{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,29]],"date-time":"2026-09-29T09:43:09Z","timestamp":1790674989709,"version":"4.1.0"},"reference-count":38,"publisher":"Journal of Neurosurgery Publishing Group (JNSPG)","issue":"4","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2024,10,1]]},"abstract":"<jats:sec>\n<jats:title>OBJECTIVE<\/jats:title>\n<jats:p>The authors evaluated the impact of the timing of epilepsy surgery on postoperative neurocognitive outcomes in a cohort of children followed in the multiinstitutional Tuberous Sclerosis Complex (TSC) Autism Center of Excellence Research Network (TACERN) study.<\/jats:p>\n<\/jats:sec>\n<jats:sec>\n<jats:title>METHODS<\/jats:title>\n<jats:p>Twenty-seven of 159 patients in the TACERN cohort had drug-refractory epilepsy and underwent surgery. Ages at surgery ranged from 15.86 to 154.14 weeks (median 91.93 weeks). Changes in patients\u2019 first preoperative (10\u201358 weeks) to last postoperative (155\u2013188 weeks) scores on three neuropsychological tests\u2014the Mullen Scales of Early Learning (MSEL), the Vineland Adaptive Behavior Scales, 2nd edition (VABS-2), and the Preschool Language Scales, 5th edition (PLS-5)\u2014were calculated. Pearson correlation and multivariate linear regression models were used to correlate test outcomes separately with age at surgery and duration of epilepsy prior to surgery. Analyses were separately conducted for patients whose seizure burdens decreased postoperatively (n = 21) and those whose seizure burdens did not (n = 6). Regression analysis was specifically focused on the 21 patients who achieved successful seizure control.<\/jats:p>\n<\/jats:sec>\n<jats:sec>\n<jats:title>RESULTS<\/jats:title>\n<jats:p>Age at surgery was significantly negatively correlated with the change in the combined verbal subtests of the MSEL (R = \u22120.45, p = 0.039) and predicted this score in a multivariate linear regression model (\u03b2 = \u22120.09, p = 0.035). Similar trends were seen in the total language score of the PLS-5 (R = \u22120.4, p = 0.089; \u03b2 = \u22120.12, p = 0.014) and in analyses examining the duration of epilepsy prior to surgery as the independent variable of interest. Associations between age at surgery and duration of epilepsy prior to surgery with changes in the verbal subscores of VABS-2 were more variable (R = \u22120.15, p = 0.52; \u03b2 = \u22120.05, p = 0.482).<\/jats:p>\n<\/jats:sec>\n<jats:sec>\n<jats:title>CONCLUSIONS<\/jats:title>\n<jats:p>Earlier surgery and shorter epilepsy duration prior to surgery were associated with greater improvement in postoperative language in patients with TSC. Prospective or comparative effectiveness clinical trials are needed to further elucidate surgical timing impacts on neurocognitive outcomes.<\/jats:p>\n<\/jats:sec>","DOI":"10.3171\/2024.4.peds2481","type":"journal-article","created":{"date-parts":[[2024,7,13]],"date-time":"2024-07-13T02:11:25Z","timestamp":1720836685000},"page":"384-392","source":"Crossref","is-referenced-by-count":6,"title":["Association of earlier surgery with improved postoperative language development in children with tuberous sclerosis complex"],"prefix":"10.3171","volume":"34","author":[{"given":"Sina","family":"Sadeghzadeh","sequence":"first","affiliation":[{"name":"Departments of Neurosurgery and"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Thomas M.","family":"Johnstone","sequence":"additional","affiliation":[{"name":"Departments of Neurosurgery and"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jurriaan M.","family":"Peters","sequence":"additional","affiliation":[{"name":"Department of Neurology, Harvard Medical School, Boston, Massachusetts; and"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Brenda E.","family":"Porter","sequence":"additional","affiliation":[{"name":"Neurology, Stanford University School of Medicine, Stanford, California;"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"S. Katie Z.","family":"Ihnen","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Division of Neurology, Cincinnati Children\u2019s Hospital Medical Center, University of Cincinnati College of Medicine, Cincinnati, Ohio"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1754","reference":[{"issue":"1","key":"b1","series-title":"Orphanet J Rare Dis","first-page":"23","article-title":"A systematic review on the burden of illness in individuals with tuberous sclerosis complex (TSC)","volume":"15","author":"Z\u00f6llner JP","year":"2020","unstructured":"Z\u00f6llner JP, Franz DN, Hertzberg C,  A systematic review on the burden of illness in individuals with tuberous sclerosis complex (TSC). Orphanet J Rare Dis. 2020;15(1):23."},{"issue":"7","key":"b2","series-title":"Epilepsia","first-page":"1236","article-title":"The natural history of epilepsy in tuberous sclerosis complex","volume":"51","author":"Chu-Shore CJ","year":"2010","unstructured":"Chu-Shore CJ, Major P, Camposano S, Muzykewicz D, Thiele EA. The natural history of epilepsy in tuberous sclerosis complex. Epilepsia. 2010;51(7):1236\u20131241."},{"issue":"12","key":"b3","series-title":"Epilepsia","first-page":"2134","article-title":"Epilepsy in TSC: certain etiology does not mean certain prognosis","volume":"54","author":"Vignoli A","year":"2013","unstructured":"Vignoli A, La Briola F, Turner K,  Epilepsy in TSC: certain etiology does not mean certain prognosis. Epilepsia. 2013;54(12):2134\u20132142."},{"key":"b4","series-title":"Pediatr Neurol","first-page":"1","article-title":"Epilepsy is heterogeneous in early-life tuberous sclerosis complex","volume":"123","author":"Ihnen SKZ","year":"2021","unstructured":"Ihnen SKZ, Capal JK, Horn PS,  Epilepsy is heterogeneous in early-life tuberous sclerosis complex. Pediatr Neurol. 2021;123:1\u20139."},{"issue":"3","key":"b5","series-title":"Rev Neurol (Paris)","first-page":"163","article-title":"Resective surgery in tuberous sclerosis complex, from Penfield to 2018: a critical review","volume":"175","author":"Ostrowsky-Coste K","year":"2019","unstructured":"Ostrowsky-Coste K, Neal A, Guenot M,  Resective surgery in tuberous sclerosis complex, from Penfield to 2018: a critical review. Rev Neurol (Paris). 2019;175(3):163\u2013182."},{"issue":"1","key":"b6","series-title":"Epilepsia Open","first-page":"73","article-title":"Epilepsy in tuberous sclerosis complex: findings from the TOSCA study","volume":"4","author":"Nabbout R","year":"2018","unstructured":"Nabbout R, Belousova E, Benedik MP,  Epilepsy in tuberous sclerosis complex: findings from the TOSCA study. Epilepsia Open. 2018;4(1):73\u201384."},{"issue":"3","key":"b7","series-title":"Dev Med Child Neurol","first-page":"322","article-title":"Long-term cognitive outcomes in tuberous sclerosis complex","volume":"62","author":"Tye C","year":"2020","unstructured":"Tye C, Mcewen FS, Liang H,  Long-term cognitive outcomes in tuberous sclerosis complex. Dev Med Child Neurol. 2020;62(3):322\u2013329."},{"key":"b8","series-title":"Front Genet","first-page":"997461","article-title":"Shared mechanisms of neural circuit disruption in tuberous sclerosis across lifespan: bridging neurodevelopmental and neurodegenerative pathology","volume":"13","author":"Litwa K","year":"2022","unstructured":"Litwa K. Shared mechanisms of neural circuit disruption in tuberous sclerosis across lifespan: bridging neurodevelopmental and neurodegenerative pathology. Front Genet. 2022;13:997461."},{"key":"b9","series-title":"Pediatr Neurol","first-page":"50","article-title":"Updated international tuberous sclerosis complex diagnostic criteria and surveillance and management recommendations","volume":"123","author":"Northrup H","year":"2021","unstructured":"Northrup H, Aronow ME, Bebin EM,  Updated international tuberous sclerosis complex diagnostic criteria and surveillance and management recommendations. Pediatr Neurol. 2021;123:50\u201366."},{"key":"b10","series-title":"Pediatr Neurol","first-page":"77","article-title":"Beyond the guidelines: how we can improve healthcare for people with tuberous sclerosis complex around the world","volume":"123","author":"Stuart C","year":"2021","unstructured":"Stuart C, Fladrowski C, Flinn J,  Beyond the guidelines: how we can improve healthcare for people with tuberous sclerosis complex around the world. Pediatr Neurol. 2021;123:77\u201384."},{"issue":"9","key":"b11","series-title":"Epilepsia","first-page":"1206","article-title":"Epilepsy surgery in children with tuberous sclerosis complex: presurgical evaluation and outcome","volume":"41","author":"Koh S","year":"2000","unstructured":"Koh S, Jayakar P, Dunoyer C,  Epilepsy surgery in children with tuberous sclerosis complex: presurgical evaluation and outcome. Epilepsia. 2000;41(9):1206\u20131213."},{"issue":"5","key":"b12","series-title":"Neurology","first-page":"392","article-title":"Noninvasive testing, early surgery, and seizure freedom in tuberous sclerosis complex","volume":"74","author":"Wu JY","year":"2010","unstructured":"Wu JY, Salamon N, Kirsch HE,  Noninvasive testing, early surgery, and seizure freedom in tuberous sclerosis complex. Neurology. 2010;74(5):392\u2013398."},{"issue":"1","key":"b13","series-title":"J Neurosurg Pediatr","first-page":"26","article-title":"Long-term outcomes of resective epilepsy surgery after invasive presurgical evaluation in children with tuberous sclerosis complex and bilateral multiple lesions","volume":"15","author":"Arya R","year":"2015","unstructured":"Arya R, Tenney JR, Horn PS,  Long-term outcomes of resective epilepsy surgery after invasive presurgical evaluation in children with tuberous sclerosis complex and bilateral multiple lesions. J Neurosurg Pediatr. 2015;15(1):26\u201333."},{"key":"b14","series-title":"Seizure","first-page":"71","article-title":"Refractory epilepsy in preschool children with tuberous sclerosis complex: early surgical treatment and outcome","volume":"60","author":"Fohlen M","year":"2018","unstructured":"Fohlen M, Taussig D, Ferrand-Sorbets S,  Refractory epilepsy in preschool children with tuberous sclerosis complex: early surgical treatment and outcome. Seizure. 2018;60:71\u201379."},{"issue":"10","key":"b15","series-title":"Childs Nerv Syst","first-page":"2519","article-title":"Epilepsy surgery in tuberous sclerosis complex (TSC): emerging techniques and redefinition of treatment goals","volume":"36","author":"Treiber JM","year":"2020","unstructured":"Treiber JM, Curry DJ, Weiner HL, Roth J. Epilepsy surgery in tuberous sclerosis complex (TSC): emerging techniques and redefinition of treatment goals. Childs Nerv Syst. 2020;36(10):2519\u20132525."},{"key":"b16","series-title":"Pediatr Neurol","first-page":"39","article-title":"Pilot study of neurodevelopmental impact of early epilepsy surgery in tuberous sclerosis complex","volume":"109","author":"Grayson LE","year":"2020","unstructured":"Grayson LE, Peters JM, McPherson T,  Pilot study of neurodevelopmental impact of early epilepsy surgery in tuberous sclerosis complex. Pediatr Neurol. 2020;109:39\u201346."},{"key":"b17","article-title":"Mullen Scales of Early Learning Manual","author":"Mullen EM","year":"1995","unstructured":"Mullen EM. Mullen Scales of Early Learning Manual. American Guidance Service; 1995."},{"key":"b18","article-title":"Preschool Language Scales","author":"Zimmerman I","year":"2011","unstructured":"Zimmerman I, Steiner V, Pond RE. Preschool Language Scales. 5th ed. Pearson;2011."},{"key":"b19","first-page":"10","article-title":"Vineland Adaptive Behavior Scales II: Survey Interview Form\/Caregiver Rating Form","author":"Sparrow SS","year":"2005","unstructured":"Sparrow SS, Cicchetti DV, Balla DA. Vineland Adaptive Behavior Scales II: Survey Interview Form\/Caregiver Rating Form. Pearson Assessments; 2005:10."},{"issue":"Pt A","key":"b20","series-title":"Epilepsy Behav","first-page":"245","article-title":"Influence of seizures on early development in tuberous sclerosis complex","volume":"70","author":"Capal JK","year":"2017","unstructured":"Capal JK, Bernardino-Cuesta B, Horn PS,  Influence of seizures on early development in tuberous sclerosis complex. Epilepsy Behav. 2017;70(Pt A):245\u201352."},{"issue":"2","key":"b21","series-title":"Neurology","first-page":"160","article-title":"Early developmental trajectories associated with ASD in infants with tuberous sclerosis complex","volume":"83","author":"Spurling Jeste S","year":"2014","unstructured":"Spurling Jeste S, Wu JY, Senturk D,  Early developmental trajectories associated with ASD in infants with tuberous sclerosis complex. Neurology. 2014;83(2):160\u2013168."},{"issue":"4","key":"b22","series-title":"Ann Neurol","first-page":"726","article-title":"Tuber locations associated with infantile spasms map to a common brain network","volume":"89","author":"Cohen AL","year":"2021","unstructured":"Cohen AL, Mulder BPF, Prohl AK,  Tuber locations associated with infantile spasms map to a common brain network. Ann Neurol. 2021;89(4):726\u2013739."},{"issue":"11","key":"b23","series-title":"Psychol Med","first-page":"2321","article-title":"Intellectual abilities in tuberous sclerosis complex: risk factors and correlates from the Tuberous Sclerosis 2000 Study","volume":"45","author":"Bolton PF","year":"2015","unstructured":"Bolton PF, Clifford M, Tye C,  Intellectual abilities in tuberous sclerosis complex: risk factors and correlates from the Tuberous Sclerosis 2000 Study. Psychol Med. 2015;45(11):2321\u20132331."},{"issue":"12","key":"b24","series-title":"Neurology","first-page":"916","article-title":"Cognitive impairment in tuberous sclerosis complex is a multifactorial condition","volume":"70","author":"Jansen FE","year":"2008","unstructured":"Jansen FE, Vincken KL, Algra A,  Cognitive impairment in tuberous sclerosis complex is a multifactorial condition. Neurology. 2008;70(12):916\u2013923."},{"issue":"2","key":"b25","series-title":"Psychol Med","first-page":"335","article-title":"Learning disability and epilepsy in an epidemiological sample of individuals with tuberous sclerosis complex","volume":"33","author":"Joinson C","year":"2003","unstructured":"Joinson C, O\u2019Callaghan FJ, Osborne JP, Martyn C, Harris T, Bolton PF. Learning disability and epilepsy in an epidemiological sample of individuals with tuberous sclerosis complex. Psychol Med. 2003;33(2):335\u2013344."},{"key":"b26","series-title":"Epilepsy Behav","first-page":"108210","article-title":"Epilepsy associated with tuberous sclerosis complex in childhood: long-term outcome in children after epilepsy surgery and children non-eligible for epilepsy surgery","volume":"122","author":"Stomberg M","year":"2021","unstructured":"Stomberg M, Bien CG, Kalbhenn T, Polster T, May TW. Epilepsy associated with tuberous sclerosis complex in childhood: long-term outcome in children after epilepsy surgery and children non-eligible for epilepsy surgery. Epilepsy Behav. 2021;122:108210."},{"issue":"6","key":"b27","series-title":"J Neurol","first-page":"1146","article-title":"Long-term outcomes of epilepsy surgery in tuberous sclerosis complex","volume":"264","author":"Liang S","year":"2017","unstructured":"Liang S, Zhang J, Yang Z,  Long-term outcomes of epilepsy surgery in tuberous sclerosis complex. J Neurol. 2017;264(6):1146\u20131154."},{"key":"b28","series-title":"J Neurol Sci","first-page":"117506","article-title":"Seizure outcome after epilepsy surgery in tuberous sclerosis complex: results and analysis of predictors from a multicenter study","volume":"427","author":"Vannicola C","year":"2021","unstructured":"Vannicola C, Tassi L, Barba C,  Seizure outcome after epilepsy surgery in tuberous sclerosis complex: results and analysis of predictors from a multicenter study. J Neurol Sci. 2021;427:117506."},{"issue":"4","key":"b29","series-title":"Am J Intellect Dev Disabil","first-page":"291","article-title":"The construct of adaptive behavior: its conceptualization, measurement, and use in the field of intellectual disability","volume":"117","author":"Tass\u00e9 MJ","year":"2012","unstructured":"Tass\u00e9 MJ, Schalock RL, Balboni G,  The construct of adaptive behavior: its conceptualization, measurement, and use in the field of intellectual disability. Am J Intellect Dev Disabil. 2012;117(4):291\u2013303."},{"issue":"1","key":"b30","series-title":"J Spec Educ","first-page":"37","article-title":"Research with adaptive behavior scales","volume":"21","author":"Harrison PL","year":"1987","unstructured":"Harrison PL. Research with adaptive behavior scales. J Spec Educ. 1987;21(1):37\u201368."},{"issue":"1","key":"b31","series-title":"J Appl Sch Psychol","first-page":"83","article-title":"A systematic review and psychometric evaluation of adaptive behavior scales and recommendations for practice","volume":"31","author":"Floyd RG","year":"2015","unstructured":"Floyd RG, Shands EI, Alfonso VC,  A systematic review and psychometric evaluation of adaptive behavior scales and recommendations for practice. J Appl Sch Psychol. 2015;31(1):83\u2013113."},{"issue":"1","key":"b32","series-title":"Epilepsia","first-page":"81","article-title":"Epileptogenicity in tuberous sclerosis complex: a stereoelectroencephalographic study","volume":"61","author":"Neal A","year":"2020","unstructured":"Neal A, Ostrowsky-Coste K, Jung J,  Epileptogenicity in tuberous sclerosis complex: a stereoelectroencephalographic study. Epilepsia. 2020;61(1):81\u201395."},{"issue":"4","key":"b33","series-title":"Ann Neurol","first-page":"478","article-title":"mTOR cascade activation distinguishes tubers from focal cortical dysplasia","volume":"56","author":"Baybis M","year":"2004","unstructured":"Baybis M, Yu J, Lee A,  mTOR cascade activation distinguishes tubers from focal cortical dysplasia. Ann Neurol. 2004;56(4):478\u2013487."},{"issue":"11","key":"b34","series-title":"Childs Nerv Syst","first-page":"1885","article-title":"Cognitive and epilepsy outcomes after epilepsy surgery caused by focal cortical dysplasia in children: early intervention maybe better","volume":"30","author":"Chen HH","year":"2014","unstructured":"Chen HH, Chen C, Hung SC,  Cognitive and epilepsy outcomes after epilepsy surgery caused by focal cortical dysplasia in children: early intervention maybe better. 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