Literature DB >> 31019230

Effect of enzyme replacement therapy on the growth of patients with Morquio A.

Caitlin Doherty1,2, Molly Stapleton1,2, Matthew Piechnik1,2, Robert W Mason1,2, William G Mackenzie1, Seiji Yamaguchi3, Hironori Kobayashi3, Yasuyuki Suzuki4, Shunji Tomatsu5,6,7.   

Abstract

Mucopolysaccharidosis IVA (MPS IVA) is a degenerative systemic skeletal dysplasia, in which children exhibit marked short stature and become physically handicapped. This study evaluated the growth patterns of patients treated with enzyme replacement therapy (ERT), compared with those of untreated patients. Cross-sectional and longitudinal data of heights and weights were collected from 128 MPS IVA patients and compared with the growth charts of MPS IVA. Twelve patients (six males, six females) starting ERT before 5 years old were treated for at least 2 years. Six out of 12 patients (50%) with ERT over 2 years stopped growing between 94 and 98 cm (mean height of 95.1 ± 2.2 cm) from 5.0 years to 9.0 years of age (mean age of 6.2 ± 1.6 years). The other patients, except one attenuated case, exhibited a marked slow growth velocity from 3.6 years to 7.7 years. Treated and untreated patients with severe phenotype reached their final heights by ~10 years of age. Patients treated with ERT exhibited a reduced pubertal growth spurt analogous to their untreated counterparts, which contributes to the marked short stature associated with MPS IVA. Compared with the growth charts for untreated patients, patients treated with ERT did not show any significant increase in growth in any age group. Overall, ERT-treated patients do not experience growth improvement and continue to exhibit poor growth despite early ERT intervention before 5 years of age. These findings indicate that current intravenous ERT is ineffective at correcting abnormal growth in MPS IVA.

Entities:  

Mesh:

Substances:

Year:  2019        PMID: 31019230     DOI: 10.1038/s10038-019-0604-6

Source DB:  PubMed          Journal:  J Hum Genet        ISSN: 1434-5161            Impact factor:   3.172


  43 in total

1.  Incidence of inborn errors of metabolism in British Columbia, 1969-1996.

Authors:  D A Applegarth; J R Toone; R B Lowry
Journal:  Pediatrics       Date:  2000-01       Impact factor: 7.124

2.  Obstructive airway in Morquio A syndrome, the past, the present and the future.

Authors:  Shunji Tomatsu; Lauren W Averill; Kazuki Sawamoto; William G Mackenzie; Michael B Bober; Christian Pizarro; Christopher J Goff; Li Xie; Tadao Orii; Mary Theroux
Journal:  Mol Genet Metab       Date:  2015-09-21       Impact factor: 4.797

3.  Epidemiology of mucopolysaccharidoses.

Authors:  Shaukat A Khan; Hira Peracha; Diana Ballhausen; Alfred Wiesbauer; Marianne Rohrbach; Matthias Gautschi; Robert W Mason; Roberto Giugliani; Yasuyuki Suzuki; Kenji E Orii; Tadao Orii; Shunji Tomatsu
Journal:  Mol Genet Metab       Date:  2017-05-26       Impact factor: 4.797

4.  Glycosaminoglycans analysis in blood and urine of patients with mucopolysaccharidosis.

Authors:  Shaukat A Khan; Robert W Mason; Roberto Giugliani; Kenji Orii; Toshiyuki Fukao; Yasuyuki Suzuki; Seiji Yamaguchi; Hironori Kobayashi; Tadao Orii; Shunji Tomatsu
Journal:  Mol Genet Metab       Date:  2018-05-17       Impact factor: 4.797

Review 5.  Review of clinical presentation and diagnosis of mucopolysaccharidosis IVA.

Authors:  C J Hendriksz; P Harmatz; M Beck; S Jones; T Wood; R Lachman; C G Gravance; T Orii; S Tomatsu
Journal:  Mol Genet Metab       Date:  2013-04-10       Impact factor: 4.797

Review 6.  Mucopolysaccharidosis type II (Hunter syndrome): a clinical review and recommendations for treatment in the era of enzyme replacement therapy.

Authors:  J Edmond Wraith; Maurizio Scarpa; Michael Beck; Olaf A Bodamer; Linda De Meirleir; Nathalie Guffon; Allan Meldgaard Lund; Gunilla Malm; Ans T Van der Ploeg; Jiri Zeman
Journal:  Eur J Pediatr       Date:  2007-11-23       Impact factor: 3.183

7.  Natural history of Morquio A patient with tracheal obstruction from birth to death.

Authors:  Caitlin Doherty; Lauren W Averill; Mary Theroux; William G Mackenzie; Christian Pizarro; Robert W Mason; Shunji Tomatsu
Journal:  Mol Genet Metab Rep       Date:  2017-12-22

8.  Enzyme replacement therapy for treating mucopolysaccharidosis type IVA (Morquio A syndrome): effect and limitations.

Authors:  Shunji Tomatsu; Kazuki Sawamoto; Tsutomu Shimada; Michael B Bober; Francyne Kubaski; Eriko Yasuda; Robert W Mason; Shaukat Khan; Carlos J Alméciga-Díaz; Luis A Barrera; William G Mackenzie; Tadao Orii
Journal:  Expert Opin Orphan Drugs       Date:  2015-10-29       Impact factor: 0.694

9.  Efficacy and safety of enzyme replacement therapy with BMN 110 (elosulfase alfa) for Morquio A syndrome (mucopolysaccharidosis IVA): a phase 3 randomised placebo-controlled study.

Authors:  Christian J Hendriksz; Barbara Burton; Thomas R Fleming; Paul Harmatz; Derralynn Hughes; Simon A Jones; Shuan-Pei Lin; Eugen Mengel; Maurizio Scarpa; Vassili Valayannopoulos; Roberto Giugliani; Peter Slasor; Debra Lounsbury; Wolfgang Dummer
Journal:  J Inherit Metab Dis       Date:  2014-05-09       Impact factor: 4.982

10.  Safety and clinical activity of elosulfase alfa in pediatric patients with Morquio A syndrome (mucopolysaccharidosis IVA) less than 5 y.

Authors:  Simon A Jones; Martin Bialer; Rossella Parini; Ken Martin; Hui Wang; Ke Yang; Adam J Shaywitz; Paul Harmatz
Journal:  Pediatr Res       Date:  2015-09-02       Impact factor: 3.756

View more
  17 in total

Review 1.  Enzyme replacement therapy for mucopolysaccharidoses; past, present, and future.

Authors:  Hui Hsuan Chen; Kazuki Sawamoto; Robert W Mason; Hironori Kobayashi; Seiji Yamaguchi; Yasuyuki Suzuki; Kenji Orii; Tadao Orii; Shunji Tomatsu
Journal:  J Hum Genet       Date:  2019-08-27       Impact factor: 3.172

2.  Bromocriptine as a Novel Pharmacological Chaperone for Mucopolysaccharidosis IV A.

Authors:  Sergio Olarte-Avellaneda; Jacobo Cepeda Del Castillo; Andrés Felipe Rojas-Rodriguez; Oscar Sánchez; Alexander Rodríguez-López; Diego A Suárez García; Luz Mary Salazar Pulido; Carlos J Alméciga-Díaz
Journal:  ACS Med Chem Lett       Date:  2020-06-24       Impact factor: 4.345

Review 3.  Bone-Specific Drug Delivery for Osteoporosis and Rare Skeletal Disorders.

Authors:  Kazuki Sawamoto; J Víctor Álvarez; Angélica María Herreño; Francisco J Otero-Espinar; Maria L Couce; Carlos J Alméciga-Díaz; Shunji Tomatsu
Journal:  Curr Osteoporos Rep       Date:  2020-10       Impact factor: 5.096

Review 4.  Therapeutic Options for Mucopolysaccharidoses: Current and Emerging Treatments.

Authors:  Kazuki Sawamoto; Molly Stapleton; Carlos J Alméciga-Díaz; Angela J Espejo-Mojica; Juan Camilo Losada; Diego A Suarez; Shunji Tomatsu
Journal:  Drugs       Date:  2019-07       Impact factor: 11.431

5.  Liver-Targeted AAV8 Gene Therapy Ameliorates Skeletal and Cardiovascular Pathology in a Mucopolysaccharidosis IVA Murine Model.

Authors:  Kazuki Sawamoto; Subha Karumuthil-Melethil; Shaukat Khan; Molly Stapleton; Joseph T Bruder; Olivier Danos; Shunji Tomatsu
Journal:  Mol Ther Methods Clin Dev       Date:  2020-05-22       Impact factor: 6.698

6.  Pathophysiology of Hip Disorders in Patients with Mucopolysaccharidosis IVA.

Authors:  Zhigang Wang; Yunlan Xu; Enze Jiang; Jianmin Wang; Shunji Tomatsu; Kaiying Shen
Journal:  Diagnostics (Basel)       Date:  2020-04-29

7.  A Case Report of a Japanese Boy with Morquio A Syndrome: Effects of Enzyme Replacement Therapy Initiated at the Age of 24 Months.

Authors:  Akari Nakamura-Utsunomiya; Toshio Nakamae; Reiko Kagawa; Shuhei Karakawa; Sonoko Sakata; Fumiaki Sakura; Chihiro Tani; Yoshiko Matsubara; Takashi Ishino; Go Tajima; Satoshi Okada
Journal:  Int J Mol Sci       Date:  2020-02-02       Impact factor: 5.923

8.  Enzyme-Loaded Gel Core Nanostructured Lipid Carriers to Improve Treatment of Lysosomal Storage Diseases: Formulation and In Vitro Cellular Studies of Elosulfase Alfa-Loaded Systems.

Authors:  J Víctor Álvarez; Carolina Herrero Filgueira; Alexandre de la Fuente González; Cristóbal Colón Mejeras; Andrés Beiras Iglesias; Shunji Tomatsu; José Blanco Méndez; Asteria Luzardo Álvarez; María Luz Couce; Francisco J Otero Espinar
Journal:  Pharmaceutics       Date:  2019-10-11       Impact factor: 6.321

9.  Plasma Proteomic Analysis in Morquio A Disease.

Authors:  José V Álvarez; Susana B Bravo; María Pilar Chantada-Vázquez; Sofía Barbosa-Gouveia; Cristóbal Colón; Olalla López-Suarez; Shunji Tomatsu; Francisco J Otero-Espinar; María L Couce
Journal:  Int J Mol Sci       Date:  2021-06-07       Impact factor: 5.923

10.  Proteomic Analysis in Morquio A Cells Treated with Immobilized Enzymatic Replacement Therapy on Nanostructured Lipid Systems.

Authors:  J Víctor Álvarez; Susana B Bravo; María García-Vence; María J De Castro; Asteria Luzardo; Cristóbal Colón; Shunji Tomatsu; Francisco J Otero-Espinar; María L Couce
Journal:  Int J Mol Sci       Date:  2019-09-18       Impact factor: 5.923

View more

北京卡尤迪生物科技股份有限公司 © 2022-2023.