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Case Reports and Small Case Series
May 2000

Sarcoid Inflammation and Obstruction of the Nasolacrimal System

Arch Ophthalmol. 2000;118(5):719-720. doi:

We saw a patient with a medial canthal lesion and epiphora of the left eye of 18 months' duration. Evaluation led to the diagnosis of sarcoidosis, but not before progressive cardiac involvement occurred, with a fatal outcome.

Report of a Case

A 58-year-old white woman was referred to our service for evaluation of a medial canthal lesion (Figure 1) and tearing of the left eye. Previous unsuccessful treatment by a community ophthalmologist included several months of oral antibiotics, antifungals, and topical corticosteroids, culminating in a left dacryocystorhinostomy. The incision included removal of the cutaneous lesion, which was found to be attached to the lacrimal sac. Biopsy specimens of lacrimal sac mucosa demonstrated noncaseating granulomas with epithelioid histiocytes (Figure 2). Three weeks postoperatively, the cutaneous lesion recurred and enlarged.

Figure 1.
A diffusely elevated, erythematous lesion is located over the left lacrimal sac.

A diffusely elevated, erythematous lesion is located over the left lacrimal sac.

Figure 2.
Photomicrograph demonstrates the presence of noncaseating granulomas with epithelioid histiocytes and multinucleated giant cells within lacrimal sac mucosa (hematoxylin-eosin, original magnification ×100).

Photomicrograph demonstrates the presence of noncaseating granulomas with epithelioid histiocytes and multinucleated giant cells within lacrimal sac mucosa (hematoxylin-eosin, original magnification ×100).

Her medical history was significant for an unspecified cardiac dysrhythmia treated with digoxin. Examination disclosed a 10×10-mm broadly elevated, erythematous mass with firm nodular components over the lacrimal sac. Nasal mucosa appeared normal.

Within 2 weeks of presentation, the patient was admitted to another hospital with atrial fibrillation. We recommended a serum angiotensin-converting enzyme determination and a gallium scan, which confirmed the diagnosis of sarcoidosis. During cardioversion, an embolus created a middle cerebral artery occlusion, which was fatal.

Comment

Since a review of cases of histologically verified sarcoidosis of the lacrimal sac in 1981,1 similar descriptions have appeared in the literature.2 In each case, it has been postulated that granulomatous inflammation of lacrimal tract mucosa, contiguous with similar infiltration of nasal mucosa, led to obstruction of outflow. Clinically significant blockage occurs in approximately 1.8% of patients with sarcoidosis.3

The differential diagnosis for granulomatous inflammation of the nasolacrimal system includes atypical mycobacterial infection, fungal infection, and Wegener granulomatosis. Several features of this case suggested a diagnosis other than sarcoidosis. Respiratory or ocular symptoms were absent. The medial canthal lesion was solitary; cutaneous involvement in sarcoidosis, while occurring in 9% to 37% of patients,4 more typically consists of erythema nodosum or clusters of small subcutaneous nodules. Nasal mucosal examination did not disclose the diffusely grayish appearance characteristic of sarcoid infiltration.

Patients with sarcoid-related obstruction of lacrimal outflow are at increased risk for failure after dacryocystorhinostomy. Of the 13 patients with histologically verified nasolacrimal system sarcoidosis reported in the literature, 9 had a total of 13 primary dacrocystorhinostomies. Four of the 13 surgeries resulted in failure. Although follow-up intervals and surgical technique (including placement of lacrimal stents) varied, the high failure rate suggests the presence of ongoing inflammation at the site ofnasolacrimal anastomosis. In the absolacrimal anastomosis. In the absence of long-term steroid administration, stents should probably be maintained for prolonged intervals.

Chronic cardiac dysrhythmia led to the patient's death 18 months after the onset of her medial canthal lesion; 5% of patients with sarcoidosis have life-threatening cardiac dysfunction secondary to granulomatous inflammation, including dysrhythmias. Prompt diagnosis of sarcoid inflammation and obstruction of the nasolacrimal system may lead to earlier detection and management of the vision- and life-threatening complications of sarcoidosis.

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Article Information

Corresponding author: George H. Garcia, MD, 925 N 87th St, Milwaukee, WI 53226.

References
1.
Harris  GJWilliams  GAClarke  GP Sarcoidosis of the lacrimal sac. Arch Ophthalmol. 1981;991198- 1201Article
2.
Vasquez  RJLinberg  JVMcCormick  SA Histopathology of nasolacrimal duct obstruction compatible with localized sarcoidosis. Ophthalmic Plast Reconstr Surg. 1988;4147- 151Article
3.
Karma  A Sarcoidosis of the lacrimal sac [letter]. Arch Ophthalmol. 1982;100664Article
4.
Mana  JMarcoval  JGraells  JSalazar  APeyri  JPujol  R Cutaneous involvement in sarcoidosis: relationship to systemic disease. Arch Dermatol. 1997;133882- 888Article
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