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New Clinicopathologic Findings in 4 Cases

Kimberly Peele Cockerham, MD; Ahmed A. Hidayat, MD; Glenn C. Cockerham, MD; Mark H. Depper, MD; Scott Sorensen, MD; Albert S. Cytryn, MD; Paul T. Gavaris, MD

Arch Ophthalmol. 2000;118:227-232.

Objective  To define the clinicopathologic features of eyelid involvement in Melkersson-Rosenthal syndrome (MRS).

Methods  Four patients with eyelid edema consistent with MRS were evaluated clinically, including diagnostic imaging in 2 patients. Eyelid tissue from these patients was examined by light microscopy and immunohistochemistry. Polymerase chain reaction for herpes simplex virus was performed in 1 case.

Results  The 3 men and 1 woman ranged in age from 33 to 74 years. All patients had insidious, painless, nonpitting eyelid edema. Three patients had unilateral edema; one had bilateral, asymmetric involvement. Ipsilateral lip edema was present in 1 case. Computed tomography demonstrated periorbital heterogeneous thickening that corresponded to the microscopic finding of scattered granulomas. All 4 patients demonstrated epithelioid granulomas inside and adjacent to dilated lymphatic vessels. Polymerase chain reaction testing was negative for herpes simplex virus.

Conclusions  Isolated eyelid swelling that mimics thyroid-associated ophthalmopathy may occur in MRS. Computed tomography may be useful in the diagnosis. Biopsy should be performed in all cases of unexplained nonpitting eyelid edema. In the eyelid, MRS is characterized histopathologically by a granulomatous lymphangitis, a finding that seems to be unique to this condition.

From Allegheny Orbital and Ophthalmology Associates, Pittsburgh, Pa (Dr K. P. Cockerham); Department of Ophthalmic Pathology, Armed Forces Institute of Pathology, Washington, DC (Dr Hidayat); Ophthalmology Service, Andrews Air Force Base, Maryland (Dr G. C. Cockerham); Department of Radiology (Dr Depper) and the Ophthalmology Service (Dr Sorensen), Walter Reed Army Medical Center, Ophthalmic Plastic and Orbital Consultants (Dr Cytryn), and Eye Associates of Washington (Dr Gavaris), Washington, DC.

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