Journal
MEDICINE
Volume 100, Issue 42, Pages -Publisher
LIPPINCOTT WILLIAMS & WILKINS
DOI: 10.1097/MD.0000000000027578
Keywords
anti-vascular endothelial growth factor therapy; bilateral diffuse uveal melanocytic proliferation; combination therapy; photodynamic therapy
Categories
Funding
- Japan Society for the Promotion of Science, Tokyo, Japan [18K09444, 21K09716]
- Grants-in-Aid for Scientific Research [21K09716] Funding Source: KAKEN
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Bilateral diffuse uveal melanocytic proliferation (BDUMP) is a rare retinal exudative disease with no established treatment standard. In 3 cases of BDUMP, combined anti-VEGF therapy and photodynamic therapy may be an effective option for treatment-resistant exudate. Early diagnosis and prompt administration of combination therapy are crucial for better outcomes.
Rationale: Bilateral diffuse uveal melanocytic proliferation (BDUMP) is an extremely rare retinal exudative disease with physical disorders and no established treatment standard. We describe treatment courses in 3 cases of BDUMP. Patients concerns: Three male patients complained active vision loss. One male patient in his 70s (patient 1) was treated with prednisolone, mesalazine, and ciclosporin for hypoplastic anemia and ulcerous colitis. One male patient in his 60s (patient 2) was on prednisolone therapy for adult Still disease. Another male patient in his 70s (patient 3) was on prednisolone therapy for polymyalgia rheumatica, giant cell arteritis, and pancreatic body tumor. Diagnoses: Retinal specialists diagnosed these patients with BDUMP based on characteristic fundus findings of multiple red patches and retinal exudate. Interventions: Two patients (patients 1 and 2) with poor response to anti-vascular endothelial growth factor (VEGF) monotherapy and/or triamcinolone acetonide sub-Tenon injection were treated with combined anti-VEGF therapy and photodynamic therapy. One patient (patient 3) was treated with 3 rounds of monthly anti-VEGF monotherapy. Outcomes: Retinal exudates were resolved in all patients. No recurrence of retinal exudates was observed for at least 10 months, 2 years, or 4 months after the therapy in patients 1, 2, and 3, respectively. However, best-corrected visual acuity of the right eye was low (20/200) compared with that of the left eye (20/22) in patient 2 despite exudate resolution, due to permanent outer retinal damage secondary to long-term retinal exudate. Lessons subsections: Combined anti-VEGF therapy and photodynamic therapy may be a feasible therapeutic option for treatment-resistant exudate in patients with BDUMP. Early diagnosis of BDUMP and prompt administration of combination therapy are crucial.
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