{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,10,10]],"date-time":"2025-10-10T01:37:00Z","timestamp":1760060220806,"version":"build-2065373602"},"reference-count":25,"publisher":"MDPI AG","issue":"16","license":[{"start":{"date-parts":[[2025,8,15]],"date-time":"2025-08-15T00:00:00Z","timestamp":1755216000000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Diagnostics"],"abstract":"<jats:p>Introduction: Hypertrophic olivary degeneration (HOD) is a rare form of trans-synaptic degeneration involving the Guillain\u2013Mollaret triangle, characterized by enlargement of the inferior olivary nucleus\u2014unlike the atrophy typical of most neurodegenerative processes. It is usually associated with stroke, surgical injury, or demyelination, but rarely follows hemorrhage from a cavernous malformation (CM). This report presents a case of HOD secondary to a mesencephalic CM hemorrhage, with emphasis on imaging findings and diagnostic considerations. Case Description: A 55-year-old woman presented with acute-onset, right-sided facial, torso, and limb hypoesthesia, along with gait instability. Neurological examination revealed sensory impairment in the right maxillary (V2) and mandibular (V3) trigeminal territories, as well as diminished pain and temperature sensation throughout the right hemibody. MRI revealed a hemorrhage in the posterior mesencephalon near the left red nucleus, leading to the diagnosis of a CM with an associated venous angioma. She was managed conservatively and improved clinically. Six months later, MRI showed hypertrophy and T2\/FLAIR hyperintensity of the left inferior olive, consistent with developing HOD. At 1.5 years follow-up, olivary enlargement had progressed\u2014now consistent with stage 2 HOD\u2014and a bilateral palatal tremor was observed, more pronounced on the right side. DTI revealed asymmetric volume loss in the left brainstem fiber pathways at the level of the medulla oblongata, confirming trans-synaptic degeneration. Conclusions: This case highlights HOD as a rare but important complication of mesencephalic CM hemorrhage. Recognition of its characteristic imaging features\u2014olivary hypertrophy with persistent T2\/FLAIR hyperintensity\u2014is essential for accurate diagnosis. DTI supports the trans-synaptic mechanism, helping distinguish HOD from other pathologies and preventing unnecessary investigations.<\/jats:p>","DOI":"10.3390\/diagnostics15162048","type":"journal-article","created":{"date-parts":[[2025,8,15]],"date-time":"2025-08-15T10:33:42Z","timestamp":1755254022000},"page":"2048","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Another Rare Cause of Hypertrophic Olivary Degeneration Following Cavernous Malformation Hemorrhage: A Case Report"],"prefix":"10.3390","volume":"15","author":[{"ORCID":"https:\/\/orcid.org\/0009-0004-0119-1352","authenticated-orcid":false,"given":"Sigita","family":"Skrasti\u0146a","sequence":"first","affiliation":[{"name":"Faculty of Medicine, Riga Stradins University, 16 Dzirciema Street, LV-1007 Riga, Latvia"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9416-1600","authenticated-orcid":false,"given":"Marija","family":"Roddate","sequence":"additional","affiliation":[{"name":"Clinic of Neurology, Pauls Stradins Clinical University Hospital, 13 Pilsonu Street, LV-1002 Riga, Latvia"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kristaps","family":"Ranc\u0101ns","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery, Pauls Stradins Clinical University Hospital, 13 Pilsonu Street, LV-1002 Riga, Latvia"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4862-6726","authenticated-orcid":false,"given":"Evija","family":"Migl\u0101ne","sequence":"additional","affiliation":[{"name":"Clinic of Neurology, Pauls Stradins Clinical University Hospital, 13 Pilsonu Street, LV-1002 Riga, Latvia"},{"name":"Department of Neurology and Neurosurgery, Riga Stradins University, 16 Dzirciema Street, LV-1007 Riga, Latvia"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Aleksandrs","family":"Kalni\u0146\u0161","sequence":"additional","affiliation":[{"name":"Division of Neuroradiology, Department of Radiology, Pritzker School of Medicine, The University of Chicago Medical Center, 5841 S. Maryland Avenue, Chicago, IL 60637, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7457-1901","authenticated-orcid":false,"given":"Arturs","family":"Balodis","sequence":"additional","affiliation":[{"name":"Institute of Diagnostic Radiology, Pauls Stradins Clinical University Hospital, 13 Pilsonu Street, LV-1002 Riga, Latvia"},{"name":"Department of Radiology, Riga Stradins University, 16 Dzirciema Street, LV-1007 Riga, Latvia"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1968","published-online":{"date-parts":[[2025,8,15]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"53","DOI":"10.1016\/j.brainres.2019.04.024","article-title":"Hypertrophic olivary degeneration: A comprehensive review focusing on etiology","volume":"1718","author":"Wang","year":"2019","journal-title":"Brain Res."},{"key":"ref_2","doi-asserted-by":"crossref","first-page":"71","DOI":"10.5334\/jbr-btr.1065","article-title":"Imaging Features of Hypertrophic Olivary Degeneration","volume":"100","author":"Lemmerling","year":"2016","journal-title":"J. Belg. Soc. Radiol."},{"key":"ref_3","doi-asserted-by":"crossref","first-page":"341","DOI":"10.1007\/s11060-008-9523-7","article-title":"Hypertrophic olivary degeneration after resection of a cerebellar tumor","volume":"87","author":"Akar","year":"2008","journal-title":"J. Neurooncol."},{"key":"ref_4","doi-asserted-by":"crossref","unstructured":"Tilikete, C., and Desestret, V. (2017). Hypertrophic olivary degeneration and palatal or oculopalatal tremor. Front. Neurol., 8.","DOI":"10.3389\/fneur.2017.00302"},{"key":"ref_5","doi-asserted-by":"crossref","first-page":"401","DOI":"10.4103\/0971-3026.143902","article-title":"MRI and MR tractography in bilateral hypertrophic olivary degeneration","volume":"24","author":"Sen","year":"2014","journal-title":"Indian J. Radiol. Imaging"},{"key":"ref_6","first-page":"1073","article-title":"Hypertrophic olivary degeneration: Metaanalysis of the temporal evolution of MR findings","volume":"21","author":"Goyal","year":"2000","journal-title":"AJNR Am. J. Neuroradiol."},{"key":"ref_7","doi-asserted-by":"crossref","first-page":"3480","DOI":"10.21037\/qims-21-1048","article-title":"Hypertrophic olivary degeneration: A description of four cases of and a literature analysis","volume":"12","author":"Gao","year":"2022","journal-title":"Quant. Imaging Med. Surg."},{"key":"ref_8","doi-asserted-by":"crossref","first-page":"235","DOI":"10.1007\/s00062-009-9001-4","article-title":"Hypertrophic olivary degeneration following pontine cavernoma hemorrhage: A typical change accompanying lesions in the Guillain-Mollaret triangle","volume":"19","author":"MacHt","year":"2009","journal-title":"Clin. Neuroradiol."},{"key":"ref_9","doi-asserted-by":"crossref","first-page":"413","DOI":"10.1007\/s00234-011-0897-4","article-title":"Hypertrophic olivary degeneration after pontine hemorrhage","volume":"54","author":"Asal","year":"2012","journal-title":"Neuroradiology"},{"key":"ref_10","doi-asserted-by":"crossref","first-page":"1213","DOI":"10.1016\/j.jocn.2015.02.005","article-title":"Hypertrophic olivary degeneration secondary to pontine haemorrhage","volume":"22","author":"Wein","year":"2015","journal-title":"J. Clin. Neurosci."},{"key":"ref_11","doi-asserted-by":"crossref","first-page":"286","DOI":"10.1016\/j.wneu.2020.02.068","article-title":"Hypertrophic Olivary Degeneration and Holmes Tremor: Case Report and Review of the Literature","volume":"137","author":"Dogan","year":"2020","journal-title":"World Neurosurg."},{"key":"ref_12","doi-asserted-by":"crossref","unstructured":"Schaller-Paule, M.A., Steidl, E., Shrestha, M., Deichmann, R., Steinmetz, H., Seiler, A., Lapa, S., Steiner, T., Thonke, S., and Weidauer, S. (2021). Multicenter Prospective Analysis of Hypertrophic Olivary Degeneration Following Infratentorial Stroke (HOD-IS): Evaluation of Disease Epidemiology, Clinical Presentation, and MR-Imaging Aspects. Front. Neurol., 12.","DOI":"10.3389\/fneur.2021.675123"},{"key":"ref_13","first-page":"292","article-title":"Unusual co-occurrence of hypertrophic inferior olivary degeneration with infratentorial cavernomatosis and orbital cavernous hemangioma","volume":"20","author":"Ridah","year":"2024","journal-title":"Radiol. Case Rep."},{"key":"ref_14","doi-asserted-by":"crossref","first-page":"3538","DOI":"10.1016\/j.radcr.2024.04.078","article-title":"Hypertrophic olivary degeneration secondary to a Guillain Mollaret triangle cavernoma: Two case report","volume":"19","author":"Marrakchi","year":"2024","journal-title":"Radiol. Case Rep."},{"key":"ref_15","doi-asserted-by":"crossref","first-page":"59","DOI":"10.1055\/s-0035-1566114","article-title":"Hypertrophic Olivary Degeneration: A Neurosurgical Point of View","volume":"77","author":"Carvalho","year":"2015","journal-title":"J. Neurol. Surg. A Cent. Eur. Neurosurg."},{"key":"ref_16","first-page":"24","article-title":"Hypertrophic olivary degeneration after resection of a pontine cavernous malformation: A case report","volume":"5","author":"Gatlin","year":"2011","journal-title":"J. Radiol. Case Rep."},{"key":"ref_17","doi-asserted-by":"crossref","first-page":"717","DOI":"10.3171\/jns.2004.100.4.0717","article-title":"Hypertrophic Olivary Degeneration after Resection of a Pontine Cavernoma: Case Illustration","volume":"100","author":"Harter","year":"2004","journal-title":"J. Neurosurg."},{"key":"ref_18","doi-asserted-by":"crossref","first-page":"149","DOI":"10.1007\/s00701-007-1470-0","article-title":"Hypertrophic olivary degeneration after surgical removal of cavernous malformations of the brain stem: Report of four cases and review of the literature","volume":"150","author":"Hornyak","year":"2008","journal-title":"Acta Neurochir."},{"key":"ref_19","doi-asserted-by":"crossref","first-page":"137","DOI":"10.1017\/cjn.2018.375","article-title":"An Acute Clinical Presentation Associated with Hypertrophic Olivary Degeneration","volume":"46","author":"Howard","year":"2019","journal-title":"Can. J. Neurol. Sci."},{"key":"ref_20","doi-asserted-by":"crossref","first-page":"2751","DOI":"10.2169\/internalmedicine.53.3679","article-title":"Hypertrophic Olivary Degeneration with an Egg-shaped Appearance","volume":"53","author":"Kosaka","year":"2014","journal-title":"Intern. Med."},{"key":"ref_21","doi-asserted-by":"crossref","first-page":"108422","DOI":"10.1016\/j.clineuro.2024.108422","article-title":"Postoperative brainstem cavernous malformations complicated with LGI1 encephalitis and hypertrophic olivary degeneration: A case report and literature review","volume":"244","author":"Zhu","year":"2024","journal-title":"Clin. Neurol. Neurosurg."},{"key":"ref_22","doi-asserted-by":"crossref","first-page":"40","DOI":"10.5334\/tohm.188","article-title":"Palatal Tremor\u2014Pathophysiology, Clinical Features, Investigations, Management and Future Challenges","volume":"10","author":"Bhattacharjee","year":"2020","journal-title":"Tremor Other Hyperkinetic Mov."},{"key":"ref_23","doi-asserted-by":"crossref","first-page":"857","DOI":"10.1007\/s00415-006-0039-9","article-title":"Botulinum toxin is effective and safe for palatal tremor: A report of five cases and a review of the literature","volume":"253","author":"Penney","year":"2006","journal-title":"J. Neurol."},{"key":"ref_24","first-page":"414620","article-title":"Essential Palatal Tremor Managed by Cognitive Behavioral Therapy","volume":"2015","author":"Kitamura","year":"2015","journal-title":"Case Rep. Dent."},{"key":"ref_25","doi-asserted-by":"crossref","first-page":"264","DOI":"10.5334\/tohm.210","article-title":"Hypertrophic Olivary Degeneration and Holmes\u2019 Tremor Secondary to Bleeding of Cavernous Malformation in the Midbrain","volume":"4","author":"Cury","year":"2014","journal-title":"Tremor Other Hyperkinetic Mov."}],"container-title":["Diagnostics"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.mdpi.com\/2075-4418\/15\/16\/2048\/pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,10,9]],"date-time":"2025-10-09T18:28:08Z","timestamp":1760034488000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.mdpi.com\/2075-4418\/15\/16\/2048"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,8,15]]},"references-count":25,"journal-issue":{"issue":"16","published-online":{"date-parts":[[2025,8]]}},"alternative-id":["diagnostics15162048"],"URL":"https:\/\/doi.org\/10.3390\/diagnostics15162048","relation":{},"ISSN":["2075-4418"],"issn-type":[{"type":"electronic","value":"2075-4418"}],"subject":[],"published":{"date-parts":[[2025,8,15]]}}}