AJR 2005; 184:321-323
© American Roentgen Ray Society
Bilateral Dacryops: Correlation of Clinical, Radiologic, and Histopathologic Features
Apostolos John Tsiouris1,
Monica Deshmukh2,
Pina C. Sanelli1 and
Brian G. Brazzo3
1 Department of Radiology, New York Presbyterian Hospital, Weill Medical College
of Cornell University, 520 E 70th St., Starr Pavilion 630, New York, NY
10021.
2 University of Medicine and Dentistry of New Jersey, Robert Wood Johnson
Medical School, Camden, NJ 08103.
3 Department of Ophthalmology, New York Presbyterian Hospital, Weill Medical
College of Cornell University, New York, NY 10021.
Received January 15, 2004;
accepted after revision March 15, 2004.
Address correspondence to A. J. Tsiouris
(apt9001{at}med.cornell.edu).
Introduction
We report a case of bilateral lacrimal gland enlargement due to dacryops.
Dedicated contrast-enhanced CT of the orbits showed bilateral peripherally
enhancing cystic structures that were contiguous with the palpebral lobes of
the lacrimal glands. Histopathology confirmed the diagnosis of bilateral
dacryops, an extremely rare clinical entity and even rarer bilaterally.
Tears are produced by the lacrimal and accessory lacrimal glands and drain
through the lacrimal ducts. Their function is to keep the ocular surface moist
and clear of debris and bacteria. The major lacrimal glands are located at the
anterior superolateral aspects of the orbits, adjacent to the tendons of the
superior and lateral rectus muscles. The gland is separated into two lobes:
the orbital and the palpebral
[1]. Loss of lacrimal ductule
contractility combined with distention of the duct wall results in a condition
called "dacryops"
[2]. Dacryops refers to a cyst
of the lacrimal gland and can develop anywhere the lacrimal tissue is present.
Cysts develop insidiously and present as painless masses on the eyelids. Their
clinical presentation is rare, and their bilateral presentation is encountered
even less frequently. We present the clinical, histopathologic, and CT
appearance of bilateral dacryops as a unique case.
Case Report
A 48-year-old Korean American woman was referred to the oculoplastics
department at our institution with bilateral eyelid swelling. The swelling had
been present for the past 5 years but had progressed over the past year. She
had no associated pain, erythema, or discharge. She denied any history of
orbital trauma or infection.
The patient's visual acuity was 20/20 right and left with corrective eye
glasses. Findings from an external examination were normal: no inflammation,
masses, or proptosis. The findings of the ocular motility examination was
normal. At eyelid eversion examination, small cystic masses were identified on
the anterior aspect of the palpebral lobe of the lacrimal glands. The cysts
appeared smooth and translucent and were attached to the underlying
conjunctiva of the globe and eyelids bilaterally. No evidence of lacrimal
gland inflammation was detected.
Axial and direct coronal CT examination of the orbits with sagittal oblique
reformations was performed after the administration of nonionic IV contrast
material. CT showed bilateral cystic lesions contiguous with the inferior
palpebral lobe of the lacrimal glands. The right cyst measured 10 mm in
diameter; the left cyst was slightly smaller, measuring 8 mm in diameter. The
right and left cysts measured 9 H and 7 H, respectively, which is consistent
with fluid density. The cysts showed thin peripheral enhancement without
nodular or irregular enhancement. The globes and the remaining lacrimal gland
tissue were normal bilaterally. No associated periorbital or soft-tissue
inflammation was present.
The patient was admitted for biopsy of both lacrimal glands via lid
incision, which was conducted while the patient was under sedation. Histologic
examination established the diagnosis of bilateral dacryops. These findings
showed that both masses had large cystic spaces lined by benign columnar
epithelium of lacrimal duct type.
Discussion
The major lacrimal glandular tissue is located in the anterior
superolateral region of the orbit and is responsible for tear production and
secretion. Obstruction of the lacrimal gland ductules leads to the formation
of dacryops, ductal cysts of the lacrimal glands. Dacryops is a rare clinical
phenomenon, most commonly presenting unilaterally in the palpebral lobe.
Patients frequently complain of painless swelling in the lateral portion of
the upper eyelid. A history of trauma or inflammation of the conjunctiva or a
congenital anomaly of the excretory duct can be the precipitating factor to
cyst formation [3].
The cysts appear as smooth-walled, bluish gray areas in the superolateral
region of the orbit on gross inspection. Histologically, lacrimal ductal cysts
are composed of two layers of cells: an inner cuboidal layer containing goblet
cells surrounded by an outer myoepithelial layer. Fibrotic tissue, often
containing lymphocytes, also can be present in the histologic specimen. The
cysts usually are surrounded by lacrimal glandular tissue and dilated lacrimal
gland ducts [4].
The pathogenesis behind cyst formation is related to periductal or
conjunctival inflammation or trauma, which results in lacrimal gland
hypersecretion and damage to ductule walls and secondary ductule obstruction.
The damaged walls then passively dilate and form cysts
[2]. Cysts generally grow
slowly but can display rapid growth at times, especially in the setting of
superimposed infection. Cyst growth eventually can lead to other symptoms,
including proptosis, and to mass effect with eyelid and globe displacement
[5].
CT findings for dacryops have been reported in the literature
[6,
7]. Contrast-enhanced CT shows
masses with fluid density in the region of the lacrimal glands, lateral to the
globes. There is no evidence of associated bone abnormality
[6,
7]. CT evaluation can reveal
the attenuation values and enhancement of the cysts. Although there are no CT
attenuation values specific for dacryops, the literature has reported fluid
and soft-tissue densities for these cysts
[3,
4]. In our patient, CT
examination (field of view, 15 cm; 120 kVp; 230 mA; 1.3-mm slice thickness;
matrix, 512 x 512; axial and direct coronal imaging with sagittal
reformations) showed bilateral fluid-density masses located in the palpebral
lobe of the lacrimal glands. No evidence of bone erosion or adjacent
inflammation was detected (Figs.
1A and
1B
).
View larger version (85K):
[in this window]
[in a new window]
[as a PowerPoint slide]
|
Fig. 1A. —48-year-old Korean American woman who presented with
complaints of worsening bilateral eyelid swelling. Axial contrast-enhanced CT
scan of orbits shows peripherally enhancing cystic lesions (arrows)
along lateral aspects of globes bilaterally.
|
|
View larger version (110K):
[in this window]
[in a new window]
[as a PowerPoint slide]
|
Fig. 1B. —48-year-old Korean American woman who presented with
complaints of worsening bilateral eyelid swelling. Coronal contrast-enhanced
CT scan of orbits shows relationship of cysts (arrows) with inferior
margin of lacrimal glands.
|
|
View larger version (92K):
[in this window]
[in a new window]
[as a PowerPoint slide]
|
Fig. 1C. —48-year-old Korean American woman who presented with
complaints of worsening bilateral eyelid swelling. Photograph of gross
specimen shows small cystic mass (arrow) at lateral aspect of
orbit.
|
|
View larger version (150K):
[in this window]
[in a new window]
[as a PowerPoint slide]
|
Fig. 1D. —48-year-old Korean American woman who presented with
complaints of worsening bilateral eyelid swelling. Photomicrograph shows
cystic space (arrowheads) lined by columnar epithelium. (H and E)
|
|
The bilateral nature of our patient's presentation suggests that systemic
diseases also should be considered. Differential considerations for bilateral
lacrimal gland disease include Sjögren's syndrome, Mikulicz's disease,
sarcoidosis [8], and
lymphoproliferative diseases. These entities may present with nonspecific
diffuse enlargement of the lacrimal glands. However, lymphoma may show
discrete nodular masses. Additional orbital cystic lesions may be considered,
such as dermoid cysts, posttraumatic hemorrhagic cysts, conjunctival cysts,
adenoid cystic carcinomas, and abscesses. Overall, the diagnosis relies on
biopsy results and the patient's clinical history. In our patient, the
bilaterality, fluid attenuation of the lesions, lack of internal enhancement,
and associated inflammation are not typical of these entities. Therefore,
other possibilities were considered such as dacryops because of its classic
location in the orbit. Even though these lesions often are precipitated by
previous traumatic injury or infection, the patient may not recall a subtle
inciting event or these lesions may be an idiopathic occurrence.
In summary, dacryops, or a lacrimal gland ductal cyst, is a rare entity in
clinical medicine, and bilateral presentation is even more unusual. However,
this entity does occur, and it is important to understand its clinical,
histologic, and radiologic appearance. Clinicians and radiologists should be
aware of the presentation of dacryops and the possibility of its bilaterality,
so they will include this entity in the differential diagnosis of lacrimal
gland masses.
References
- Newell FW. Lacrimal apparatus. In: Ophthalmology:
principles and concepts, 6th ed. St. Louis, MO: Mosby,1986
: 254
- Duke-Elder S, MacFaul PA. Systems of ophthalmology
series: the ocular adnexa, vol. 13. St.
Louis, MO: Mosby, 1974:638
-642
- Bullock JD, Fleishman JA, Rosset JS. Lacrimal ductal cysts.
Ophthalmology1986; 93:1355
-1360[Medline]
- Smith S, Rootman J. Lacrimal ductal cysts: presentation and
management. Surv Ophthalmol1986; 30:245
-250[Medline]
- Hornblass A, Herschorn BJ. Lacrimal gland duct cysts.
Ophthalmic Surg1985; 16:301
-306[Medline]
- Bradey N, Hayward JM. Bilateral lacrimal gland enlargement: an
unusual manifestation of dacryops. Clin Radiol1991; 43:280
-281[Medline]
- Khoury N, Haddad MC, Tawil A. Ductal cysts of the accessory
lacrimal glands: CT findings. AJNR1999; 20:1140
-1142[Abstract/Free Full Text]
- Sacher M, Lanzieri CF, Sobel LI, Som PM. Computed tomography of
bilateral lacrimal gland sarcoidosis. J Comput Assist
Tomogr 1984;8:213
-215[Medline]
CiteULike 
Complore 
Connotea 
Del.icio.us 
Digg 
Reddit 
Technorati What's this?
Top
Introduction
Case Report
