{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,12]],"date-time":"2026-06-12T04:58:56Z","timestamp":1781240336881,"version":"3.54.1"},"reference-count":33,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2026,6,12]],"date-time":"2026-06-12T00:00:00Z","timestamp":1781222400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["frontiersin.org"],"crossmark-restriction":true},"short-container-title":["Front. Med."],"abstract":"<jats:p>Optic nerve infiltration as the initial manifestation of isolated central nervous system (CNS) relapse in acute myeloid leukemia (AML) has rarely been reported. We present the case of a 70-years-old man with AML in clinical remission who presented with the sudden onset of a visual field defect and vision loss in the left eye. Initially the patient was diagnosed with non-arteritic anterior ischemic optic neuropathy (NAION) at a local hospital based on findings of decreased vision, optic disc edema and an inferior sectoral visual field defect connected to the physiological blind spot in the left eye, the patient subsequently progressed rapidly to no light perception. Fundus examination showed sequential features consistent with central retinal vein occlusion (CRVO) followed by central retinal artery occlusion (CRAO). Magnetic resonance imaging (MRI) demonstrated thickening and enhancement of the left optic nerve with involvement of the intraconal fat. Cerebrospinal fluid (CSF) cytology confirmed leukemic infiltration, while bone marrow aspiration remained negative for malignant cells, establishing a definitive diagnosis of isolated CNS relapse with optic nerve infiltration. Following intrathecal and systemic chemotherapy, leukemic cells were cleared from the CSF, although the left eye remained without light perception. This case describes the previously unreported sequential onset of optic nerve infiltration, CRVO, and subsequent CRAO in AML, highlighting the need for clinicians to recognize such rare ocular manifestations to detect occult leukemia recurrence early. Atypical or unexplained optic neuropathy in AML patients should raise suspicion of leukemic infiltration. Timely performance of CSF cytology and cranial MRI, together with a multidisciplinary diagnostic and therapeutic approach, is essential for improving visual prognosis and survival rate.<\/jats:p>","DOI":"10.3389\/fmed.2026.1853833","type":"journal-article","created":{"date-parts":[[2026,6,12]],"date-time":"2026-06-12T04:42:35Z","timestamp":1781239355000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":0,"title":["Case Report: Optic nerve infiltration associated with sequential CRVO and CRAO in isolated CNS relapse of AML"],"prefix":"10.3389","volume":"13","author":[{"given":"Yu","family":"Wang","sequence":"first","affiliation":[{"name":"Department of Ophthalmology, Shandong Provincial Hospital Affiliated to Shandong First Medical University, Jinan","place":["Shandong, China"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yiwei","family":"Li","sequence":"additional","affiliation":[{"name":"Department of Ophthalmology, Shandong Provincial Hospital Affiliated to Shandong First Medical University, Jinan","place":["Shandong, China"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jiaxuan","family":"Zhang","sequence":"additional","affiliation":[{"name":"Department of Ophthalmology, Shandong Provincial Hospital Affiliated to Shandong First Medical University, Jinan","place":["Shandong, China"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Bojia","family":"Niu","sequence":"additional","affiliation":[{"name":"Department of Ophthalmology, Shandong Provincial Hospital Affiliated to Shandong First Medical University, Jinan","place":["Shandong, China"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yujie","family":"Jiang","sequence":"additional","affiliation":[{"name":"Department of Hematology, Shandong Provincial Hospital Affiliated to Shandong First Medical University, Jinan","place":["Shandong, China"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ying","family":"Xiao","sequence":"additional","affiliation":[{"name":"Department of Ophthalmology, Shandong Provincial Hospital Affiliated to Shandong First Medical University, Jinan","place":["Shandong, China"]}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1965","published-online":{"date-parts":[[2026,6,12]]},"reference":[{"key":"B1","doi-asserted-by":"crossref","first-page":"1 051","DOI":"10.1056\/NEJM199909303411407","article-title":"Acute myeloid leukemia.","volume":"341","author":"Lowenberg","year":"1999","journal-title":"N Engl J Med."},{"key":"B2","doi-asserted-by":"publisher","first-page":"211","DOI":"10.1016\/0039-6257(83)90123-6","article-title":"Ocular and orbital involvement in leukemia.","volume":"27","author":"Kincaid","year":"1983","journal-title":"Surv Ophthalmol."},{"key":"B3","doi-asserted-by":"publisher","first-page":"663","DOI":"10.1038\/sj.eye.6701308","article-title":"Ophthalmic manifestations of acute leukaemias: the ophthalmologist\u2019s role.","volume":"18","author":"Sharma","year":"2004","journal-title":"Eye."},{"key":"B4","doi-asserted-by":"publisher","first-page":"2119","DOI":"10.2147\/opth.S342720","article-title":"Early ophthalmological manifestations of acute myeloid leukemia: current perspectives.","volume":"16","author":"El Salloukh","year":"2022","journal-title":"Clin Ophthalmol."},{"key":"B5","doi-asserted-by":"publisher","first-page":"7506","DOI":"10.3390\/jcm14217506","article-title":"Multifocal ocular manifestations heralding relapse of acute myeloid leukemia: a case report and literature review.","volume":"14","author":"Mastrogiuseppe","year":"2025","journal-title":"J Clin Med."},{"key":"B6","doi-asserted-by":"publisher","first-page":"2073","DOI":"10.1016\/s0140-6736(23)00108-3","article-title":"Acute myeloid leukaemia.","volume":"401","author":"DiNardo","year":"2023","journal-title":"Lancet."},{"key":"B7","doi-asserted-by":"publisher","first-page":"1","DOI":"10.1155\/2016\/3794284","article-title":"Acute myeloid leukemia relapse presenting as complete monocular vision loss due to optic nerve involvement.","volume":"2016","author":"Patel","year":"2016","journal-title":"Case Rep Hematol."},{"key":"B8","doi-asserted-by":"publisher","first-page":"219","DOI":"10.1136\/bjo-2025-327835","article-title":"Ophthalmic manifestations and treatment outcomes in leukaemia: a comparative analysis of clinical spectrum, visual prognosis and predictors of disease remission across subtypes.","volume":"110","author":"Agarwal","year":"2026","journal-title":"Br J Ophthalmol."},{"key":"B9","doi-asserted-by":"publisher","first-page":"796","DOI":"10.1097\/iae.0000000000004771","article-title":"Leukemia and retinal vascular occlusion: a systematic review and meta-analysis of ocular manifestations and vascular complications.","volume":"46","author":"Chen","year":"2026","journal-title":"Retina."},{"key":"B10","doi-asserted-by":"publisher","first-page":"3519","DOI":"10.1007\/s00277-024-05861-2","article-title":"Highlights of ophthalmological manifestations in newly diagnosed acute leukemia: a correlation with hematological parameters.","volume":"103","author":"Laimon","year":"2024","journal-title":"Ann Hematol."},{"key":"B11","doi-asserted-by":"publisher","first-page":"17","DOI":"10.1016\/j.oret.2017.05.009","article-title":"Ocular manifestations in leukemias and their correlation with hematologic parameters at a tertiary care setting in South India.","volume":"2","author":"Soman","year":"2018","journal-title":"Ophthalmol Retina."},{"key":"B12","doi-asserted-by":"publisher","first-page":"899","DOI":"10.1016\/s0161-6420(83)80013-x","article-title":"Ocular manifestations of leukemia.","volume":"90","author":"Ralph Rosenthal","year":"1983","journal-title":"Ophthalmology."},{"key":"B13","doi-asserted-by":"publisher","first-page":"81","DOI":"10.1046\/j.1600-0420.2003.00197.x","article-title":"Retinopathy in acute leukaemia at initial diagnosis: correlation of fundus lesions and haematological parameters.","volume":"82","author":"Reddy","year":"2003","journal-title":"Acta Ophthalmol Scand."},{"key":"B14","doi-asserted-by":"publisher","first-page":"530","DOI":"10.1016\/j.survophthal.2021.07.001","article-title":"Ophthalmologic manifestations as the initial presentation of chronic myeloid leukemia: a review.","volume":"67","author":"Yassin","year":"2022","journal-title":"Surv Ophthalmol."},{"key":"B15","doi-asserted-by":"publisher","first-page":"490","DOI":"10.1001\/archopht.1961.00960010492010","article-title":"Ocular involvement inleukemia and allied disorders.","volume":"66","author":"Allen","year":"1961","journal-title":"Arch Ophthalmol."},{"key":"B16","doi-asserted-by":"publisher","first-page":"427","DOI":"10.1007\/s11912-022-01220-4","article-title":"Central nervous system involvement in adults with acute leukemia: diagnosis, prevention, and management.","volume":"24","author":"Paul","year":"2022","journal-title":"Curr Oncol Rep."},{"key":"B17","doi-asserted-by":"publisher","first-page":"311","DOI":"10.1007\/s12185-020-03065-8","article-title":"The eyes have it: CNS leukemia presenting as optic neuritis.","volume":"113","author":"Quann","year":"2021","journal-title":"Intern J Hematol."},{"key":"B18","doi-asserted-by":"publisher","first-page":"9172","DOI":"10.1200\/jco.2005.02.7482","article-title":"Risk factors and therapy for isolated central nervous system relapse of pediatric acute myeloid leukemia.","volume":"23","author":"Johnston","year":"2005","journal-title":"J Clin Oncol."},{"key":"B19","doi-asserted-by":"publisher","first-page":"1392","DOI":"10.3109\/10428194.2014.953148","article-title":"Incidence of and risk factors for involvement of the central nervous system in acute myeloid leukemia.","volume":"56","author":"Rozovski","year":"2014","journal-title":"Leukemia Lymphoma."},{"key":"B20","doi-asserted-by":"publisher","first-page":"112","DOI":"10.1002\/cncr.26253","article-title":"Cytogenetic profile of patients with acute myeloid leukemia and central nervous system disease.","volume":"118","author":"Shihadeh","year":"2011","journal-title":"Cancer."},{"key":"B21","doi-asserted-by":"crossref","first-page":"1071","DOI":"10.1182\/blood.V65.5.1071.1071","article-title":"A unique pattern of central nervous system leukemia in acute myelomonocytic leukemia associated with Inv(16)(P13q22).","volume":"65","author":"Holmes","year":"1985","journal-title":"Blood."},{"key":"B22","doi-asserted-by":"publisher","first-page":"2446","DOI":"10.1016\/j.ophtha.2016.08.017","article-title":"Demographic, systemic, and ocular factors associated with nonarteritic anterior ischemic optic neuropathy.","volume":"123","author":"Cestari","year":"2016","journal-title":"Ophthalmology."},{"key":"B23","doi-asserted-by":"publisher","first-page":"790","DOI":"10.1016\/j.ophtha.2024.01.011","article-title":"Ophthalmic and systemic factors of acute nonarteritic anterior ischemic optic neuropathy in the Quark207 treatment trial.","volume":"131","author":"Kupersmith","year":"2024","journal-title":"Ophthalmology."},{"key":"B24","doi-asserted-by":"publisher","first-page":"121","DOI":"10.4103\/meajo.meajo_69_23","article-title":"Bilateral retinal infiltration and ischemia as the first presenting sign of chronic myeloid leukemia: a case report with multimodal imaging.","volume":"30","author":"Alzuabi","year":"2023","journal-title":"Middle East Afr J Ophthalmol."},{"key":"B25","doi-asserted-by":"publisher","first-page":"759","DOI":"10.3129\/i07-133","article-title":"Bilateral central retinal artery occlusion associated with leukemic optic neuropathy.","volume":"42","author":"Lin","year":"2007","journal-title":"Can J Ophthalmol."},{"key":"B26","doi-asserted-by":"publisher","first-page":"e25625","DOI":"10.7759\/cureus.25625","article-title":"Isolated infiltrative optic neuropathy in an acute lymphoblastic leukemia relapse.","volume":"14","author":"Kuan","year":"2022","journal-title":"Cureus."},{"key":"B27","doi-asserted-by":"publisher","first-page":"101493","DOI":"10.1016\/j.ajoc.2022.101493","article-title":"Unilateral isolated optic nerve infiltration combined with central retinal artery occlusion in a patient with acute myeloid leukemia.","volume":"26","author":"Zhao","year":"2022","journal-title":"Am J Ophthalmol Case Rep."},{"key":"B28","doi-asserted-by":"publisher","first-page":"37","DOI":"10.1159\/000309220","article-title":"Leukemic optic neuropathy.","volume":"185","author":"Takahashi","year":"1982","journal-title":"Ophthalmologica."},{"key":"B29","doi-asserted-by":"publisher","first-page":"231","DOI":"10.2174\/1573399811666150526151241","article-title":"The interactions between diabetes mellitus and myelodysplastic syndromes: current state of evidence and future directions.","volume":"12","author":"Hamoudeh","year":"2016","journal-title":"Curr Diab Rev."},{"key":"B30","doi-asserted-by":"publisher","first-page":"540","DOI":"10.1007\/s00125-020-05338-7","article-title":"Association between diabetes and haematological malignancies: a population-based study.","volume":"64","author":"Gong","year":"2021","journal-title":"Diabetologia."},{"key":"B31","doi-asserted-by":"publisher","first-page":"e239795","DOI":"10.1136\/bcr-2020-239795","article-title":"Bilateral retinal artery occlusions as the first manifestation of extramedullary central nervous system involvement in relapsed acute myeloid leukaemia.","volume":"14","author":"Khair","year":"2021","journal-title":"BMJ Case Rep."},{"key":"B32","doi-asserted-by":"publisher","first-page":"1744","DOI":"10.1002\/1097-0142(197610)38:4<1744::aid-cncr2820380449>3.0.co;2-3","article-title":"Leukemic ophthalmopathy in children.","volume":"38","author":"Ridgway","year":"1976","journal-title":"Cancer."},{"key":"B33","doi-asserted-by":"publisher","first-page":"431","DOI":"10.1017\/cjn.2020.31","article-title":"Complete recovery of vision after optic nerve relapse of acute lymphoblastic leukemia.","volume":"47","author":"Hu","year":"2020","journal-title":"Can J Neurol Sci."}],"container-title":["Frontiers in Medicine"],"original-title":[],"link":[{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fmed.2026.1853833\/full","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,6,12]],"date-time":"2026-06-12T04:42:59Z","timestamp":1781239379000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fmed.2026.1853833\/full"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,6,12]]},"references-count":33,"alternative-id":["10.3389\/fmed.2026.1853833"],"URL":"https:\/\/doi.org\/10.3389\/fmed.2026.1853833","relation":{},"ISSN":["2296-858X"],"issn-type":[{"value":"2296-858X","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,6,12]]},"article-number":"1853833"}}