Literature DB >> 17361416

Abnormalities in the hair morphology of patients with some but not all types of mucopolysaccharidoses.

Marcelina Malinowska1, Joanna Jakóbkiewicz-Banecka, Anna Kloska, Anna Tylki-Szymańska, Barbara Czartoryska, Ewa Piotrowska, Alicja Wegrzyn, Grzegorz Wegrzyn.   

Abstract

Mucopolysaccharidoses (MPS) are a group of inherited, progressive, metabolic diseases, caused by the deficiency of one of the enzymes involved in the degradation of glycosaminoglycans (GAGs). The disease is usually fatal, with the life span of most untreated MPS patients being between one and two decades. In this report, on the basis of scanning electron microscopy (SEM) studies, we demonstrate that, besides the many other symptoms of MPS, there are characteristic abnormalities in the hair morphology of patients suffering from some types of this disease (MPS I, MPS II, MPS IIIA, MPS IIIB), but not from other types (MPS IVA, MPS IVB, MPS VI), where the changes are minor, if any. Different GAGs accumulate in the tissues of patients suffering from the various MPS types, and analysis of the disease types in which severe hair abnormalities occur or not could suggest that the accumulation of heparan sulfate, rather than dermatan sulfate or keratan sufate, may be responsible for the major changes in hair morphology. Considerable abnormalities in hair morphology occur in patients suffering from MPS I, MPS II, MPS IIIA, and MPS IIIB, but not in patients suffering from MPS IVA, MPS IVB, and MPS VI; this feature might potentially be used as an additional test for the assessment of the efficacy of treatments for MPS patients (types I, II, IIIA, and IIIB).

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Year:  2007        PMID: 17361416     DOI: 10.1007/s00431-007-0462-7

Source DB:  PubMed          Journal:  Eur J Pediatr        ISSN: 0340-6199            Impact factor:   3.183


  18 in total

1.  Physicochemical hair conformation of patients with Sanfilippo disease type IIIA.

Authors:  R K Charan; G Nauer; U Wagner; A Klabuschnig; G Lubec
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Journal:  J Pediatr       Date:  2006-04       Impact factor: 4.406

3.  Simple method for cutting transverse sections of hair. Comments on shape of hair in Hurler and Sanfilippo syndromes.

Authors:  I A Crump; D M Danks
Journal:  Arch Dis Child       Date:  1971-06       Impact factor: 3.791

4.  Changes in hair morphology of mucopolysaccharidosis I patients treated with recombinant human alpha-L-iduronidase (laronidase, Aldurazyme).

Authors:  Anna Kloska; Jerzy Bohdanowicz; Grazyna Konopa; Anna Tylki-Szymńska; Joanna Jakóbkiewicz-Banecka; Barbara Czartoryska; Anna Liberek; Alicja Wegrzyn; Grzegorz Wegrzyn
Journal:  Am J Med Genet A       Date:  2005-12-15       Impact factor: 2.802

5.  Inhibition of glycosaminoglycan synthesis using rhodamine B in a mouse model of mucopolysaccharidosis type IIIA.

Authors:  Ainslie L K Roberts; Belinda J Thomas; Ashley S Wilkinson; Janice M Fletcher; Sharon Byers
Journal:  Pediatr Res       Date:  2006-07-20       Impact factor: 3.756

6.  Hair as a diagnostic tool in dysmorphology.

Authors:  M Silengo; M Valenzise; L Sorasio; G B Ferrero
Journal:  Clin Genet       Date:  2002-10       Impact factor: 4.438

Review 7.  New prospects for the treatment of lysosomal storage diseases.

Authors:  Raphael Schiffmann; Roscoe O Brady
Journal:  Drugs       Date:  2002       Impact factor: 9.546

8.  Observations on hair shaft morphology in mucopolysaccharidoses.

Authors:  M Teschler-Nicola; W Killian
Journal:  J Ment Defic Res       Date:  1982-09

9.  Enzyme replacement therapy for mucopolysaccharidosis I: a randomized, double-blinded, placebo-controlled, multinational study of recombinant human alpha-L-iduronidase (laronidase).

Authors:  James E Wraith; Lorne A Clarke; Michael Beck; Edwin H Kolodny; Gregory M Pastores; Joseph Muenzer; David M Rapoport; Kenneth I Berger; Stuart J Swiedler; Emil D Kakkis; Tanja Braakman; Elenie Chadbourne; Karen Walton-Bowen; Gerald F Cox
Journal:  J Pediatr       Date:  2004-05       Impact factor: 4.406

10.  A phase II/III clinical study of enzyme replacement therapy with idursulfase in mucopolysaccharidosis II (Hunter syndrome).

Authors:  Joseph Muenzer; James E Wraith; Michael Beck; Roberto Giugliani; Paul Harmatz; Christine M Eng; Ashok Vellodi; Rick Martin; Uma Ramaswami; Muge Gucsavas-Calikoglu; Suresh Vijayaraghavan; Susanne Wendt; Suzanne Wendt; Ana Cristina Puga; Antonio Puga; Brian Ulbrich; Marwan Shinawi; Maureen Cleary; Diane Piper; Anne Marie Conway; Ann Marie Conway; Alan Kimura
Journal:  Genet Med       Date:  2006-08       Impact factor: 8.822

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  8 in total

1.  The use of elevated doses of genistein-rich soy extract in the gene expression-targeted isoflavone therapy for Sanfilippo disease patients.

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Journal:  JIMD Rep       Date:  2011-12-11

2.  Genistein supplementation in patients affected by Sanfilippo disease.

Authors:  Verónica Delgadillo; Maria Del Mar O'Callaghan; Rafael Artuch; Raquel Montero; Mercedes Pineda
Journal:  J Inherit Metab Dis       Date:  2011-05-10       Impact factor: 4.982

3.  Lichen Planopilaris: The first biopsy layer microbiota inspection.

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4.  Heparan sulfate regulates hair follicle and sebaceous gland morphogenesis and homeostasis.

Authors:  Vivien Jane Coulson-Thomas; Tarsis Ferreira Gesteira; Jeffrey Esko; Winston Kao
Journal:  J Biol Chem       Date:  2014-07-22       Impact factor: 5.157

5.  Genistin-rich soy isoflavone extract in substrate reduction therapy for Sanfilippo syndrome: An open-label, pilot study in 10 pediatric patients.

Authors:  Ewa Piotrowska; Joanna Jakóbkiewicz-Banecka; Anna Tylki-Szymanska; Anna Liberek; Agnieszka Maryniak; Marcelina Malinowska; Barbara Czartoryska; Ewa Puk; Anna Kloska; Tomasz Liberek; Sylwia Baranska; Alicja Wegrzyn; Grzegorz Wegrzyn
Journal:  Curr Ther Res Clin Exp       Date:  2008-04

Review 6.  Advances in Understanding Hair Growth.

Authors:  Bruno A Bernard
Journal:  F1000Res       Date:  2016-02-08

7.  Predictive Metagenomic Profiling, Urine Metabolomics, and Human Marker Gene Expression as an Integrated Approach to Study Alopecia Areata.

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Journal:  Front Cell Infect Microbiol       Date:  2020-04-29       Impact factor: 5.293

8.  Easy-to-use algorithm would provide faster diagnoses for mucopolysaccharidosis type I and enable patients to receive earlier treatment.

Authors:  Anna Tylki-Szymańska; Linda De Meirleir; Maja Di Rocco; Waseem M Fathalla; Nathalie Guffon; Christina Lampe; Allan M Lund; Rossella Parini; Frits A Wijburg; Jiri Zeman; Maurizio Scarpa
Journal:  Acta Paediatr       Date:  2018-08       Impact factor: 2.299

  8 in total

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