{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,3,6]],"date-time":"2026-03-06T07:15:37Z","timestamp":1772781337563,"version":"3.50.1"},"reference-count":38,"publisher":"Springer Science and Business Media LLC","issue":"6","license":[{"start":{"date-parts":[[2013,9,3]],"date-time":"2013-09-03T00:00:00Z","timestamp":1378166400000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/www.springer.com\/tdm"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Pediatr Res"],"published-print":{"date-parts":[[2013,12]]},"DOI":"10.1038\/pr.2013.158","type":"journal-article","created":{"date-parts":[[2013,9,3]],"date-time":"2013-09-03T15:55:52Z","timestamp":1378223752000},"page":"712-720","source":"Crossref","is-referenced-by-count":18,"title":["Immune response to intrathecal enzyme replacement therapy in mucopolysaccharidosis I patients"],"prefix":"10.1038","volume":"74","author":[{"given":"Moin","family":"Vera","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Steven","family":"Le","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Shih-hsin","family":"Kan","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hermes","family":"Garban","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"David","family":"Naylor","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Anton","family":"Mlikotic","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ilkka","family":"Kaitila","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Paul","family":"Harmatz","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Agnes","family":"Chen","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Patricia","family":"Dickson","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"297","published-online":{"date-parts":[[2013,9,3]]},"reference":[{"key":"BFpr2013158_CR1","first-page":"3421","volume-title":"The Metabolic and Molecular Bases of Inherited Disease","author":"EF Neufeld","year":"2001","unstructured":"Neufeld EF, Meunzer J . The Mucopolysaccharidoses. In: Scriver CR, Beaudet AL, Sly WS, Valle D, eds. The Metabolic and Molecular Bases of Inherited Disease. New York: McGraw-Hill, 2001:3421\u201352."},{"key":"BFpr2013158_CR2","doi-asserted-by":"publisher","first-page":"579","DOI":"10.1007\/978-3-642-15720-2_40","volume-title":"Inborn Metabolic Diseases: Diagnosis and Treatment","author":"JE Wraith","year":"2012","unstructured":"Wraith JE . Mucopolysaccharidoses and oligosaccharidoses. In: Saudubray J-M, van den Berghe G, Walter JH, eds. Inborn Metabolic Diseases: Diagnosis and Treatment, 5th ed. Berlin: Springer-Verlag, 2012:579\u201390."},{"key":"BFpr2013158_CR3","doi-asserted-by":"publisher","first-page":"182","DOI":"10.1056\/NEJM200101183440304","volume":"344","author":"ED Kakkis","year":"2001","unstructured":"Kakkis ED, Muenzer J, Tiller GE, et al. Enzyme-replacement therapy in mucopolysaccharidosis I. N Engl J Med 2001;344:182\u20138.","journal-title":"N Engl J Med"},{"key":"BFpr2013158_CR4","doi-asserted-by":"publisher","first-page":"581","DOI":"10.1016\/j.jpeds.2004.01.046","volume":"144","author":"JE Wraith","year":"2004","unstructured":"Wraith JE, Clarke LA, Beck M, et al. Enzyme replacement therapy for mucopolysaccharidosis I: a randomized, double-blinded, placebo-controlled, multinational study of recombinant human alpha-L-iduronidase (laronidase). J Pediatr 2004;144:581\u20138.","journal-title":"J Pediatr"},{"key":"BFpr2013158_CR5","doi-asserted-by":"publisher","first-page":"E12","DOI":"10.3171\/FOC\/2008\/24\/3-4\/E11","volume":"24","author":"GM Enns","year":"2008","unstructured":"Enns GM, Huhn SL . Central nervous system therapy for lysosomal storage disorders. Neurosurg Focus 2008;24:E12.","journal-title":"Neurosurg Focus"},{"key":"BFpr2013158_CR6","first-page":"223","volume":"47","author":"E Kachur","year":"2000","unstructured":"Kachur E, Del Maestro R . Mucopolysaccharidoses and spinal cord compression: case report and review of the literature with implications of bone marrow transplantation. Neurosurgery 2000;47:223\u20138; discussion 228\u20139.","journal-title":"Neurosurgery"},{"key":"BFpr2013158_CR7","doi-asserted-by":"publisher","first-page":"946","DOI":"10.2174\/138920111795542642","volume":"12","author":"PI Dickson","year":"2011","unstructured":"Dickson PI, Chen AH . Intrathecal enzyme replacement therapy for mucopolysaccharidosis I: translating success in animal models to patients. Curr Pharm Biotechnol 2011;12:946\u201355.","journal-title":"Curr Pharm Biotechnol"},{"key":"BFpr2013158_CR8","doi-asserted-by":"publisher","first-page":"194","DOI":"10.1111\/j.1469-8749.1973.tb15160.x","volume":"15","author":"P Kennedy","year":"1973","unstructured":"Kennedy P, Swash M, Dean MF . Cervical cord compression in mucopolysaccharidosis. Dev Med Child Neurol 1973;15:194\u20139.","journal-title":"Dev Med Child Neurol"},{"key":"BFpr2013158_CR9","doi-asserted-by":"publisher","first-page":"404","DOI":"10.1016\/S0090-3019(82)80004-9","volume":"17","author":"HH Kaufman","year":"1982","unstructured":"Kaufman HH, Rosenberg HS, Scott CI, Lee YY, Pruessner JL, Butler IJ . Cervical myelopathy due to dural compression in mucopolysaccharidosis. Surg Neurol 1982;17:404\u201310.","journal-title":"Surg Neurol"},{"key":"BFpr2013158_CR10","doi-asserted-by":"publisher","first-page":"2520","DOI":"10.1096\/fj.06-6169com","volume":"21","author":"WC Lee","year":"2007","unstructured":"Lee WC, Tsoi YK, Troendle FJ, et al. Single-dose intracerebroventricular administration of galactocerebrosidase improves survival in a mouse model of globoid cell leukodystrophy. FASEB J 2007;21:2520\u20137.","journal-title":"FASEB J"},{"key":"BFpr2013158_CR11","doi-asserted-by":"publisher","first-page":"349","DOI":"10.1016\/j.expneurol.2008.10.021","volume":"215","author":"JC Dodge","year":"2009","unstructured":"Dodge JC, Clarke J, Treleaven CM, et al. Intracerebroventricular infusion of acid sphingomyelinase corrects CNS manifestations in a mouse model of Niemann-Pick A disease. Exp Neurol 2009;215:349\u201357.","journal-title":"Exp Neurol"},{"key":"BFpr2013158_CR12","doi-asserted-by":"publisher","first-page":"649","DOI":"10.1038\/mt.2008.9","volume":"16","author":"M Chang","year":"2008","unstructured":"Chang M, Cooper JD, Sleat DE, et al. Intraventricular enzyme replacement improves disease phenotypes in a mouse model of late infantile neuronal ceroid lipofuscinosis. Mol Ther 2008;16:649\u201356.","journal-title":"Mol Ther"},{"key":"BFpr2013158_CR13","doi-asserted-by":"publisher","first-page":"313","DOI":"10.1016\/j.ymgme.2006.10.005","volume":"90","author":"KM Hemsley","year":"2007","unstructured":"Hemsley KM, King B, Hopwood JJ . Injection of recombinant human sulfamidase into the CSF via the cerebellomedullary cistern in MPS IIIA mice. Mol Genet Metab 2007;90:313\u201328.","journal-title":"Mol Genet Metab"},{"key":"BFpr2013158_CR14","doi-asserted-by":"publisher","first-page":"39","DOI":"10.1038\/pr.2011.13","volume":"71","author":"D Auclair","year":"2012","unstructured":"Auclair D, Finnie J, Walkley SU, et al. Intrathecal recombinant human 4-sulfatase reduces accumulation of glycosaminoglycans in dura of mucopolysaccharidosis VI cats. Pediatr Res 2012;71:39\u201345.","journal-title":"Pediatr Res"},{"key":"BFpr2013158_CR15","doi-asserted-by":"publisher","first-page":"163","DOI":"10.1016\/j.ymgme.2004.07.003","volume":"83","author":"E Kakkis","year":"2004","unstructured":"Kakkis E, McEntee M, Vogler C, et al. Intrathecal enzyme replacement therapy reduces lysosomal storage in the brain and meninges of the canine model of MPS I. Mol Genet Metab 2004;83:163\u201374.","journal-title":"Mol Genet Metab"},{"key":"BFpr2013158_CR16","doi-asserted-by":"publisher","first-page":"61","DOI":"10.1016\/j.ymgme.2006.12.012","volume":"91","author":"P Dickson","year":"2007","unstructured":"Dickson P, McEntee M, Vogler C, et al. Intrathecal enzyme replacement therapy: successful treatment of brain disease via the cerebrospinal fluid. Mol Genet Metab 2007;91:61\u20138.","journal-title":"Mol Genet Metab"},{"key":"BFpr2013158_CR17","doi-asserted-by":"publisher","first-page":"68","DOI":"10.1016\/j.ymgme.2012.02.003","volume":"106","author":"PI Dickson","year":"2012","unstructured":"Dickson PI, Ellinwood NM, Brown JR, et al. Specific antibody titer alters the effectiveness of intrathecal enzyme replacement therapy in canine mucopolysaccharidosis I. Mol Genet Metab 2012;106:68\u201372.","journal-title":"Mol Genet Metab"},{"key":"BFpr2013158_CR18","doi-asserted-by":"publisher","first-page":"901","DOI":"10.1038\/nbt.1484","volume":"26","author":"J Wang","year":"2008","unstructured":"Wang J, Lozier J, Johnson G, et al. Neutralizing antibodies to therapeutic enzymes: considerations for testing, prevention and treatment. Nat Biotechnol 2008;26:901\u20138.","journal-title":"Nat Biotechnol"},{"key":"BFpr2013158_CR19","first-page":"2868","volume":"118","author":"P Dickson","year":"2008","unstructured":"Dickson P, Peinovich M, McEntee M, et al. Immune tolerance improves the efficacy of enzyme replacement therapy in canine mucopolysaccharidosis I. J Clin Invest 2008;118:2868\u201376.","journal-title":"J Clin Invest"},{"key":"BFpr2013158_CR20","first-page":"347","volume-title":"Immunobiology: The Immune System in Health and Disease","author":"CA Janeway","year":"2005","unstructured":"Janeway CA, Travers P, Walport M, Shlomchik MJ, eds. Immunobiology: The Immune System in Health and Disease, 6th ed. New York: Garland, 2005:347\u201351."},{"key":"BFpr2013158_CR21","doi-asserted-by":"publisher","first-page":"559","DOI":"10.1007\/s00415-011-6190-y","volume":"259","author":"JE Bromberg","year":"2012","unstructured":"Bromberg JE, Doorduijn JK, Baars JW, van Imhoff GW, Enting R, van den Bent MJ . Acute painful lumbosacral paresthesia after intrathecal rituximab. J Neurol 2012;259:559\u201361.","journal-title":"J Neurol"},{"key":"BFpr2013158_CR22","doi-asserted-by":"publisher","first-page":"1185","DOI":"10.1001\/archinte.159.11.1185","volume":"159","author":"G Moris","year":"1999","unstructured":"Moris G, Garcia-Monco JC . The challenge of drug-induced aseptic meningitis. Arch Intern Med 1999;159:1185\u201394.","journal-title":"Arch Intern Med"},{"key":"BFpr2013158_CR23","doi-asserted-by":"publisher","first-page":"562","DOI":"10.1016\/j.jocn.2004.08.024","volume":"12","author":"L Kepa","year":"2005","unstructured":"Kepa L, Oczko-Grzesik B, Stolarz W, Sobala-Szczygiel B . Drug-induced aseptic meningitis in suspected central nervous system infections. J Clin Neurosci 2005;12:562\u20134.","journal-title":"J Clin Neurosci"},{"key":"BFpr2013158_CR24","doi-asserted-by":"publisher","first-page":"98","DOI":"10.1172\/JCI113317","volume":"81","author":"EH Schuchman","year":"1988","unstructured":"Schuchman EH, Desnick RJ . Mucopolysaccharidosis type I subtypes. Presence of immunologically cross-reactive material and in vitro enhancement of the residual alpha-L-iduronidase activities. J Clin Invest 1988;81:98\u2013105.","journal-title":"J Clin Invest"},{"key":"BFpr2013158_CR25","doi-asserted-by":"publisher","first-page":"105","DOI":"10.1136\/jnnp.s1-2.6.105","volume":"2","author":"JG Greenfield","year":"1921","unstructured":"Greenfield JG . Original Papers: on Froin\u2019s syndrome, and its relation to allied conditions in the cerebrospinal fluid. J Neurol Psychopathol 1921;2:105\u201341.","journal-title":"J Neurol Psychopathol"},{"key":"BFpr2013158_CR26","doi-asserted-by":"publisher","first-page":"600","DOI":"10.1016\/j.crad.2007.07.027","volume":"63","author":"S Mirza","year":"2008","unstructured":"Mirza S, Adams WM, Corkhill RA . Froin\u2019s syndrome revisited, 100 years on. Pseudo-Froin\u2019s syndrome on MRI. Clin Radiol 2008;63:600\u20134.","journal-title":"Clin Radiol"},{"key":"BFpr2013158_CR27","doi-asserted-by":"publisher","first-page":"143","DOI":"10.1111\/j.1442-200X.2010.03215.x","volume":"53","author":"T Asano","year":"2011","unstructured":"Asano T, Ichiki K, Koizumi S, et al. Enhanced expression of cytokines\/chemokines in cerebrospinal fluids in mumps meningitis in children. Pediatr Int 2011;53:143\u20136.","journal-title":"Pediatr Int"},{"key":"BFpr2013158_CR28","doi-asserted-by":"publisher","first-page":"198","DOI":"10.4269\/ajtmh.2008.78.198","volume":"78","author":"CC John","year":"2008","unstructured":"John CC, Panoskaltsis-Mortari A, Opoka RO, et al. Cerebrospinal fluid cytokine levels and cognitive impairment in cerebral malaria. Am J Trop Med Hyg 2008;78:198\u2013205.","journal-title":"Am J Trop Med Hyg"},{"key":"BFpr2013158_CR29","doi-asserted-by":"publisher","first-page":"731","DOI":"10.1016\/j.braindev.2008.11.005","volume":"31","author":"T Ichiyama","year":"2009","unstructured":"Ichiyama T, Ito Y, Kubota M, Yamazaki T, Nakamura K, Furukawa S . Serum and cerebrospinal fluid levels of cytokines in acute encephalopathy associated with human herpesvirus-6 infection. Brain Dev 2009;31:731\u20138.","journal-title":"Brain Dev"},{"key":"BFpr2013158_CR30","doi-asserted-by":"publisher","first-page":"156","DOI":"10.1016\/S1413-8670(11)70162-3","volume":"15","author":"VL Pinto Junior","year":"2011","unstructured":"Pinto Junior VL, Rebelo MC, Gomes RN, Assis EF, Castro-Faria-Neto HC, B\u00f3ia MN . IL-6 and IL-8 in cerebrospinal fluid from patients with aseptic meningitis and bacterial meningitis: their potential role as a marker for differential diagnosis. Braz J Infect Dis 2011;15:156\u20138.","journal-title":"Braz J Infect Dis"},{"key":"BFpr2013158_CR31","doi-asserted-by":"publisher","first-page":"67","DOI":"10.1016\/0165-5728(91)90073-G","volume":"32","author":"D Maimone","year":"1991","unstructured":"Maimone D, Gregory S, Arnason BG, Reder AT . Cytokine levels in the cerebrospinal fluid and serum of patients with multiple sclerosis. J Neuroimmunol 1991;32:67\u201374.","journal-title":"J Neuroimmunol"},{"key":"BFpr2013158_CR32","doi-asserted-by":"publisher","first-page":"498","DOI":"10.1177\/096120330000900704","volume":"9","author":"E Trysberg","year":"2000","unstructured":"Trysberg E, Carlsten H, Tarkowski A . Intrathecal cytokines in systemic lupus erythematosus with central nervous system involvement. Lupus 2000;9:498\u2013503.","journal-title":"Lupus"},{"key":"BFpr2013158_CR33","doi-asserted-by":"publisher","first-page":"13","DOI":"10.1016\/0304-3940(96)12706-3","volume":"211","author":"M Mogi","year":"1996","unstructured":"Mogi M, Harada M, Narabayashi H, Inagaki H, Minami M, Nagatsu T . Interleukin (IL)-1 beta, IL-2, IL-4, IL-6 and transforming growth factor-alpha levels are elevated in ventricular cerebrospinal fluid in juvenile parkinsonism and Parkinson\u2019s disease. Neurosci Lett 1996;211:13\u20136.","journal-title":"Neurosci Lett"},{"key":"BFpr2013158_CR34","doi-asserted-by":"crossref","first-page":"2994","DOI":"10.4049\/jimmunol.159.6.2994","volume":"159","author":"BB Mittleman","year":"1997","unstructured":"Mittleman BB, Castellanos FX, Jacobsen LK, Rapoport JL, Swedo SE, Shearer GM . Cerebrospinal fluid cytokines in pediatric neuropsychiatric disease. J Immunol 1997;159:2994\u20139.","journal-title":"J Immunol"},{"key":"BFpr2013158_CR35","doi-asserted-by":"publisher","first-page":"1268","DOI":"10.1136\/jnnp.2007.134528","volume":"79","author":"J Ludwig","year":"2008","unstructured":"Ludwig J, Binder A, Steinmann J, Wasner G, Baron R . Cytokine expression in serum and cerebrospinal fluid in non-inflammatory polyneuropathies. J Neurol Neurosurg Psychiatr 2008;79:1268\u201373.","journal-title":"J Neurol Neurosurg Psychiatr"},{"key":"BFpr2013158_CR36","doi-asserted-by":"publisher","first-page":"829","DOI":"10.1073\/pnas.0305480101","volume":"101","author":"E Kakkis","year":"2004","unstructured":"Kakkis E, Lester T, Yang R, et al. Successful induction of immune tolerance to enzyme replacement therapy in canine mucopolysaccharidosis I. Proc Natl Acad Sci USA 2004;101:829\u201334.","journal-title":"Proc Natl Acad Sci USA"},{"key":"BFpr2013158_CR37","doi-asserted-by":"publisher","first-page":"156","DOI":"10.1006\/bmme.1996.0044","volume":"58","author":"ED Kakkis","year":"1996","unstructured":"Kakkis ED, McEntee MF, Schmidtchen A, et al. Long-term and high-dose trials of enzyme replacement therapy in the canine model of mucopolysaccharidosis I. Biochem Mol Med 1996;58:156\u201367.","journal-title":"Biochem Mol Med"},{"key":"BFpr2013158_CR38","volume-title":"Johns Hopkins: The Harriet Lane Handbook","author":"MM Tschudy","year":"2012","unstructured":"Tschudy MM, Arcara KM, eds. Johns Hopkins: The Harriet Lane Handbook, 19th ed. Philadelphia: Mosby, 2012."}],"container-title":["Pediatric Research"],"original-title":[],"language":"en","link":[{"URL":"http:\/\/www.nature.com\/articles\/pr2013158.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"http:\/\/www.nature.com\/articles\/pr2013158","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"http:\/\/www.nature.com\/articles\/pr2013158.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,7,4]],"date-time":"2023-07-04T08:51:58Z","timestamp":1688460718000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.nature.com\/articles\/pr2013158"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2013,9,3]]},"references-count":38,"journal-issue":{"issue":"6","published-print":{"date-parts":[[2013,12]]}},"alternative-id":["BFpr2013158"],"URL":"https:\/\/doi.org\/10.1038\/pr.2013.158","relation":{},"ISSN":["0031-3998","1530-0447"],"issn-type":[{"value":"0031-3998","type":"print"},{"value":"1530-0447","type":"electronic"}],"subject":[],"published":{"date-parts":[[2013,9,3]]}}}