmologe 2005 Apr;102(4):399-423;
quiz 424-5.
16- Ohtsuka K, Hashimoto M, Suzuki Y. A
review of 244 orbital tumors in Japanese
patients during a 21-year period: origins and
locations. Jpn J Ophthalmol 2005 JanFeb;49(1):49-55.

References

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of

Essentially, in all orbital lesions in which the

orbital apex and osseous optic canal were
involved, the transcranial approach needed
to unroof the canal and totally resected the
tumor.Otherwise in all orbital lesions limited
to the orbital cavity various methods of
anterolateral orbitotomies may give the best
results.
Conclusion
The crucial aspect of management of all
lacrimal fossa tumors is to suspect
pleomorphic adenoma. Although this tumor
is histologically benign, incomplete excision
will likely result in relentless recurrence and
even malignant transformation.
Therefore, when pleomorphic adenoma is
suspected, a lateral orbitotomy is mandatory.
The entire tumor with its pseudocapsule,
surrounding levator aponeurosis, conjunctiva
and periorbita must be excised enbloc
(11,12,13,14,15,16) to avoid recurrences and
long-term misery for patients.

Etezad Razavi M, and

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1- Academy of Ophthalmology: Basic and

Clinical Science course. Section 7.2000.
P.72-73.
2- Albert and Jakobiec. Principle and
practice of ophthalmology. Vol3,4. 1998. P.
1956, 2349t,1955-1956.
3- Ostrosky A, Klurfan FJ et al. Pleomorphic
adenoma of the lacrimal gland.Case
report.Med Oral Patol Oral Cir Bucal 2005
Jan-Feb;10(1):88-9; 86-8.
4- Marshall AF, White DR, Shockley WW.
Pleomorphic adenoma in the palpebral lobe
of the lacrimal gland. Otolaryngol Head
Neck Surg 2005 Jan; 132(1):141-3.
5-Wright JE. Factors affecting the survival
of patients with lacrimal gland tumors. Can J
Ophthalmol 1983;17:3.
6- Miyazaki T, Yamasaki T et al. Unusual
progression of pleomorphic adenoma of the
lacrimal gland: case report. Neurol Med Chir
(Tokyo) 2005 Aug; 45(8):407-1.
7- Sadick H, Riedel F et al. Benign mixed
tumor of the lacrimal gland. Clinical
diagnosis and surgical management. ORL J

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No.41, Autumn-2005

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