{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,4,17]],"date-time":"2026-04-17T13:57:07Z","timestamp":1776434227770,"version":"3.51.2"},"reference-count":43,"publisher":"Wiley","issue":"2","license":[{"start":{"date-parts":[[2025,11,19]],"date-time":"2025-11-19T00:00:00Z","timestamp":1763510400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"},{"start":{"date-parts":[[2025,11,19]],"date-time":"2025-11-19T00:00:00Z","timestamp":1763510400000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"funder":[{"DOI":"10.13039\/100000065","name":"National Institute of Neurological Disorders and Stroke","doi-asserted-by":"publisher","award":["U01NS082320"],"award-info":[{"award-number":["U01NS082320"]}],"id":[{"id":"10.13039\/100000065","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Epilepsia"],"published-print":{"date-parts":[[2026,2]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Objective<\/jats:title>\n                    <jats:p>This study was undertaken to develop a fully automated algorithm for tuber segmentation and quantification of tuber volume that performs similarly to the gold standard human neuroradiologist.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>We used brain magnetic resonance imaging (MRI) from patients with tuberous sclerosis complex (TSC) to train and validate a convolutional neural network (CNN), which was evaluated on segmentation with the Dice\u2013S\u00f8rensen similarity coefficient (DSSC) and on tuber burden quantification with Spearman correlation coefficient against a neuroradiologist's gold standard in the test set.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      We collected 263 MRIs from 196 patients (57% males) with median (25th percentile\u201375th percentile) age of 4.3 (3.0\u201310.1) years: 176 MRIs in the train set, 39 in the validation set, and 48 in the test set. The final model achieved in the test set a DSSC of .820 (95% confidence interval [CI] = .799\u2013.840) in the whole brain\u00a0and in the different lobes the following: .831 (95% CI = .804\u2013.850) in left frontal, .827 (95% CI = .799\u2013.853) in right frontal, .817 (95% CI = .779\u2013.842) in left temporal, .834 (95% CI = .812\u2013.849) in right temporal, .821 (95% CI = .783\u2013.856) in left parietal, .840 (95% CI = .810\u2013.865) in right parietal, .832 (95% CI = .808\u2013.851) in left occipital, and\u00a0.856 (95% CI = .838\u2013.871) in right occipital. CNN tuber volume quantification nearly perfectly correlated (Spearman correlation coefficient) with the neuroradiologist's across the whole brain (.984, 95% CI = .971\u2013.991) and in the different lobes:\u00a0.966 (95% CI = .940\u2013.981) in left frontal, .973 (95% CI = .952\u2013.985) in right frontal, .936 (95% CI = .888\u2013.964) in left temporal, .967 (95% CI = .942\u2013.982) in right temporal,\u00a0.989 (95% CI = .980\u2013.994) in left parietal, .983 (95% CI = .970\u2013.990) in right parietal, .992 (95% CI = .985\u2013.995) in left occipital, and .982 (95% CI = .968\u2013.990) in right occipital (all\n                      <jats:italic>p<\/jats:italic>\n                      &lt; .00001).\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Significance<\/jats:title>\n                    <jats:p>We generated, trained, validated, and made publicly available a CNN that achieves a near\u2010perfect correlation with a neuroradiologist gold standard quantification of tuber burden, allows for objective tuber segmentation, and increases rigor and reproducibility in TSC research across institutions.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1111\/epi.70007","type":"journal-article","created":{"date-parts":[[2025,11,19]],"date-time":"2025-11-19T13:43:59Z","timestamp":1763559839000},"page":"830-845","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Convolutional neural networks for automatic tuber segmentation and quantification of tuber burden in tuberous sclerosis complex"],"prefix":"10.1111","volume":"67","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-4313-002X","authenticated-orcid":false,"given":"Iv\u00e1n","family":"S\u00e1nchez Fern\u00e1ndez","sequence":"first","affiliation":[{"name":"Localization Laboratory, Division of Epilepsy and Clinical Neurophysiology, Department of Neurology, Boston Children's Hospital Harvard Medical School  Boston Massachusetts USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1544-4398","authenticated-orcid":false,"given":"Matheus D.","family":"Soldatelli","sequence":"additional","affiliation":[{"name":"Department of Radiology, Boston Children's Hospital Harvard Medical School  Boston Massachusetts USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0009-0001-6306-8163","authenticated-orcid":false,"given":"Gillian N.","family":"Miller","sequence":"additional","affiliation":[{"name":"Laboratory of Translational Neuroimaging, Boston Children's Hospital Harvard Medical School  Boston Massachusetts USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0009-0000-3463-9521","authenticated-orcid":false,"given":"Catharina F.","family":"Gout","sequence":"additional","affiliation":[{"name":"Localization Laboratory, Division of Epilepsy and Clinical Neurophysiology, Department of Neurology, Boston Children's Hospital Harvard Medical School  Boston Massachusetts USA"},{"name":"Department of Pediatrics Noordwest Ziekenhuisgroep  Alkmaar the Netherlands"},{"name":"Vrije Universiteit  Amsterdam the Netherlands"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0009-0004-2459-1885","authenticated-orcid":false,"given":"Ellen C.","family":"Broekhuizen","sequence":"additional","affiliation":[{"name":"Localization Laboratory, Division of Epilepsy and Clinical Neurophysiology, Department of Neurology, Boston Children's Hospital Harvard Medical School  Boston Massachusetts USA"},{"name":"Utrecht University  Utrecht the Netherlands"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0009-0002-2531-763X","authenticated-orcid":false,"given":"Iris C. 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