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Am. J. Trop. Med. Hyg., 102(1), 2020, pp. 78 doi:10.4269/ajtmh.19-0539

Copyright © 2020 by The American Society of Tropical Medicine and Hygiene

Images in Clinical Tropical Medicine Intraocular Infiltration

Koju Kamoi* and Kyoko Ohno-Matsui

Department of Ophthalmology and Visual Science, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University, Tokyo, Japan

A 58-year-old man presented with general malaise and showed positive results for human T-cell leukemia virus type 1 (HTLV-1) infection. As blood tests revealed abnor- mal leukocytes and flower cells, smoldering-type adult T-cell leukemia/lymphoma (ATL) was diagnosed. After 7 years, the condition changed to acute-type ATL. Blood tests identied an increase in CD4 from 995 cells/μL to 1,256 cells/μL and monoclonal integration of HTLV-1 pro- virus into tumor cells. Treatment was started with chemo- therapy and allogeneic bone marrow transplantation.

During follow-up, he was referred to the ophthalmology

department after experiencing sudden visual loss in the right eye.

Slit-lamp examination revealed cellular inltration in the anterior chamber and vitreous humor. Fundus examination showed yellowish-white inltrative foci associated with protrusions in the retina. Optical coherence tomography revealed massive solid inltrative foci below the retinal pigment epithelium (Figure 1). As differential diagnoses for co-infections of the eye, our recent nationwide survey revealed cytomegalovirus (CMV) as a major cause of op- portunistic infection in the eye of ATL patients, followed by herpesvirus and toxoplasma.1In addition, co-infections of the eye in immunocompetent patients are not frequently conrmed using serologic diagnostic methods.2Multiplex polymerase chain reaction (PCR) and broad-range PCR of a sample of aqueous humor, therefore, ruled out viral (human simplex virus-1 and 2, varicella zoster virus, EpsteinBarr virus, CMV, and human herpesvirus 6–8), toxoplasma, tu- berculosis, syphilis, and bacterial and fungal infections, ruling out opportunistic infection and allowing the diagnosis

FIGURE1. Yellowish-white inltrative foci associated with protru- sions in the retina (uppergure). Optical coherence tomography re- veals massive solid inltrative foci below the retinal pigment epithelium (lowergure). White line represents scan line. Thisgure appears in color at www.ajtmh.org.

FIGURE2. Inltration is reduced 6 months after vitreous injection of methotrexate concomitant with radiotherapy. Thisgure appears in color at www.ajtmh.org.

* Address correspondence to Koju Kamoi, Department of Ophthal- mology and Visual Science, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University, 1-5-45 Yushima, Bunkyo-ku, Tokyo 113-8519, Japan. E-mail: [email protected]

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of intraocular leukemic cell inltration.3Elevated lesions on the retina improved after two vitreous injections of 400μg/

0.1 mL of methotrexate and subsided withve sessions of 2-Gy radiotherapy. Corresponding to this combination therapy, retinal lesions gradually resolved within 6 months (Figure 2).

Globally, HTLV-1 infection is now the focus of attention, since the discovery that more than 40% of adults from Ab- original communities in central Australia are infected with HTLV-1.4This infection causes ATL and also visual impair- ment by ATL-related ocular manifestations.5As HTLV-1 associated ATL is a neoplasia characterized by massive invasion of leukemic cells into various organs, physicians should keep in mind that intraocular inltration is the most frequent manifestation observed among HTLV-1–associated ATL patients.1

Received July 21, 2019. Accepted for publication September 26, 2019.

Authors addresses: Koju Kamoi and Kyoko Ohno-Matsui, Department of Ophthalmology and Visual Science, Graduate

School of Medical and Dental Sciences, Tokyo Medical and Dental University, Tokyo, Japan, E-mails: [email protected] and [email protected].

This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.

REFERENCES

1. Kamoi K, Okayama A, Izumo S, Hamaguchi I, Uchimaru K, Tojo A, Ohno-Matsui K, 2018. Adult T-cell leukemia/lymphoma-related ocular manifestations: analysis of therst large-scale nation- wide survey.Front Microbiol 9:3240.

2. de-la-Torre A et al, 2019. Coinfections and differential diagnosis in immunocompetent patients with uveitis of infectious origin.

BMC Infect Dis 19:91.

3. Mochizuki M, Sugita S, Kamoi K, Takase H, 2017. A new era of uveitis: impact of polymerase chain reaction in intraocular in- ammatory diseases.Jpn J Ophthalmol 61:120.

4. Martin F, Tagaya Y, Gallo R, 2018. Time to eradicate HTLV-1: an open letter to WHO.Lancet 391:18931894.

5. Kamoi K, Mochizuki M, 2012. HTLV infection and the eye.Curr Opin Ophthalmol 23:557561.

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