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Iduronate-2-Sulfatase with Anti-human Transferrin Receptor Antibody for Neuropathic Mucopolysaccharidosis II: A Phase 1/2 Trial.
Okuyama T, Eto Y, Sakai N, Minami K, Yamamoto T, Sonoda H, Yamaoka M, Tachibana K, Hirato T, Sato Y. Okuyama T, et al. Mol Ther. 2019 Feb 6;27(2):456-464. doi: 10.1016/j.ymthe.2018.12.005. Epub 2018 Dec 8. Mol Ther. 2019. PMID: 30595526 Free PMC article. Clinical Trial.
Hunter syndrome (mucopolysaccharidosis II [MPS II]), a deficiency of iduronate-2-sulfatase (IDS), causes an accumulation of glycosaminoglycans, giving rise to multiple systemic and CNS symptoms. The currently available therapies, idursulfase and idursulfase beta, ar …
Hunter syndrome (mucopolysaccharidosis II [MPS II]), a deficiency of iduronate-2-sulfatase (IDS), causes an accumulation of glycosaminoglyca …
A Phase 2/3 Trial of Pabinafusp Alfa, IDS Fused with Anti-Human Transferrin Receptor Antibody, Targeting Neurodegeneration in MPS-II.
Okuyama T, Eto Y, Sakai N, Nakamura K, Yamamoto T, Yamaoka M, Ikeda T, So S, Tanizawa K, Sonoda H, Sato Y. Okuyama T, et al. Mol Ther. 2021 Feb 3;29(2):671-679. doi: 10.1016/j.ymthe.2020.09.039. Epub 2020 Sep 30. Mol Ther. 2021. PMID: 33038326 Free article. Clinical Trial.
Serum HS and DS concentrations, liver and spleen volumes, and other assessments suggested the peripheral efficacy of pabinafusp alfa was comparable to that of idursulfase. Drug-related adverse events were mild or moderate in severity, transient, and manageable. ...
Serum HS and DS concentrations, liver and spleen volumes, and other assessments suggested the peripheral efficacy of pabinafusp alfa was com …
Phase I/II clinical trial of enzyme replacement therapy with idursulfase beta in patients with mucopolysaccharidosis II (Hunter syndrome).
Sohn YB, Cho SY, Park SW, Kim SJ, Ko AR, Kwon EK, Han SJ, Jin DK. Sohn YB, et al. Orphanet J Rare Dis. 2013 Mar 18;8:42. doi: 10.1186/1750-1172-8-42. Orphanet J Rare Dis. 2013. PMID: 23497636 Free PMC article. Clinical Trial.
Patients were randomized to active comparator infusions (N=11), 0.5 mg/kg idursulfase beta infusions (N=10), or 1.0 mg/kg idursulfase beta infusions (N=10). ...Both idursulfase beta infusions were generally safe and well tolerated, and elicited no serious adv …
Patients were randomized to active comparator infusions (N=11), 0.5 mg/kg idursulfase beta infusions (N=10), or 1.0 mg/kg idursulf
Immunogenicity of idursulfase and clinical outcomes in very young patients (16 months to 7.5 years) with mucopolysaccharidosis II (Hunter syndrome).
Pano A, Barbier AJ, Bielefeld B, Whiteman DA, Amato DA. Pano A, et al. Orphanet J Rare Dis. 2015 Apr 24;10:50. doi: 10.1186/s13023-015-0265-2. Orphanet J Rare Dis. 2015. PMID: 25902842 Free PMC article. Clinical Trial.
BACKGROUND: Twenty-eight treatment-naive mucopolysaccharidosis II patients (16 months-7.5 years) received 0.5 mg/kg idursulfase weekly for one year in NCT00607386. Serum anti-idursulfase immunoglobulin G antibodies (Abs) were seen in 68% of patients. ...Liver size a …
BACKGROUND: Twenty-eight treatment-naive mucopolysaccharidosis II patients (16 months-7.5 years) received 0.5 mg/kg idursulfase weekl …
The relationship between anti-idursulfase antibody status and safety and efficacy outcomes in attenuated mucopolysaccharidosis II patients aged 5 years and older treated with intravenous idursulfase.
Barbier AJ, Bielefeld B, Whiteman DA, Natarajan M, Pano A, Amato DA. Barbier AJ, et al. Mol Genet Metab. 2013 Nov;110(3):303-10. doi: 10.1016/j.ymgme.2013.08.002. Epub 2013 Aug 9. Mol Genet Metab. 2013. PMID: 23988379 Free article. Clinical Trial.
In the pivotal phase II/III trial of idursulfase administered intravenously to treat mucopolysaccharidosis II, approximately half of the patients developed antibodies to idursulfase. ...A total of 63 treatment-naive patients received 0.5 mg/kg of intravenous idur
In the pivotal phase II/III trial of idursulfase administered intravenously to treat mucopolysaccharidosis II, approximately half of …
Development of the multi-attribute Adolescent Health Utility Measure (AHUM).
Beusterien KM, Yeung JE, Pang F, Brazier J. Beusterien KM, et al. Health Qual Life Outcomes. 2012 Aug 28;10:102. doi: 10.1186/1477-7525-10-102. Health Qual Life Outcomes. 2012. PMID: 22929184 Free PMC article. Clinical Trial.

The random effects model was statistically significant (p < 0.0001; adjusted R2 = 0.361; RMSE = 0.194). When AHUM utilities were applied to the idursulfase trial, mean utilities in the idursulfase weekly and placebo groups improved +0.087 and +0.006, respectively

The random effects model was statistically significant (p < 0.0001; adjusted R2 = 0.361; RMSE = 0.194). When AHUM utilities were applied

Safety and efficacy of enzyme replacement therapy with idursulfase beta in children aged younger than 6 years with Hunter syndrome.
Sohn YB, Cho SY, Lee J, Kwun Y, Huh R, Jin DK. Sohn YB, et al. Mol Genet Metab. 2015 Feb;114(2):156-60. doi: 10.1016/j.ymgme.2014.08.009. Epub 2014 Aug 30. Mol Genet Metab. 2015. PMID: 25219292 Clinical Trial.
Idursulfase beta (Hunterase) has been used for enzyme replacement therapy (ERT) of patients with mucopolysaccharidosis II (MPS II, Hunter syndrome) aged 6 years or older since 2012 in Korea. ...Idursulfase beta (0.5mg/kg/week) was administered intravenously for 52 w
Idursulfase beta (Hunterase) has been used for enzyme replacement therapy (ERT) of patients with mucopolysaccharidosis II (MPS II, Hu
Japan Elaprase Treatment (JET) study: idursulfase enzyme replacement therapy in adult patients with attenuated Hunter syndrome (Mucopolysaccharidosis II, MPS II).
Okuyama T, Tanaka A, Suzuki Y, Ida H, Tanaka T, Cox GF, Eto Y, Orii T. Okuyama T, et al. Mol Genet Metab. 2010 Jan;99(1):18-25. doi: 10.1016/j.ymgme.2009.08.006. Mol Genet Metab. 2010. PMID: 19773189 Clinical Trial.
Two infusion-related reactions were assessed as serious (urticaria and vasovagal syncope). One patient died of causes unrelated to idursulfase. Anti-idursulfase antibodies developed in 60% (6/10) of patients. In summary, idursulfase treatment appears to be sa …
Two infusion-related reactions were assessed as serious (urticaria and vasovagal syncope). One patient died of causes unrelated to idursu
[Clinical study of enzyme replacement therapy with idursulfase].
Gutiérrez-Solana LG. Gutiérrez-Solana LG. Rev Neurol. 2007 Feb 19;44 Suppl 1:S7-S11. Rev Neurol. 2007. PMID: 17345556 Free article. Clinical Trial. Spanish.
DEVELOPMENT: A preclinical trial in a knockout mouse showed a decrease in glycosaminoglycans, both in urine and in tissues, following treatment with idursulfase. In a randomised, double-blind, placebo-controlled clinical study in phase I/II conducted in 12 patients with Hu …
DEVELOPMENT: A preclinical trial in a knockout mouse showed a decrease in glycosaminoglycans, both in urine and in tissues, following treatm …
A phase I/II clinical trial of enzyme replacement therapy in mucopolysaccharidosis II (Hunter syndrome).
Muenzer J, Gucsavas-Calikoglu M, McCandless SE, Schuetz TJ, Kimura A. Muenzer J, et al. Mol Genet Metab. 2007 Mar;90(3):329-37. doi: 10.1016/j.ymgme.2006.09.001. Epub 2006 Dec 20. Mol Genet Metab. 2007. PMID: 17185020 Clinical Trial.
OBJECTIVE: To evaluate the safety and explore the efficacy of idursulfase (recombinant human iduronate-2-sulfatase) treatment for mucopolysaccharidosis II (MPS II). ...Three groups of 4 patients were enrolled sequentially, with 3 patients in each group receiving idursul
OBJECTIVE: To evaluate the safety and explore the efficacy of idursulfase (recombinant human iduronate-2-sulfatase) treatment for muc …
15 results