{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,2,19]],"date-time":"2026-02-19T12:54:23Z","timestamp":1771505663037,"version":"3.50.1"},"reference-count":53,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2022,5,30]],"date-time":"2022-05-30T00:00:00Z","timestamp":1653868800000},"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. Surg."],"abstract":"<jats:sec><jats:title>Background<\/jats:title><jats:p>Hemorrhage into optic pathway\u2013hypothalamic glioma (OPHG) is rare. Variable clinical presentations and outcomes are associated with such pathology. We aim to present two infants presented with OPHG and a systematic review of the literature.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>We describe two cases of infants presenting with sudden decreased vision, poor feeding, and irritability due to OPHG. Both patients underwent urgent craniotomy and subtotal resection followed by chemotherapy. We systematically reviewed the literature using PubMed, Google Scholar, and Embase. In addition, we included all English published reports for all ages discussing the optic pathway (optic nerve and optic chiasm) or hypothalamic glioma associated with hemorrhage from the year of the first reported case (1970) to January 2022.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Of 17,949, 44 articles met the inclusion criteria of this review. A total of 56 cases were described with a mean of 21.35 years (0.5\u201370), with the male gender 52% and the female gender 45%. The hemorrhage location was sellar\/suprasellar in 43% cases. Histopathology of included cases was pilocytic astrocytoma in 41%, followed by pilomyxoid astrocytoma in 16% cases. The outcome was unfavorable; 37.5% cases showed improvement, whereas 18% cases resulted in death.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>Apoplexy of the OPHG can be fatal and associated with poor outcomes. A systematic review of the literature has shown that younger age, pilocytic or pilomexyoid astrocytoma histopathology, and chiasmal\/hypothalamic locations are associated with a higher risk of intertumoral hemorrhage and poor prognosis. Further genetic studies for OPHG may provide information for high-risk patients.<\/jats:p><\/jats:sec>","DOI":"10.3389\/fsurg.2022.891556","type":"journal-article","created":{"date-parts":[[2022,5,30]],"date-time":"2022-05-30T05:05:26Z","timestamp":1653887126000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":5,"title":["Optic Pathway\u2013Hypothalamic Glioma Apoplexy: A Report of Two Cases and Systematic Review of the Literature"],"prefix":"10.3389","volume":"9","author":[{"given":"Saleh","family":"Baeesa","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yazid","family":"Maghrabi","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Rana","family":"Moshref","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jaudah","family":"Al-Maghrabi","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1965","published-online":{"date-parts":[[2022,5,30]]},"reference":[{"key":"B1","doi-asserted-by":"publisher","first-page":"2041","DOI":"10.1007\/s00381-015-2855-7","article-title":"Optic pathway glioma in children: does visual deficit correlate with radiology in focal exophytic lesions?","volume":"31","author":"Aquilina","year":"2015","journal-title":"Childs Nerv Syst"},{"key":"B2","doi-asserted-by":"publisher","first-page":"E2","DOI":"10.3171\/FOC-07\/11\/E2","article-title":"Optic pathway gliomas: a review","volume":"23","author":"Binning","year":"2007","journal-title":"Neurosurg Focus"},{"key":"B3","doi-asserted-by":"publisher","first-page":"803","DOI":"10.1007\/s00401-016-1545-1","article-title":"The 2016 world health organization classification of tumors of the central nervous system: a summary","volume":"131","author":"Louis","year":"2016","journal-title":"Acta Neuropathol"},{"key":"B4","doi-asserted-by":"publisher","first-page":"273","DOI":"10.1227\/NEU.0000000000000257","article-title":"Optic pathway gliomas in adults","volume":"74","author":"Shofty","year":"2014","journal-title":"Neurosurgery"},{"key":"B5","doi-asserted-by":"publisher","first-page":"1917","DOI":"10.1007\/s00381-021-05060-8","article-title":"Neurosurgical experience of managing optic pathway gliomas","volume":"37","author":"Hill","year":"2021","journal-title":"Childs Nerv Syst"},{"key":"B6","doi-asserted-by":"publisher","first-page":"1407","DOI":"10.3171\/2017.8.JNS163085","article-title":"Optic pathway-hypothalamic glioma hemorrhage: a series of 9 patients and review of the literature","volume":"129","author":"van Baarsen","year":"2018","journal-title":"J Neurosurg"},{"key":"B7","doi-asserted-by":"publisher","first-page":"437","DOI":"10.1227\/00006123-198204000-00004","article-title":"Spontaneous intracranial hemorrhage caused by brain tumor: its incidence and clinical significance","volume":"10","author":"Wakai","year":"1982","journal-title":"Neurosurgery"},{"key":"B8","first-page":"201","article-title":"Bleeding cerebral neoplasms with symptomatic hematoma","volume":"47","author":"Licata","year":"2003","journal-title":"J Neurosurg Sci"},{"key":"B9","first-page":"365","article-title":"Brain tumors with hemorrhage","volume":"98","author":"Lieu","year":"1999","journal-title":"J Formos Med Assoc"},{"key":"B10","doi-asserted-by":"publisher","first-page":"1","DOI":"10.1007\/s10014-008-0243-7","article-title":"Hemorrhagic onset of pilocytic astrocytoma and pilomyxoid astrocytoma","volume":"26","author":"Shibahara","year":"2009","journal-title":"Brain Tumor Pathol"},{"key":"B11","doi-asserted-by":"publisher","first-page":"101","DOI":"10.1186\/s12887-021-02556-9","article-title":"Gliosarcoma in patients under 20\u2009years of age. A clinicopathologic study of 11 cases and a detailed review of the literature","volume":"21","author":"Din","year":"2021","journal-title":"BMC Pediatr"},{"key":"B12","doi-asserted-by":"publisher","first-page":"733","DOI":"10.3171\/2019.12.JNS192155","article-title":"Association of the FGFR1 mutation with spontaneous hemorrhage in low-grade gliomas in pediatric and young adult patients","volume":"134","author":"Ishi","year":"2020","journal-title":"J Neurosurg"},{"key":"B13","doi-asserted-by":"publisher","first-page":"462","DOI":"10.1097\/ICB.0000000000000821","article-title":"Optic disk astrocytoma unassociated with tuberous sclerosis complex managed with surgical excision and a 7-year follow-up","volume":"15","author":"Lu","year":"2021","journal-title":"Retin Cases Brief Rep"},{"key":"B14","doi-asserted-by":"publisher","first-page":"102","DOI":"10.1001\/jamaophthalmol.2019.4511","article-title":"SETD2 mutation in an aggressive optic nerve glioma","volume":"138","author":"Campbell","year":"2020","journal-title":"JAMA Ophthalmol"},{"key":"B15","doi-asserted-by":"publisher","DOI":"10.1136\/bcr-2020-235662","article-title":"Reappraisal of haemorrhagic suprasellar pilocytic astrocytoma during adulthood","volume":"13","author":"Cortez","year":"2020","journal-title":"BMJ Case Rep"},{"key":"B16","doi-asserted-by":"publisher","first-page":"2050313X18761310","DOI":"10.1177\/2050313X18761310","article-title":"Intraorbital ancient pilocytic astrocytoma of the optic nerve in neurofibromatosis type 1 patient presenting with sudden ocular pain","volume":"6","author":"Motoyama","year":"2018","journal-title":"SAGE Open Med Case Rep"},{"key":"B17","doi-asserted-by":"publisher","first-page":"368","DOI":"10.1177\/1066896917692098","article-title":"Intraocular ependymoma with blood-filled spaces: neoplasm or a reactive process with ependymal differentiation\u2014a dilemma","volume":"25","author":"Dewan","year":"2017","journal-title":"Int J Surg Pathol"},{"key":"B18","doi-asserted-by":"publisher","first-page":"167","DOI":"10.3126\/nepjoph.v8i2.17007","article-title":"Ocular myiasis in a glioma: a case report","volume":"8","author":"Mathew","year":"2016","journal-title":"Nepal J Ophthalmol"},{"key":"B19","doi-asserted-by":"publisher","first-page":"90","DOI":"10.17116\/neiro201680590-97","article-title":"[Chiasm and optic nerve glioma manifested as hemorrhage (two clinical cases and a literature review)]","volume":"80","author":"Serova","year":"2016","journal-title":"Zh Vopr Neirokhir Im N N Burdenko"},{"key":"B20","doi-asserted-by":"publisher","first-page":"217","DOI":"10.1016\/j.jocn.2016.03.033","article-title":"Spontaneous intratumoural and intraventricular haemorrhage associated with a pilomyxoid astrocytoma in the hypothalamic\/chiasmatic region","volume":"33","author":"Wang","year":"2016","journal-title":"J Clin Neurosci"},{"key":"B21","doi-asserted-by":"publisher","first-page":"1617","DOI":"10.1007\/s00381-015-2749-8","article-title":"Spontaneous hemorrhages in pediatric supratentorial pilocytic astrocytomas. Malignant presentation of a benign entity","volume":"31","author":"Kapoor","year":"2015","journal-title":"Childs Nerv Syst"},{"key":"B22","doi-asserted-by":"publisher","first-page":"136","DOI":"10.1016\/j.neucir.2013.11.001","article-title":"Chiasmal haemorrhage secondary to glioma with unusual MRI appearance","volume":"25","author":"Arrese","year":"2014","journal-title":"Neurocirugia (Astur)"},{"key":"B23","doi-asserted-by":"publisher","first-page":"623","DOI":"10.1007\/s10072-013-1624-1","article-title":"The rare event of optic-chiasmatic hemorrhagic low grade glioma in adulthood. Considerations on treatment strategy","volume":"35","author":"Della Puppa","year":"2014","journal-title":"Neurol Sci"},{"key":"B24","doi-asserted-by":"publisher","first-page":"315","DOI":"10.1097\/CAD.0b013e32835c7a47","article-title":"Long-term response in high-grade optic glioma treated with medically induced hypothyroidism and carboplatin: a case report and review of the literature","volume":"24","author":"Ashur-Fabian","year":"2013","journal-title":"Anticancer Drugs"},{"key":"B25","doi-asserted-by":"publisher","first-page":"632","DOI":"10.1016\/j.clineuro.2012.06.015","article-title":"Multiple discrete aneurysmal subarachnoid hemorrhages during multimodality management of a hypothalamic glioma\u2014case report","volume":"115","author":"Faraji","year":"2013","journal-title":"Clin Neurol Neurosurg"},{"key":"B26","doi-asserted-by":"publisher","first-page":"1610","DOI":"10.1016\/j.jocn.2012.09.044","article-title":"Late adult onset optic pathway astrocytoma","volume":"20","author":"Liu","year":"2013","journal-title":"J Clin Neurosci"},{"key":"B27","doi-asserted-by":"publisher","first-page":"279","DOI":"10.1159\/000334309","article-title":"Opticochiasmatic apoplexy in a five-year-old","volume":"47","author":"Ball","year":"2011","journal-title":"Pediatr Neurosurg"},{"key":"B28","doi-asserted-by":"publisher","first-page":"633","DOI":"10.3171\/2011.3.PEDS10562","article-title":"Tumor bleeding from a de novo aneurysm associated with optic glioma","volume":"7","author":"Vogel","year":"2011","journal-title":"J Neurosurg Pediatr"},{"key":"B29","doi-asserted-by":"publisher","first-page":"244","DOI":"10.3171\/PED\/2008\/1\/3\/244","article-title":"Pilomyxoid astrocytoma in a patient presenting with fatal hemorrhage. Case report","volume":"1","author":"Hamada","year":"2008","journal-title":"J Neurosurg Pediatr"},{"key":"B30","doi-asserted-by":"publisher","first-page":"223","DOI":"10.3171\/JNS\/2008\/108\/2\/0223","article-title":"Rate of spontaneous hemorrhage in histologically proven cases of pilocytic astrocytoma","volume":"108","author":"White","year":"2008","journal-title":"J Neurosurg"},{"key":"B31","doi-asserted-by":"publisher","first-page":"525","DOI":"10.3171\/ped.2004.100.5.0525","article-title":"Juvenile pilocytic astrocytoma presenting with subarachnoid hemorrhage. case report and review of the literature","volume":"100","author":"Garg","year":"2004","journal-title":"J Neurosurg"},{"key":"B32","doi-asserted-by":"publisher","first-page":"559","DOI":"10.2176\/nmc.43.559","article-title":"Optic nerve glioma manifesting as intratumoral hemorrhage in a pregnant woman\u2013case report","volume":"43","author":"Yokoyama","year":"2003","journal-title":"Neurol Med Chir (Tokyo)"},{"key":"B33","doi-asserted-by":"publisher","first-page":"173","DOI":"10.1007\/s003810000364","article-title":"Intracranial hemorrhage from an aneurysm encased in a pilocytic astrocytoma\u2014case report and review of the literature","volume":"17","author":"Aichholzer","year":"2001","journal-title":"Childs Nerv Syst"},{"key":"B34","doi-asserted-by":"publisher","first-page":"567","DOI":"10.1007\/s003810100449","article-title":"Hypothalamic tumour with haemorrhage","volume":"17","author":"Devi","year":"2001","journal-title":"Childs Nerv Syst"},{"key":"B35","doi-asserted-by":"publisher","first-page":"120","DOI":"10.1038\/eye.1999.27","article-title":"Chiasmal apoplexy, an unusual complication of cerebral glioblastoma","volume":"13","author":"Wright","year":"1999","journal-title":"Eye (Lond)"},{"key":"B36","doi-asserted-by":"publisher","first-page":"59","DOI":"10.1080\/02688699845564","article-title":"Low grade pilocytic astrocytoma presenting as a spontaneous intracerebral haemorrhage in a child","volume":"12","author":"Golash","year":"1998","journal-title":"Br J Neurosurg"},{"key":"B37","first-page":"784","article-title":"Hypothalamic juvenile pilocytic astrocytoma presenting with intracerebral hemorrhage","volume":"97","author":"Hwang","year":"1998","journal-title":"J Formos Med Assoc"},{"key":"B38","doi-asserted-by":"publisher","first-page":"849","DOI":"10.2176\/nmc.37.849","article-title":"Hypothalamic pilocytic astrocytoma presenting with intratumoral and subarachnoid hemorrhage","volume":"37","author":"Matsumoto","year":"1997","journal-title":"Neurol Med Chir (Tokyo)"},{"key":"B39","doi-asserted-by":"publisher","first-page":"238","DOI":"10.2176\/nmc.35.238","article-title":"Mixed cavernous angioma and glioma (angioglioma) in the hypothalamus\u2013case report","volume":"35","author":"Hasegawa","year":"1995","journal-title":"Neurol Med Chir (Tokyo)"},{"key":"B40","doi-asserted-by":"publisher","first-page":"761","DOI":"10.1136\/jnnp.58.6.761","article-title":"Transient amnestic syndrome after spontaneous haemorrhage into a hypothalamic pilocytic astrocytoma","volume":"58","author":"Sorenson","year":"1995","journal-title":"J Neurol Neurosurg Psychiatry"},{"key":"B41","doi-asserted-by":"publisher","first-page":"88","DOI":"10.1159\/000120573","article-title":"Sudden and unexpected death due to hemorrhage from occult central nervous system lesions. A pediatric autopsy study","volume":"17","author":"Byard","year":"1991","journal-title":"Pediatr Neurosurg"},{"key":"B42","first-page":"15","article-title":"Hematoma of optic nerve glioma\u2013a cause for sudden proptosis. Magnetic resonance imaging findings","volume":"9","author":"Applegate","year":"1989","journal-title":"J Clin Neuroophthalmol"},{"key":"B43","first-page":"335","article-title":"Acute visual loss due to a calcified optic nerve glioma","volume":"24","author":"Jordan","year":"1989","journal-title":"Can J Ophthalmol"},{"key":"B44","doi-asserted-by":"publisher","first-page":"65","DOI":"10.1007\/BF00271124","article-title":"Subarachnoid hemorrhage from brain tumors in childhood","volume":"3","author":"Yokota","year":"1987","journal-title":"Childs Nerv Syst"},{"key":"B45","doi-asserted-by":"publisher","first-page":"118","DOI":"10.3171\/jns.1982.56.1.0118","article-title":"Chiasmal apoplexy. Report of four cases","volume":"56","author":"Maitland","year":"1982","journal-title":"J Neurosurg"},{"key":"B46","doi-asserted-by":"publisher","first-page":"141","DOI":"10.1016\/S0090-3019(82)80043-8","article-title":"Diencephalic syndrome of emaciation (Russell\u2019s syndrome)","volume":"17","author":"Waga","year":"1982","journal-title":"Surg Neurol"},{"key":"B47","doi-asserted-by":"publisher","first-page":"691","DOI":"10.1016\/S0002-9394(14)74663-X","article-title":"Pilocytic astrocytoma of the optic nerve with hemorrhage and extreme cystic degeneration","volume":"92","author":"Charles","year":"1981","journal-title":"Am J Ophthalmol"},{"key":"B48","article-title":"Juvenile pilocytic astrocytoma (\u2018glioma\u2019) of the optic nerve","volume-title":"Ocular and adnexal tumors","author":"Yanoff","year":"1978"},{"key":"B49","first-page":"139","article-title":"Simulated pituitary apoplexy: report of an unusual case due to hemorrhage into hypothalamic astrocytoma","volume":"9","author":"Glew","year":"1977","journal-title":"Ann Ophthalmol"},{"key":"B50","doi-asserted-by":"publisher","first-page":"197","DOI":"10.3171\/jns.1970.32.2.0197","article-title":"Prechiasmal infarction associated with intrachiasmal and suprasellar tumors","volume":"32","author":"Schneider","year":"1970","journal-title":"J Neurosurg"},{"key":"B51","doi-asserted-by":"publisher","first-page":"471","DOI":"10.1177\/08830738030180070401","article-title":"Optic pathway gliomas in children with and without neurofibromatosis 1","volume":"18","author":"Czyzyk","year":"2003","journal-title":"J Child Neurol"},{"key":"B52","doi-asserted-by":"publisher","first-page":"201","DOI":"10.1055\/s-2007-1023231","article-title":"Cavernous hemangioma of the optic chiasm: a surgical review","volume":"18","author":"Crocker","year":"2008","journal-title":"Skull Base"},{"key":"B53","doi-asserted-by":"publisher","first-page":"414","DOI":"10.1159\/000149911","article-title":"Chiasmal cavernoma: a rare cause of acute visual loss improved by prompt surgery","volume":"44","author":"Newman","year":"2008","journal-title":"Pediatr Neurosurg"}],"container-title":["Frontiers in Surgery"],"original-title":[],"link":[{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fsurg.2022.891556\/full","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2022,6,6]],"date-time":"2022-06-06T13:42:12Z","timestamp":1654522932000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fsurg.2022.891556\/full"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2022,5,30]]},"references-count":53,"alternative-id":["10.3389\/fsurg.2022.891556"],"URL":"https:\/\/doi.org\/10.3389\/fsurg.2022.891556","relation":{},"ISSN":["2296-875X"],"issn-type":[{"value":"2296-875X","type":"electronic"}],"subject":[],"published":{"date-parts":[[2022,5,30]]},"article-number":"891556"}}