{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,24]],"date-time":"2026-08-24T23:09:43Z","timestamp":1787612983155,"version":"build-2736575974"},"reference-count":50,"publisher":"Journal of Neurosurgery Publishing Group (JNSPG)","issue":"5","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2025,5,1]]},"abstract":"<jats:sec>\n                    <jats:title>OBJECTIVE<\/jats:title>\n                    <jats:p>Intrathecal baclofen (ITB) is commonly used to treat secondary generalized dystonia. Intraventricular baclofen (IVB) has been shown to be a safe alternative treatment with low complications. The objective of this study was to report the long-term effects of IVB.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>METHODS<\/jats:title>\n                    <jats:p>This retrospective analysis included patients who underwent IVB therapy from April 2005 to June 2024. The decision to use IVB and the surgical technique have been previously described. Data collection included sex, race, etiology of dystonia, Gross Motor Functional Classification System scores, Barry-Albright Dystonia Scale (BADS) scores, Ashworth Scale scores, medical and surgical management of dystonia, follow-up duration, and complications. Patients whose IVB pump was removed within 1 year and those with less than 1 year of follow-up were excluded.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>RESULTS<\/jats:title>\n                    <jats:p>Thirty-six patients with IVB were identified, and 27 patients (median age 12.9 years) were ultimately included. The most common cause of secondary dystonia was cerebral palsy in 21 patients (78%), followed by metabolic and neurodegenerative disorders in 4 (14%), infection in 1 (4%), and severe traumatic brain injury (TBI) in 1 (4%). The follow-up duration ranged from 1.2 to 16.7 years, with a median of 7.7 years and mean of 8.4 years. The baclofen dosage was twice as high in patients with metabolic and neurodegenerative disorders compared with those with cerebral palsy, TBI, and infection, with median dosages of 1455 \u03bcg\/day and 725 \u03bcg\/day, respectively. Both posttreatment BADS and Ashworth Scale scores showed statistically significant improvement. Complications included infection that necessitated pump removal in 1 patient (4%), wound dehiscence that was treated with wound revision and antibiotics in 1 patient (4%), and hydrocephalus that required CSF diversion in 4 patients (15%). In 2 patients, IVB therapy was ineffective at controlling dystonia and, thus, was discontinued after 1.3 and 2.7 years. One patient asked for the pump to be removed after 1.2 years due to \"does not like the physical pump itself.\" Four patients (15%) developed hydrocephalus, which required CSF diversion via a ventriculoperitoneal shunt in 3 patients and a lumboperitoneal shunt in 1 patient, with no further shunt or IVB complications.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>CONCLUSIONS<\/jats:title>\n                    <jats:p>IVB is well tolerated in patients with generalized secondary dystonia refractory to conventional medical therapy, ITB, and deep brain stimulation. Positive long-term outcomes were reported in this cohort, with significant improvement in dystonia and overall complication rates similar to those reported with ITB.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.3171\/2024.12.peds24418","type":"journal-article","created":{"date-parts":[[2025,3,22]],"date-time":"2025-03-22T13:05:13Z","timestamp":1742648713000},"page":"480-485","source":"Crossref","is-referenced-by-count":2,"title":["Long-term outcomes of intraventricular baclofen therapy for medically refractory generalized secondary dystonia"],"prefix":"10.3171","volume":"35","author":[{"given":"Joyce","family":"Koueik","sequence":"first","affiliation":[{"name":"Department of Neurological Surgery, University of Wisconsin\u2013Madison, Wisconsin; and"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tenzing","family":"Lhamo","sequence":"additional","affiliation":[{"name":"Department of Neurological Surgery, University of Wisconsin\u2013Madison, Wisconsin; and"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Emily","family":"Meyer","sequence":"additional","affiliation":[{"name":"Department of Neurological Surgery, University of Wisconsin\u2013Madison, Wisconsin; and"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Susan","family":"Hubanks","sequence":"additional","affiliation":[{"name":"Department of Neurological Surgery, University of Wisconsin\u2013Madison, Wisconsin; and"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Brandon G.","family":"Rocque","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery, University of Alabama at Birmingham, Children\u2019s of Alabama, Birmingham, Alabama"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"A. Leland","family":"Albright","sequence":"additional","affiliation":[{"name":"Department of Neurological Surgery, University of Wisconsin\u2013Madison, Wisconsin; and"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1754","reference":[{"issue":"2","key":"b1","series-title":"J Mov Disord","first-page":"58","article-title":"Secondary dystonia-clinical clues and syndromic associations","volume":"2","author":"Schneider SA","year":"2009","unstructured":"Schneider SA, Bhatia KP. Secondary dystonia-clinical clues and syndromic associations. J Mov Disord. 2009;2(2):58\u201363."},{"issue":"1","key":"b2","series-title":"Expert Opin Pharmacother","first-page":"5","article-title":"Treatment strategies for dystonia","volume":"11","author":"Cloud LJ","year":"2010","unstructured":"Cloud LJ, Jinnah HA. Treatment strategies for dystonia. Expert Opin Pharmacother. 2010;11(1):5\u201315."},{"issue":"2","key":"b3","series-title":"Exp Neurol","first-page":"165","article-title":"Short-term effects of intrathecal baclofen in spasticity","volume":"103","author":"Latash ML","year":"1989","unstructured":"Latash ML, Penn RD, Corcos DM, Gottlieb GL. Short-term effects of intrathecal baclofen in spasticity. Exp Neurol. 1989;103(2):165\u2013172."},{"issue":"2","key":"b4","series-title":"Neurol Clin","first-page":"439","article-title":"Drug pumps for treatment of neurologic diseases and pain","volume":"3","author":"Penn RD","year":"1985","unstructured":"Penn RD. Drug pumps for treatment of neurologic diseases and pain. Neurol Clin. 1985;3(2):439\u2013451."},{"issue":"8447","key":"b5","series-title":"Lancet","first-page":"125","article-title":"Continuous intrathecal baclofen for severe spasticity","volume":"2","author":"Penn RD","year":"1985","unstructured":"Penn RD, Kroin JS. Continuous intrathecal baclofen for severe spasticity. Lancet. 1985;2(8447):125\u2013127."},{"issue":"23","key":"b6","series-title":"N Engl J Med","first-page":"1517","article-title":"Intrathecal baclofen for severe spinal spasticity","volume":"320","author":"Penn RD","year":"1989","unstructured":"Penn RD, Savoy SM, Corcos D,  Intrathecal baclofen for severe spinal spasticity. N Engl J Med. 1989;320(23):1517\u20131521."},{"issue":"1 Suppl","key":"b7","series-title":"J Neurosurg","first-page":"71","article-title":"Intraventricular baclofen infusion for dystonia. Report of two cases","volume":"105","author":"Albright AL","year":"2006","unstructured":"Albright AL. Intraventricular baclofen infusion for dystonia. Report of two cases. J Neurosurg. 2006;105(1 Suppl):71\u201374."},{"issue":"1","key":"b8","series-title":"J Neurosurg Pediatr","first-page":"11","article-title":"Intraventricular baclofen for dystonia: techniques and outcomes","volume":"3","author":"Albright AL","year":"2009","unstructured":"Albright AL, Ferson SS. Intraventricular baclofen for dystonia: techniques and outcomes. J Neurosurg Pediatr. 2009;3(1):11\u201314."},{"issue":"1","key":"b9","series-title":"Dev Med Child Neurol","first-page":"49","article-title":"Deep brain stimulation for pediatric dystonia: a meta-analysis with individual participant data","volume":"61","author":"Elkaim LM","year":"2019","unstructured":"Elkaim LM, Alotaibi NM, Sigal A,  Deep brain stimulation for pediatric dystonia: a meta-analysis with individual participant data. Dev Med Child Neurol. 2019;61(1):49\u201356."},{"issue":"2 Suppl Operative","key":"b10","series-title":"Neurosurgery","first-page":"321","article-title":"Intraventricular vs intrathecal baclofen for secondary dystonia: a comparison of complications","volume":"70","author":"Rocque BG","year":"2012","unstructured":"Rocque BG, Albright AL. Intraventricular vs intrathecal baclofen for secondary dystonia: a comparison of complications. 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Deep brain stimulation and intrathecal\/intraventricular baclofen for glutaric aciduria type 1: a scoping review, individual patient data analysis, and clinical trials review. J Inherit Metab Dis. 2023;46(4):543\u2013553."},{"issue":"3","key":"b13","series-title":"Childs Nerv Syst","first-page":"895","article-title":"Intraventricular baclofen for palliative management of acquired generalized dystonia in pediatric patients: a case series and literature review","volume":"40","author":"Krouma M","year":"2024","unstructured":"Krouma M, Soilhi AA, Desnous B, James S, Boulay C, Scavarda D. Intraventricular baclofen for palliative management of acquired generalized dystonia in pediatric patients: a case series and literature review. 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Intraventricular baclofen for treatment of severe dystonia associated with glutaryl-CoA dehydrogenase deficiency (GA1): report of two cases. Mov Disord Clin Pract (Hoboken). 2016;3(3):296\u2013299."},{"issue":"3","key":"b16","series-title":"Mov Disord Clin Pract (Hoboken)","first-page":"456","article-title":"Intraventricular baclofen following deep brain stimulation in a child with refractory status dystonicus","volume":"8","author":"Mingbunjerdsuk D","year":"2021","unstructured":"Mingbunjerdsuk D, Blume H, Browd S, Samii A. Intraventricular baclofen following deep brain stimulation in a child with refractory status dystonicus. Mov Disord Clin Pract (Hoboken). 2021;8(3):456\u2013459."},{"issue":"1","key":"b17","series-title":"Neurosurgery","first-page":"93","article-title":"Occult hydrocephalus in children with cerebral palsy","volume":"56","author":"Albright AL","year":"2005","unstructured":"Albright AL, Ferson S, Carlos S. Occult hydrocephalus in children with cerebral palsy. 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Mov Disord. 2011;26(9):1748\u20131751."},{"issue":"6","key":"b20","series-title":"J Neurosurg Pediatr","first-page":"585","article-title":"Deep brain stimulation for the treatment of childhood dystonic cerebral palsy","volume":"14","author":"Keen JR","year":"2014","unstructured":"Keen JR, Przekop A, Olaya JE, Zouros A, Hsu FP. Deep brain stimulation for the treatment of childhood dystonic cerebral palsy. J Neurosurg Pediatr. 2014;14(6):585\u2013593."},{"issue":"1","key":"b21","series-title":"Dev Med Child Neurol","first-page":"28","article-title":"Deep brain stimulation for cerebral palsy: where are we now?","volume":"62","author":"Sanger TD","year":"2020","unstructured":"Sanger TD. Deep brain stimulation for cerebral palsy: where are we now?. Dev Med Child Neurol. 2020;62(1):28\u201333."},{"issue":"e550","key":"b22","series-title":"World Neurosurg","first-page":"e556","article-title":"Intrathecal baclofen pump versus globus pallidus interna deep brain stimulation in adult patients with severe cerebral palsy","volume":"126","author":"Kim JH","year":"2019","unstructured":"Kim JH, Jung NY, Chang WS, Jung HH, Cho SR, Chang JW. Intrathecal baclofen pump versus globus pallidus interna deep brain stimulation in adult patients with severe cerebral palsy. World Neurosurg. 2019;126(e550):e556."},{"issue":"3","key":"b23","series-title":"J Neurosurg","first-page":"605","article-title":"Cerebellar deep brain stimulation for the treatment of movement disorders in cerebral palsy","volume":"139","author":"Cajigas I","year":"2023","unstructured":"Cajigas I, Morrison MA, Luciano MS, Starr PA. Cerebellar deep brain stimulation for the treatment of movement disorders in cerebral palsy. J Neurosurg. 2023;139(3):605\u2013614."},{"issue":"3","key":"b24","series-title":"Neuromodulation","first-page":"266","article-title":"Rate of complications among the recipients of intrathecal baclofen pump in Japan: a multicenter study","volume":"16","author":"Taira T","year":"2013","unstructured":"Taira T, Ueta T, Katayama Y,  Rate of complications among the recipients of intrathecal baclofen pump in Japan: a multicenter study. Neuromodulation. 2013;16(3):266\u2013272."},{"issue":"6","key":"b25","series-title":"Neurosurg Focus","first-page":"E12","article-title":"Uncovering the rate and risk factors of intrathecal baclofen pump-associated complications in the adult population","volume":"56","author":"Tiefenbach J","year":"2024","unstructured":"Tiefenbach J, Machado AG, Bethoux F. Uncovering the rate and risk factors of intrathecal baclofen pump-associated complications in the adult population. Neurosurg Focus. 2024;56(6):E12."},{"issue":"2","key":"b1","series-title":"J Mov Disord","first-page":"58","article-title":"Secondary dystonia-clinical clues and syndromic associations","volume":"2","author":"Schneider SA","year":"2009","unstructured":"Schneider SA, Bhatia KP. Secondary dystonia-clinical clues and syndromic associations. J Mov Disord. 2009;2(2):58\u201363."},{"issue":"1","key":"b2","series-title":"Expert Opin Pharmacother","first-page":"5","article-title":"Treatment strategies for dystonia","volume":"11","author":"Cloud LJ","year":"2010","unstructured":"Cloud LJ, Jinnah HA. Treatment strategies for dystonia. Expert Opin Pharmacother. 2010;11(1):5\u201315."},{"issue":"2","key":"b3","series-title":"Exp Neurol","first-page":"165","article-title":"Short-term effects of intrathecal baclofen in spasticity","volume":"103","author":"Latash ML","year":"1989","unstructured":"Latash ML, Penn RD, Corcos DM, Gottlieb GL. Short-term effects of intrathecal baclofen in spasticity. 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N Engl J Med. 1989;320(23):1517\u20131521."},{"issue":"1 Suppl","key":"b7","series-title":"J Neurosurg","first-page":"71","article-title":"Intraventricular baclofen infusion for dystonia. Report of two cases","volume":"105","author":"Albright AL","year":"2006","unstructured":"Albright AL. Intraventricular baclofen infusion for dystonia. Report of two cases. J Neurosurg. 2006;105(1 Suppl):71\u201374."},{"issue":"1","key":"b8","series-title":"J Neurosurg Pediatr","first-page":"11","article-title":"Intraventricular baclofen for dystonia: techniques and outcomes","volume":"3","author":"Albright AL","year":"2009","unstructured":"Albright AL, Ferson SS. Intraventricular baclofen for dystonia: techniques and outcomes. 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Deep brain stimulation and intrathecal\/intraventricular baclofen for glutaric aciduria type 1: a scoping review, individual patient data analysis, and clinical trials review. J Inherit Metab Dis. 2023;46(4):543\u2013553."},{"issue":"3","key":"b13","series-title":"Childs Nerv Syst","first-page":"895","article-title":"Intraventricular baclofen for palliative management of acquired generalized dystonia in pediatric patients: a case series and literature review","volume":"40","author":"Krouma M","year":"2024","unstructured":"Krouma M, Soilhi AA, Desnous B, James S, Boulay C, Scavarda D. Intraventricular baclofen for palliative management of acquired generalized dystonia in pediatric patients: a case series and literature review. 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Intraventricular baclofen for treatment of severe dystonia associated with glutaryl-CoA dehydrogenase deficiency (GA1): report of two cases. Mov Disord Clin Pract (Hoboken). 2016;3(3):296\u2013299."},{"issue":"3","key":"b16","series-title":"Mov Disord Clin Pract (Hoboken)","first-page":"456","article-title":"Intraventricular baclofen following deep brain stimulation in a child with refractory status dystonicus","volume":"8","author":"Mingbunjerdsuk D","year":"2021","unstructured":"Mingbunjerdsuk D, Blume H, Browd S, Samii A. Intraventricular baclofen following deep brain stimulation in a child with refractory status dystonicus. Mov Disord Clin Pract (Hoboken). 2021;8(3):456\u2013459."},{"issue":"1","key":"b17","series-title":"Neurosurgery","first-page":"93","article-title":"Occult hydrocephalus in children with cerebral palsy","volume":"56","author":"Albright AL","year":"2005","unstructured":"Albright AL, Ferson S, Carlos S. Occult hydrocephalus in children with cerebral palsy. Neurosurgery. 2005;56(1):93\u201397."},{"issue":"7","key":"b18","series-title":"Childs Nerv Syst","first-page":"733","article-title":"Interaction of ventriculoperitoneal shunt and baclofen pump","volume":"23","author":"Fulkerson DH","year":"2007","unstructured":"Fulkerson DH, Boaz JC, Luerssen TG. Interaction of ventriculoperitoneal shunt and baclofen pump. Childs Nerv Syst. 2007;23(7):733\u2013738."},{"issue":"9","key":"b19","series-title":"Mov Disord","first-page":"1748","article-title":"Dystonia due to cerebral palsy responds to deep brain stimulation of the globus pallidus internus","volume":"26","author":"Marks WA","year":"2011","unstructured":"Marks WA, Honeycutt J, Acosta F Jr,  Dystonia due to cerebral palsy responds to deep brain stimulation of the globus pallidus internus. Mov Disord. 2011;26(9):1748\u20131751."},{"issue":"6","key":"b20","series-title":"J Neurosurg Pediatr","first-page":"585","article-title":"Deep brain stimulation for the treatment of childhood dystonic cerebral palsy","volume":"14","author":"Keen JR","year":"2014","unstructured":"Keen JR, Przekop A, Olaya JE, Zouros A, Hsu FP. Deep brain stimulation for the treatment of childhood dystonic cerebral palsy. J Neurosurg Pediatr. 2014;14(6):585\u2013593."},{"issue":"1","key":"b21","series-title":"Dev Med Child Neurol","first-page":"28","article-title":"Deep brain stimulation for cerebral palsy: where are we now?","volume":"62","author":"Sanger TD","year":"2020","unstructured":"Sanger TD. Deep brain stimulation for cerebral palsy: where are we now?. 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