• Dong Hee Ha, Hee Sung Kim, and Jeong Kyu Lee.
• Department of Ophthalmology, College of Medicine, Chung-Ang University Hospital, Seoul, South Korea.
• Medicine (Baltimore). 2023 Nov 10; 102 (45): e36035e36035.

RationaleDiagnosing intralymphatic histiocytosis can be challenging due to its rarity. We present a case of intralymphatic histiocytosis in the upper eyelid of a Korean patient. We treated the condition by surgical debulking and intralesional triamcinolone injection.Patient ConcernsA 59-year-old man was referred to our clinic with a 7-year history of unilateral swelling in the right upper eyelid. He had previously been treated with long-term oral steroids and immunosuppressants, but his eyelid swelling persisted. Unilaterally non-pitting erythematous edema was localized on the right upper eyelid without any itching or pain. His best corrected visual acuity at presentation was 20/20 for both eyes. Enhanced orbital computerized tomography revealed edematous soft tissue thickening in the right upper eyelid. In the laboratory testing, the erythrocyte sedimentation rate showed an increase of 19, and the antinuclear antibody titer was positive with a homogeneous pattern.DiagnosesWe diagnosed the patient with intralymphatic histiocytosis based on the histopathological findings.InterventionWe attempted surgical debulking and biopsy on the right upper eyelid due to the persistent symptoms and the absence of a definitive diagnosis.OutcomesThe patient has demonstrated significant improvement after receiving an intralesional triamcinolone injection in the right upper eyelid following the surgery and is currently under follow-up with no signs of recurrence.LessonOphthalmologists should consider intralymphatic histiocytosis in cases of persistent eyelid swelling that do not respond to treatment, even in Asian patients. Surgical debulking and intralesional triamcinolone injections may be beneficial for improvement.Copyright © 2023 the Author(s). Published by Wolters Kluwer Health, Inc.

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