{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,31]],"date-time":"2026-10-31T17:40:04Z","timestamp":1793468404912,"version":"4.5.0"},"reference-count":45,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2024,3,15]],"date-time":"2024-03-15T00:00:00Z","timestamp":1710460800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100001691","name":"Japan Society for the Promotion of Science","doi-asserted-by":"publisher","award":["21K09098"],"award-info":[{"award-number":["21K09098"]}],"id":[{"id":"10.13039\/501100001691","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001691","name":"Japan Society for the Promotion of Science","doi-asserted-by":"publisher","award":["22H03020"],"award-info":[{"award-number":["22H03020"]}],"id":[{"id":"10.13039\/501100001691","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001691","name":"Japan Society for the Promotion of Science","doi-asserted-by":"publisher","award":["22H00190"],"award-info":[{"award-number":["22H00190"]}],"id":[{"id":"10.13039\/501100001691","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001700","name":"Ministry of Education, Culture, Sports, Science and Technology","doi-asserted-by":"publisher","award":["JPMXP1020230118"],"award-info":[{"award-number":["JPMXP1020230118"]}],"id":[{"id":"10.13039\/501100001700","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["frontiersin.org"],"crossmark-restriction":true},"short-container-title":["Front. Aging Neurosci."],"abstract":"<jats:sec><jats:title>Background<\/jats:title><jats:p>Disproportionately enlarged subarachnoid-space hydrocephalus (DESH) is a key feature for Hakim disease (idiopathic normal pressure hydrocephalus: iNPH), but subjectively evaluated. To develop automatic quantitative assessment of DESH with automatic segmentation using combined deep learning models.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>This study included 180 participants (42 Hakim patients, 138 healthy volunteers; 78 males, 102 females). Overall, 159 three-dimensional (3D) T1-weighted and 180 T2-weighted MRIs were included. As a semantic segmentation, 3D MRIs were automatically segmented in the total ventricles, total subarachnoid space (SAS), high-convexity SAS, and Sylvian fissure and basal cistern on the 3D U-Net model. As an image classification, DESH, ventricular dilatation (VD), tightened sulci in the high convexities (THC), and Sylvian fissure dilatation (SFD) were automatically assessed on the multimodal convolutional neural network (CNN) model. For both deep learning models, 110 T1- and 130 T2-weighted MRIs were used for training, 30 T1- and 30 T2-weighted MRIs for internal validation, and the remaining 19 T1- and 20 T2-weighted MRIs for external validation. Dice score was calculated as (overlapping area)\u2009\u00d7\u20092\/total area.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Automatic region extraction from 3D T1- and T2-weighted MRI was accurate for the total ventricles (mean Dice scores: 0.85 and 0.83), Sylvian fissure and basal cistern (0.70 and 0.69), and high-convexity SAS (0.68 and 0.60), respectively. Automatic determination of DESH, VD, THC, and SFD from the segmented regions on the multimodal CNN model was sufficiently reliable; all of the mean softmax probability scores were exceeded by 0.95. All of the areas under the receiver-operating characteristic curves of the DESH, Venthi, and Sylhi indexes calculated by the segmented regions for detecting DESH were exceeded by 0.97.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>Using 3D U-Net and a multimodal CNN, DESH was automatically detected with automatically segmented regions from 3D MRIs. Our developed diagnostic support tool can improve the precision of Hakim disease (iNPH) diagnosis.<\/jats:p><\/jats:sec>","DOI":"10.3389\/fnagi.2024.1362637","type":"journal-article","created":{"date-parts":[[2024,3,15]],"date-time":"2024-03-15T04:59:55Z","timestamp":1710478795000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":21,"title":["Automatic assessment of disproportionately enlarged subarachnoid-space hydrocephalus from 3D MRI using two deep learning models"],"prefix":"10.3389","volume":"16","author":[{"given":"Shigeki","family":"Yamada","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hirotaka","family":"Ito","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hironori","family":"Matsumasa","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Satoshi","family":"Ii","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tomohiro","family":"Otani","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Motoki","family":"Tanikawa","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Chifumi","family":"Iseki","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yoshiyuki","family":"Watanabe","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shigeo","family":"Wada","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marie","family":"Oshima","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mitsuhito","family":"Mase","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1965","published-online":{"date-parts":[[2024,3,15]]},"reference":[{"key":"ref1","doi-asserted-by":"publisher","first-page":"117","DOI":"10.1056\/NEJM196507152730301","article-title":"Symptomatic occult hydrocephalus with \u201cNormal\u201d cerebrospinal-fluid Pressure. A treatable syndrome","volume":"273","author":"Adams","year":"1965","journal-title":"N. Engl. J. Med."},{"key":"ref2","doi-asserted-by":"publisher","first-page":"1153","DOI":"10.1111\/ene.14671","article-title":"Early shunt surgery improves survival in idiopathic normal pressure hydrocephalus","volume":"28","author":"Andren","year":"2021","journal-title":"Eur. J. Neurol."},{"key":"ref3","doi-asserted-by":"publisher","first-page":"640","DOI":"10.1007\/s00415-019-09598-1","article-title":"Survival in treated idiopathic normal pressure hydrocephalus","volume":"267","author":"Andren","year":"2020","journal-title":"J. Neurol."},{"key":"ref4","doi-asserted-by":"publisher","first-page":"1","DOI":"10.1212\/WNL.0000000000208037","article-title":"Prevalence of possible idiopathic Normal pressure hydrocephalus in Sweden: a population-based MRI study in 791 70-year-old participants","volume":"102","author":"Constantinescu","year":"2023","journal-title":"Neurology"},{"key":"ref5","doi-asserted-by":"publisher","first-page":"3998","DOI":"10.1038\/s41598-019-39809-8","article-title":"Lifespan changes of the human brain in Alzheimer\u2019s disease","volume":"9","author":"Coupe","year":"2019","journal-title":"Sci. Rep."},{"key":"ref6","doi-asserted-by":"publisher","first-page":"2463","DOI":"10.1007\/s00701-020-04447-x","article-title":"Semantic segmentation of cerebrospinal fluid and brain volume with a convolutional neural network in pediatric hydrocephalus-transfer learning from existing algorithms","volume":"162","author":"Grimm","year":"2020","journal-title":"Acta Neurochir."},{"key":"ref7","doi-asserted-by":"publisher","first-page":"101605","DOI":"10.1016\/j.nicl.2018.11.015","article-title":"Automated detection of imaging features of disproportionately enlarged subarachnoid space hydrocephalus using machine learning methods","volume":"21","author":"Gunter","year":"2019","journal-title":"Neuroimage Clin"},{"key":"ref8","doi-asserted-by":"publisher","first-page":"18","DOI":"10.1186\/1743-8454-7-18","article-title":"Diagnosis of idiopathic normal pressure hydrocephalus is supported by MRI-based scheme: a prospective cohort study","volume":"7","author":"Hashimoto","year":"2010","journal-title":"Cerebrospinal Fluid Res."},{"key":"ref9","doi-asserted-by":"publisher","first-page":"351","DOI":"10.2463\/mrms.mp.2019-0106","article-title":"A novel deep learning approach with a 3D convolutional ladder network for differential diagnosis of idiopathic Normal pressure hydrocephalus and Alzheimer\u2019s disease","volume":"19","author":"Irie","year":"2020","journal-title":"Magn. Reson. Med. Sci."},{"key":"ref10","doi-asserted-by":"publisher","first-page":"54","DOI":"10.1016\/j.jns.2008.10.004","article-title":"Asymptomatic ventriculomegaly with features of idiopathic normal pressure hydrocephalus on MRI (AVIM) in the elderly: a prospective study in a Japanese population","volume":"277","author":"Iseki","year":"2009","journal-title":"J. Neurol. Sci."},{"key":"ref11","doi-asserted-by":"publisher","first-page":"680","DOI":"10.1111\/ane.13710","article-title":"Prevalence and development of idiopathic normal pressure hydrocephalus: a 16-year longitudinal study in Japan","volume":"146","author":"Iseki","year":"2022","journal-title":"Acta Neurol. Scand."},{"key":"ref12","doi-asserted-by":"publisher","first-page":"222","DOI":"10.2176\/nmc.44.222","article-title":"Clinical guidelines for idiopathic normal pressure hydrocephalus","volume":"44","author":"Ishikawa","year":"2004","journal-title":"Neurol. Med. Chir. (Tokyo)"},{"key":"ref13","doi-asserted-by":"publisher","first-page":"119","DOI":"10.2176\/nmc.50.119","article-title":"Clinical significance of cerebrospinal fluid tap test and magnetic resonance imaging\/computed tomography findings of tight high convexity in patients with possible idiopathic normal pressure hydrocephalus","volume":"50","author":"Ishikawa","year":"2010","journal-title":"Neurol. Med. Chir. (Tokyo)"},{"key":"ref14","doi-asserted-by":"publisher","first-page":"1449","DOI":"10.1212\/WNL.0000000000000342","article-title":"Prevalence of idiopathic normal-pressure hydrocephalus","volume":"82","author":"Jaraj","year":"2014","journal-title":"Neurology"},{"key":"ref15","doi-asserted-by":"publisher","first-page":"e00635","DOI":"10.1002\/brb3.635","article-title":"Nationwide hospital-based survey of idiopathic normal pressure hydrocephalus in Japan: epidemiological and clinical characteristics","volume":"7","author":"Kuriyama","year":"2017","journal-title":"Brain Behav."},{"key":"ref16","doi-asserted-by":"publisher","first-page":"2849","DOI":"10.1111\/ene.15904","article-title":"Brain linear measurements for differentiating normal pressure hydrocephalus from Alzheimer\u2019s disease: an exploratory study","volume":"30","author":"Luca","year":"2023","journal-title":"Eur. J. Neurol."},{"key":"ref17","doi-asserted-by":"publisher","first-page":"237","DOI":"10.1007\/3-211-32318-X_48","article-title":"Guidelines for management of idiopathic normal pressure hydrocephalus: progress to date","volume":"95","author":"Marmarou","year":"2005","journal-title":"Acta Neurochir. Suppl."},{"key":"ref18","doi-asserted-by":"publisher","first-page":"151","DOI":"10.1080\/13554794.2019.1617319","article-title":"Disproportionately enlarged subarachnoid-space hydrocephalus (DESH) in normal pressure hydrocephalus misinterpreted as atrophy: autopsy and radiological evidence","volume":"25","author":"Mccarty","year":"2019","journal-title":"Neurocase"},{"key":"ref19","doi-asserted-by":"publisher","first-page":"775","DOI":"10.2176\/nmc.52.775","article-title":"Guidelines for Management of Idiopathic Normal Pressure Hydrocephalus: second edition","volume":"52","author":"Mori","year":"2012","journal-title":"Neurol. Med. Chir. (Tokyo)"},{"key":"ref20","doi-asserted-by":"publisher","first-page":"466","DOI":"10.1002\/mdc3.13638","article-title":"Turning and sitting in early parkinsonism: differences between idiopathic Normal pressure hydrocephalus associated with parkinsonism and Parkinson\u2019s disease","volume":"10","author":"Mostile","year":"2023","journal-title":"Mov. Disord. Clin. Pract."},{"key":"ref21","doi-asserted-by":"publisher","first-page":"63","DOI":"10.2176\/nmc.st.2020-0292","article-title":"Guidelines for management of idiopathic normal pressure hydrocephalus (Third edition): endorsed by the Japanese society of normal pressure hydrocephalus","volume":"61","author":"Nakajima","year":"2021","journal-title":"Neurol Med Chir (Tokyo)"},{"key":"ref22","doi-asserted-by":"publisher","first-page":"1831","DOI":"10.3174\/ajnr.A4838","article-title":"High-convexity tightness predicts the shunt response in idiopathic normal pressure hydrocephalus","volume":"37","author":"Narita","year":"2016","journal-title":"Am. J. Neuroradiol."},{"key":"ref23","doi-asserted-by":"publisher","first-page":"74","DOI":"10.1111\/ene.13766","article-title":"Cognitive disorders in normal pressure hydrocephalus with initial parkinsonism in comparison with de novo Parkinson\u2019s disease","volume":"26","author":"Picascia","year":"2019","journal-title":"Eur. J. Neurol."},{"key":"ref24","doi-asserted-by":"publisher","first-page":"117","DOI":"10.1007\/s00234-007-0318-x","article-title":"Narrow CSF space at high convexity and high midline areas in idiopathic normal pressure hydrocephalus detected by axial and coronal MRI","volume":"50","author":"Sasaki","year":"2008","journal-title":"Neuroradiology"},{"key":"ref25","doi-asserted-by":"publisher","first-page":"1436","DOI":"10.3171\/2016.9.JNS161080","article-title":"Utility of MRI-based disproportionately enlarged subarachnoid space hydrocephalus scoring for predicting prognosis after surgery for idiopathic normal pressure hydrocephalus: clinical research","volume":"127","author":"Shinoda","year":"2017","journal-title":"J. Neurosurg."},{"key":"ref26","doi-asserted-by":"publisher","first-page":"289","DOI":"10.1136\/jnnp.70.3.289","article-title":"Comparative analysis of the gait disorder of normal pressure hydrocephalus and Parkinson\u2019s disease","volume":"70","author":"Stolze","year":"2001","journal-title":"J. Neurol. Neurosurg. Psychiatry"},{"key":"ref27","doi-asserted-by":"publisher","first-page":"1415","DOI":"10.3390\/cancers13061415","article-title":"Fine-tuning approach for segmentation of gliomas in brain magnetic resonance images with a machine learning method to normalize image differences among facilities","volume":"13","author":"Takahashi","year":"2021","journal-title":"Cancers (Basel)"},{"key":"ref28","doi-asserted-by":"publisher","first-page":"113","DOI":"10.1016\/j.wneu.2023.12.094","article-title":"Classification of chronic hydrocephalus in adults: a systematic review and analysis","volume":"183","author":"Tullberg","year":"2024","journal-title":"World Neurosurg."},{"key":"ref29","doi-asserted-by":"publisher","first-page":"90","DOI":"10.1111\/jon.12932","article-title":"Midbrain area and the hummingbird sign from brain MRI in progressive supranuclear palsy and idiopathic normal pressure hydrocephalus","volume":"32","author":"Virhammar","year":"2021","journal-title":"J. Neuroimaging"},{"key":"ref30","doi-asserted-by":"publisher","first-page":"43","DOI":"10.1038\/s41746-022-00577-x","article-title":"A high-generalizability machine learning framework for predicting the progression of Alzheimer\u2019s disease using limited data","volume":"5","author":"Wang","year":"2022","journal-title":"NPJ Digit. Med."},{"key":"ref31","doi-asserted-by":"publisher","first-page":"4454","DOI":"10.1007\/s00330-020-06825-6","article-title":"Cerebrospinal fluid dynamics in idiopathic normal pressure hydrocephalus on four-dimensional flow imaging","volume":"30","author":"Yamada","year":"2020","journal-title":"Eur. Radiol."},{"key":"ref32","doi-asserted-by":"publisher","first-page":"39070","DOI":"10.1038\/srep39070","article-title":"Choroidal fissure acts as an overflow device in cerebrospinal fluid drainage: morphological comparison between idiopathic and secondary normal-pressure hydrocephalus","volume":"6","author":"Yamada","year":"","journal-title":"Sci. Rep."},{"key":"ref33","doi-asserted-by":"publisher","first-page":"189","DOI":"10.1111\/ane.12580","article-title":"Disease duration: the key to accurate CSF tap test in iNPH","volume":"135","author":"Yamada","year":"","journal-title":"Acta Neurol. Scand."},{"key":"ref34","doi-asserted-by":"publisher","first-page":"798488","DOI":"10.3389\/fneur.2021.798488","article-title":"Reconsidering ventriculoperitoneal shunt surgery and postoperative shunt valve pressure adjustment: our approaches learned from past challenges and failures","volume":"12","author":"Yamada","year":"","journal-title":"Front. Neurol."},{"key":"ref35","doi-asserted-by":"publisher","first-page":"20","DOI":"10.1186\/s12987-021-00243-6","article-title":"Exploring mechanisms of ventricular enlargement in idiopathic normal pressure hydrocephalus: a role of cerebrospinal fluid dynamics and motile cilia","volume":"18","author":"Yamada","year":"","journal-title":"Fluids Barriers CNS"},{"key":"ref36","doi-asserted-by":"publisher","first-page":"17318","DOI":"10.1038\/s41598-019-53888-7","article-title":"Longitudinal morphological changes during recovery from brain deformation due to idiopathic normal pressure hydrocephalus after ventriculoperitoneal shunt surgery","volume":"9","author":"Yamada","year":"2019","journal-title":"Sci. Rep."},{"key":"ref37","doi-asserted-by":"publisher","first-page":"2262","DOI":"10.3174\/ajnr.A4440","article-title":"Optimal diagnostic indices for idiopathic normal pressure hydrocephalus based on the 3D quantitative volumetric analysis for the cerebral ventricle and subarachnoid space","volume":"36","author":"Yamada","year":"2015","journal-title":"AJNR Am. J. Neuroradiol."},{"key":"ref38","doi-asserted-by":"publisher","first-page":"1249","DOI":"10.3174\/ajnr.A4695","article-title":"Comparison of CSF distribution between idiopathic normal pressure hydrocephalus and Alzheimer disease","volume":"37","author":"Yamada","year":"","journal-title":"AJNR Am. J. Neuroradiol."},{"key":"ref39","doi-asserted-by":"publisher","first-page":"583","DOI":"10.3389\/fneur.2017.00583","article-title":"Fluid distribution pattern in adult-onset congenital, idiopathic and secondary normal-pressure hydrocephalus: implications for clinical care","volume":"8","author":"Yamada","year":"","journal-title":"Front. Neurol."},{"key":"ref40","doi-asserted-by":"publisher","first-page":"479","DOI":"10.3174\/ajnr.A6941","article-title":"Quantification of oscillatory shear stress from reciprocating CSF motion on 4D flow imaging","volume":"42","author":"Yamada","year":"","journal-title":"AJNR Am. J. Neuroradiol."},{"key":"ref41","doi-asserted-by":"publisher","first-page":"e427","DOI":"10.1016\/j.wneu.2023.05.077","article-title":"Tightened sulci in the high convexities as a noteworthy feature of idiopathic Normal pressure hydrocephalus","volume":"176","author":"Yamada","year":"","journal-title":"World Neurosurg."},{"key":"ref42","doi-asserted-by":"publisher","first-page":"351","DOI":"10.1016\/j.wneu.2023.07.110","article-title":"Age-related changes in cerebrospinal fluid dynamics in the pathogenesis of chronic hydrocephalus in adults","volume":"178","author":"Yamada","year":"","journal-title":"World Neurosurg"},{"key":"ref43","doi-asserted-by":"publisher","first-page":"2002","DOI":"10.3171\/2016.5.JNS16377","article-title":"Disability risk or unimproved symptoms following shunt surgery in patients with idiopathic normal-pressure hydrocephalus: post hoc analysis of SINPHONI-2","volume":"126","author":"Yamada","year":"","journal-title":"J. Neurosurg."},{"key":"ref44","doi-asserted-by":"publisher","first-page":"141","DOI":"10.2176\/jns-nmc.2022-0331","article-title":"Cerebrospinal fluid production and absorption and ventricular enlargement mechanisms in hydrocephalus","volume":"63","author":"Yamada","year":"2023","journal-title":"Neurol. Med. Chir. (Tokyo)"},{"key":"ref45","doi-asserted-by":"publisher","first-page":"7099","DOI":"10.1007\/s00330-023-09632-x","article-title":"Aging-related volume changes in the brain and cerebrospinal fluid using artificial intelligence-automated segmentation","volume":"33","author":"Yamada","year":"","journal-title":"Eur. Radiol."}],"container-title":["Frontiers in Aging Neuroscience"],"original-title":[],"link":[{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fnagi.2024.1362637\/full","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2024,3,15]],"date-time":"2024-03-15T05:00:00Z","timestamp":1710478800000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fnagi.2024.1362637\/full"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,3,15]]},"references-count":45,"alternative-id":["10.3389\/fnagi.2024.1362637"],"URL":"https:\/\/doi.org\/10.3389\/fnagi.2024.1362637","relation":{},"ISSN":["1663-4365"],"issn-type":[{"value":"1663-4365","type":"electronic"}],"subject":[],"published":{"date-parts":[[2024,3,15]]},"article-number":"1362637"}}