Literature DB >> 26920906

Clinical Evolution After Enzyme Replacement Therapy in Twins with the Severe Form of Maroteaux-Lamy Syndrome.

M Pineda1, M O'Callaghan2, A Fernandez Lopez3, M J Coll4, R Ullot5, G Garcia-Fructuoso6.   

Abstract

Mucopolysaccharidosis type VI (MPS VI) is a progressive, autosomal, recessive lysosomal disorder. This disorder, due to a deficiency in N-acetylgalactosamine-4-sulfatase (ASB), results in an accumulation of glycosaminoglycan (GAG), causing multiple organ failures. In this study, monochorionic biamniotic twins with the severe form of MPS VI underwent enzyme replacement therapy (ERT) with weekly infusions of recombinant human ASB (galsulfase) at 1 mg/kg. After 9 years of ERT, a comprehensive clinical examination was performed. Several types of biochemical, immunological, and genetic investigations were also conducted. Both twins showed the typical symptoms and signs of MPS VI at baseline, including short stature, progressive dysmorphic facial features, and dysostosis multiplex. Twin 2 presented stronger multisystemic involvement, with marked musculoskeletal, neurological, and odontological components. She also developed an ischemic spinal cord lesion after surgery, which is the first case described in the literature in Maroteaux-Lamy syndrome. However, the extent of disease was found to be equally stabilized in the two sisters, concretely the cardiac and respiratory functions and body length. The early diagnosis and treatment of MPS VI are critical for an optimal clinical outcome, and further evidence for the new treatment strategies is needed.

Entities:  

Year:  2016        PMID: 26920906      PMCID: PMC5110444          DOI: 10.1007/8904_2016_530

Source DB:  PubMed          Journal:  JIMD Rep        ISSN: 2192-8304


  24 in total

1.  [A NEW DYSOSTOSIS WITH URINARY ELIMINATION OF CHONDROITIN SULFATE B].

Authors:  P MAROTEAUX; B LEVEQUE; J MARIE; M LAMY
Journal:  Presse Med       Date:  1963-09-25       Impact factor: 1.228

2.  Clinical characteristics of adults with slowly progressing mucopolysaccharidosis VI: a case series.

Authors:  Anke Thümler; Elke Miebach; Christina Lampe; Susanne Pitz; Wolfgang Kamin; Christoph Kampmann; Bianca Link; Eugen Mengel
Journal:  J Inherit Metab Dis       Date:  2012-03-23       Impact factor: 4.982

3.  Chromosomal localization of ARSB, the gene for human N-acetylgalactosamine-4-sulphatase.

Authors:  T Litjens; E G Baker; K R Beckmann; C P Morris; J J Hopwood; D F Callen
Journal:  Hum Genet       Date:  1989-04       Impact factor: 4.132

4.  Echocardiographic study of paediatric patients with mucopolysaccharidosis.

Authors:  Gabriela N Leal; Ana C de Paula; Cláudio Leone; Chong A Kim
Journal:  Cardiol Young       Date:  2010-04-26       Impact factor: 1.093

5.  Enzyme replacement therapy for mucopolysaccharidosis VI from 8 weeks of age--a sibling control study.

Authors:  J J McGill; A C Inwood; D J Coman; M L Lipke; D de Lore; S J Swiedler; J J Hopwood
Journal:  Clin Genet       Date:  2009-11-23       Impact factor: 4.438

Review 6.  Mucopolysaccharidosis VI.

Authors:  Vassili Valayannopoulos; Helen Nicely; Paul Harmatz; Sean Turbeville
Journal:  Orphanet J Rare Dis       Date:  2010-04-12       Impact factor: 4.123

7.  Mucopolysaccharidosis VI: the Italian experience.

Authors:  Maurizio Scarpa; Rita Barone; Agata Fiumara; Luca Astarita; Giancarlo Parenti; Angelica Rampazzo; Stefania Sala; Giovanni Sorge; Rossella Parini
Journal:  Eur J Pediatr       Date:  2009-01-07       Impact factor: 3.183

8.  Identification of the molecular defects in Spanish and Argentinian mucopolysaccharidosis VI (Maroteaux-Lamy syndrome) patients, including 9 novel mutations.

Authors:  Elena Garrido; Amparo Chabás; Maria Josep Coll; Mariana Blanco; Carmen Domínguez; Daniel Grinberg; Lluïsa Vilageliu; Bru Cormand
Journal:  Mol Genet Metab       Date:  2007-07-20       Impact factor: 4.797

9.  The natural course and the impact of therapies of cardiac involvement in the mucopolysaccharidoses.

Authors:  Vlasta Fesslová; Paola Corti; Giovanna Sersale; Attilio Rovelli; Pierluigi Russo; Savina Mannarino; Gianfranco Butera; Rossella Parini
Journal:  Cardiol Young       Date:  2009-02-06       Impact factor: 1.093

10.  Mucopolysaccharidosis type VI phenotypes-genotypes and antibody response to galsulfase.

Authors:  Marion M Brands; Marianne Hoogeveen-Westerveld; Marian A Kroos; Willemieke Nobel; George J Ruijter; Lale Özkan; Iris Plug; Daniel Grinberg; Lluïsa Vilageliu; Dicky J Halley; Ans T van der Ploeg; Arnold J Reuser
Journal:  Orphanet J Rare Dis       Date:  2013-04-04       Impact factor: 4.123

View more

北京卡尤迪生物科技股份有限公司 © 2022-2023.