0
We're unable to sign you in at this time. Please try again in a few minutes.
Retry
We were able to sign you in, but your subscription(s) could not be found. Please try again in a few minutes.
Retry
There may be a problem with your account. Please contact the AMA Service Center to resolve this issue.
Contact the AMA Service Center:
Telephone: 1 (800) 262-2350 or 1 (312) 670-7827  *   Email: subscriptions@jamanetwork.com
Error Message ......
Research Letters |

Dacryops of Krause Gland in the Inferior Fornix in a Child FREE

André Jastrzebski, MD; Seymour Brownstein, MD; David R. Jordan, MD; Joseph de Nanassy, MD
[+] Author Affiliations

Author Affiliations: Departments of Pathology (Drs Jastrzebski and Brownstein) and Ophthalmology (Drs Jastrzebski, Brownstein, and Jordan), The Ottawa Hospital and University of Ottawa, and Department of Pathology, Children's Hospital of Eastern Ontario and University of Ottawa (Dr de Nanassy), Ottawa, Ontario, Canada.


Arch Ophthalmol. 2012;130(2):252-254. doi:10.1001/archopthalmol.2011.1581.
Text Size: A A A
Published online

Dacryops of the accessory lacrimal glands are extremely rare, with only 4 previous cases reported to involve Krause glands in the last 60 years.14 Dacryops of Krause glands have not been reported in the inferior fornix. The cause is often unclear, although numerous causes of secondary dacryops are known.14

An otherwise healthy 2-year-old girl had a left lower eyelid mass, noted since age 2 months but enlarging during the previous 6 months. Her ocular and medical histories were unremarkable. Examination revealed a soft, fluctuant mass involving the left lower eyelid (Figure 1A). Computed tomography showed a cystic mass within the left lower eyelid, causing enophthalmos of the left globe (Figure 1B). Surgical dissection revealed a mass within the left inferior fornix, not involving the inferior tarsus. The lesion was excised intact.

Place holder to copy figure label and caption
Graphic Jump Location

Figure 1. Findings in a 2-year-old girl. A, Left lower eyelid swelling (asterisk). B, Computed tomographic scan shows the cystic nature of the lesion (C) extending inferiorly, with enophthalmos of the left globe (G).

Histopathologic examination revealed a cystic structure lined mostly by a double layer of cuboidal epithelium, with focal apical microvillus processes (Figure 2A). The underlying stroma focally contained small islands of normal-appearing lacrimal gland with mild chronic nongranulomatous inflammation (Figure 2B). The diagnosis was dacryops of an accessory lacrimal gland of Krause of the inferior fornix based on its location and the histopathologic findings.

Place holder to copy figure label and caption
Graphic Jump Location

Figure 2. Histopathologic findings (hematoxylin-eosin, original magnification ×400). A, Cystic cavity (asterisk) lined by a double layer of cuboidal epithelium with focal apical microvillus processes (arrows). B, Small islands of lacrimal gland tissue (arrows) and mild chronic nongranulomatous inflammation within the loose fibrous stroma adjacent to the cyst.

The term dacryops was proposed by Schmidt in 1803 and refers to lacrimal ductal cysts of the primary or accessory lacrimal glands.4 Dacryops of the accessory lacrimal glands have been reported only rarely.14 The apical microvillus processes4,5 of these lesions and the nearby presence of glandular acini1,3 are consistent with this diagnosis. Although histologically indistinguishable, Krause glands are anatomically located within the superior and inferior fornices, while Wolfring glands are found adherent or adjacent to the upper and lower tarsal plates.4,5 Wolfring gland dacryops have been reported more frequently than Krause gland dacryops.15 Weatherhead4 noted this disparity in 13 cases of Wolfring gland dacryops diagnosed during a 7-year period but only 1 case of Krause gland dacryops identified in the same period. All previously reported cases of Krause gland dacryops have occurred in the upper eyelids, where Krause glands are more abundant.14

Although originally considered secondary to ductal outlet obstruction, the pathophysiology of dacryops recently has been proposed as a complex process involving periductal inflammation and subsequent passive dilation through a combination of hypersecretion, ductule wall weakening, and impaired neuromuscular contractility.1,2,46 However, the chronic nongranulomatous inflammation, as observed in our case, also may occur secondary to the dacryops. High concentrations of IgA also have been identified in the cystic fluid, and it is postulated that ductal inflammation leads to increased IgA secretion with the resulting osmotic gradient contributing to cyst formation.4

Numerous secondary causes of dacryops have been described, including trachoma, pemphigoid, trauma, and periocular surgery, while infrequently, as in our case, the etiology remains unknown.15 Secondary cases are seen mostly in older children and young adults; idiopathic cases, as in our case, are rarely reported but are thought to occur secondary to a congenital anomaly of the duct or an intrinsic abnormality in the secreted products, possibly related to chronic inflammation.16 The treatment of dacryops, whether originating from the primary or accessory lacrimal glands, is complete excision of the cyst, ideally intact. Incomplete excision and intraoperative cyst rupture are associated with increased rates of recurrence and fistula formation.46

In summary, we report dacryops of an inferior fornix accessory lacrimal gland of Krause. To our knowledge, this is the first reported case of Krause gland dacryops involving the lower eyelid. It remains imperative that all suspicious lesions are examined histopathologically and that the ophthalmologist considers the possibility of a lacrimal ductal cyst, even within the inferior fornix.

Correspondence: Dr Brownstein, Department of Ophthalmology, University of Ottawa Eye Institute, 501 Smyth Rd, Room W6213, Ottawa, ON K1H 8L6, Canada (sbrownstein@ohri.ca).

Financial Disclosure: None reported.

Durán JA, Cuevas J. Cyst of accessory lacrimal gland.  Br J Ophthalmol. 1983;67(7):485-486
PubMed   |  Link to Article
Mathur JS, Mehra KS, Dube B, Nema HV. Retention cyst of the duct of Krause's gland.  Orient Arch Ophthalmol. 1968;6:38-40
Mortada A. Cyst of duct of Krause's gland.  Br J Ophthalmol. 1963;47:375-379
PubMed   |  Link to Article
Weatherhead RG. Wolfring dacryops.  Ophthalmology. 1992;99(10):1575-1581
PubMed
Woo KI, Kim YD. Cyst of accessory lacrimal gland.  Korean J Ophthalmol. 1995;9(2):117-121
PubMed
Bullock JD, Fleishman JA, Rosset JS. Lacrimal ductal cysts.  Ophthalmology. 1986;93(10):1355-1360
PubMed

Figures

Place holder to copy figure label and caption
Graphic Jump Location

Figure 1. Findings in a 2-year-old girl. A, Left lower eyelid swelling (asterisk). B, Computed tomographic scan shows the cystic nature of the lesion (C) extending inferiorly, with enophthalmos of the left globe (G).

Place holder to copy figure label and caption
Graphic Jump Location

Figure 2. Histopathologic findings (hematoxylin-eosin, original magnification ×400). A, Cystic cavity (asterisk) lined by a double layer of cuboidal epithelium with focal apical microvillus processes (arrows). B, Small islands of lacrimal gland tissue (arrows) and mild chronic nongranulomatous inflammation within the loose fibrous stroma adjacent to the cyst.

Tables

References

Durán JA, Cuevas J. Cyst of accessory lacrimal gland.  Br J Ophthalmol. 1983;67(7):485-486
PubMed   |  Link to Article
Mathur JS, Mehra KS, Dube B, Nema HV. Retention cyst of the duct of Krause's gland.  Orient Arch Ophthalmol. 1968;6:38-40
Mortada A. Cyst of duct of Krause's gland.  Br J Ophthalmol. 1963;47:375-379
PubMed   |  Link to Article
Weatherhead RG. Wolfring dacryops.  Ophthalmology. 1992;99(10):1575-1581
PubMed
Woo KI, Kim YD. Cyst of accessory lacrimal gland.  Korean J Ophthalmol. 1995;9(2):117-121
PubMed
Bullock JD, Fleishman JA, Rosset JS. Lacrimal ductal cysts.  Ophthalmology. 1986;93(10):1355-1360
PubMed

Correspondence

CME
Also Meets CME requirements for:
Browse CME for all U.S. States
Accreditation Information
The American Medical Association is accredited by the Accreditation Council for Continuing Medical Education to provide continuing medical education for physicians. The AMA designates this journal-based CME activity for a maximum of 1 AMA PRA Category 1 CreditTM per course. Physicians should claim only the credit commensurate with the extent of their participation in the activity. Physicians who complete the CME course and score at least 80% correct on the quiz are eligible for AMA PRA Category 1 CreditTM.
Note: You must get at least of the answers correct to pass this quiz.
Please click the checkbox indicating that you have read the full article in order to submit your answers.
Your answers have been saved for later.
You have not filled in all the answers to complete this quiz
The following questions were not answered:
Sorry, you have unsuccessfully completed this CME quiz with a score of
The following questions were not answered correctly:
Commitment to Change (optional):
Indicate what change(s) you will implement in your practice, if any, based on this CME course.
Your quiz results:
The filled radio buttons indicate your responses. The preferred responses are highlighted
For CME Course: A Proposed Model for Initial Assessment and Management of Acute Heart Failure Syndromes
Indicate what changes(s) you will implement in your practice, if any, based on this CME course.

Multimedia

Some tools below are only available to our subscribers or users with an online account.

732 Views
4 Citations
×

Related Content

Customize your page view by dragging & repositioning the boxes below.

Articles Related By Topic
Related Collections
PubMed Articles
Jobs
JAMAevidence.com

The Rational Clinical Examination: Evidence-Based Clinical Diagnosis