Literature DB >> 14979382

Diagnosis of mucopolysaccharidoses: how to avoid false positives and false negatives.

K Mahalingam1, S Janani, S Priya, E M Elango, R Maya Sundari.   

Abstract

OBJECTIVE: This paper advocates a complete procedure, which includes both quantitative and qualitative analysis of urinary GAGs in the diagnosis of MPS in a clinically suspected population.
METHODS: Urine samples from 219 clinically suspected mucopolysaccharidoses (MPS) patients and 91 controls were analysed using a combination of methods. Quantitation of isolated urinary glycosaminoglycans (GAGs) were carried out using acid alcian blue complex formation method and qualitative urinary GAG analysis by multisolvent sequential thin layer chromatography
RESULTS: Of the 219 patients analysed, 131 were confirmed to be suffering from MPS. Quantitation of urinary GAGs alone would have missed 60 low GAG excreting MPS patients and misdiagnosed 26 high GAG excreting nonMPS as MPS patients. Further qualitative analysis and enzyme estimation were needed to identify these 60 low GAG excreting MPS patients and 26 high GAG excreting non MPS patients.
CONCLUSION: These results emphasize that quantitation of urinary GAGs alone cannot diagnose MPS patients, it should be coupled with qualitative analysis and enzyme estimations for differential/definitive diagnosis.

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Year:  2004        PMID: 14979382     DOI: 10.1007/bf02725652

Source DB:  PubMed          Journal:  Indian J Pediatr        ISSN: 0019-5456            Impact factor:   1.967


  7 in total

1.  Discontinuous electrophoresis of glycosaminoglycans: a screening method for mucopolysaccharidoses.

Authors:  E M Elango; S Priya; R Mayasundari
Journal:  Indian J Pediatr       Date:  1998 Jul-Aug       Impact factor: 1.967

Review 2.  The mucopolysaccharidoses. Diagnosis, molecular genetics and treatment.

Authors:  J J Hopwood; C P Morris
Journal:  Mol Biol Med       Date:  1990-10

3.  A review and selection of simple laboratory methods used for the study of glycosaminoglycan excretion and the diagnosis of the mucopolysaccharidoses.

Authors:  C A Pennock
Journal:  J Clin Pathol       Date:  1976-02       Impact factor: 3.411

4.  Juvenile GM 1 gangliosidosis: clinical, pathological, chemical and enzymatic studies.

Authors:  J S O'Brien; M W Ho; M L Veath; J F Wilson; G Myers; J M Opitz; G M ZuRhein; J W Spranger; H A Hartmann; B Haneberg; F R Grosse
Journal:  Clin Genet       Date:  1972       Impact factor: 4.438

5.  Urinary screening for disorders of heteroglycan metabolism. Results of 10 years experience with a comprehensive system.

Authors:  A C Sewell
Journal:  Klin Wochenschr       Date:  1988-01-15

6.  The quantitative measurement of Alcian Blue-glycosaminoglycan complexes.

Authors:  P Whiteman
Journal:  Biochem J       Date:  1973-02       Impact factor: 3.857

7.  Diagnosis of mucopolysaccharidoses in a clinically selected population by urinary glycosaminoglycan analysis: a study of 2,000 urine samples.

Authors:  M Piraud; S Boyer; M Mathieu; I Maire
Journal:  Clin Chim Acta       Date:  1993-11-30       Impact factor: 3.786

  7 in total
  10 in total

Review 1.  An overview of Korean patients with mucopolysaccharidosis and collaboration through the Asia Pacific MPS Network.

Authors:  Sung Yoon Cho; Young Bae Sohn; Dong-Kyu Jin
Journal:  Intractable Rare Dis Res       Date:  2014-08

Review 2.  Misdiagnosis in mucopolysaccharidoses.

Authors:  Karolina Wiśniewska; Jakub Wolski; Lidia Gaffke; Zuzanna Cyske; Karolina Pierzynowska; Grzegorz Węgrzyn
Journal:  J Appl Genet       Date:  2022-05-13       Impact factor: 2.653

3.  Mucopolysaccharidoses diagnosis in the era of enzyme replacement therapy in Egypt.

Authors:  Ekram Fateen; Zeinab Y Abdallah; Walaa S Nazim; Mona Ibrahim; Amira Radwan
Journal:  Heliyon       Date:  2021-08-19

Review 4.  Mucopolysaccharidoses.

Authors:  Rolando Cimaz; Francesco La Torre
Journal:  Curr Rheumatol Rep       Date:  2014-01       Impact factor: 4.592

5.  Diagnosing Hunter syndrome in pediatric practice: practical considerations and common pitfalls.

Authors:  Barbara K Burton; Roberto Giugliani
Journal:  Eur J Pediatr       Date:  2012-03-01       Impact factor: 3.183

6.  Validation of Urinary Glycosaminoglycans in Iranian patients with Mucopolysaccharidase type I: The effect of urine sedimentation characteristics.

Authors:  Mohammad Abdi; Mohammad Said Hakhamaneshi; Mohammad Reza Alaei; Namam-Ali Azadi; Rahim Vakili; Daniel Zamanfar; Mohammad Taghikhani; Shohreh Khatami
Journal:  Iran J Child Neurol       Date:  2014

7.  Selective screening for detection of mucopolysaccharidoses in Malaysia; A two-year study (2014-2016).

Authors:  Affandi Omar; Julaina A Jalil; Norashareena M Shakrin; Lock H Ngu; Zabedah M Yunus
Journal:  Mol Genet Metab Rep       Date:  2019-05-10

Review 8.  Mucopolysaccharidosis type III (Sanfilippo syndrome) and misdiagnosis of idiopathic developmental delay, attention deficit/hyperactivity disorder or autism spectrum disorder.

Authors:  Frits A Wijburg; Grzegorz Węgrzyn; Barbara K Burton; Anna Tylki-Szymańska
Journal:  Acta Paediatr       Date:  2013-02-06       Impact factor: 2.299

Review 9.  Diagnosis of Mucopolysaccharidoses.

Authors:  Francyne Kubaski; Fabiano de Oliveira Poswar; Kristiane Michelin-Tirelli; Maira Graeff Burin; Diana Rojas-Málaga; Ana Carolina Brusius-Facchin; Sandra Leistner-Segal; Roberto Giugliani
Journal:  Diagnostics (Basel)       Date:  2020-03-22

10.  Presentation and Treatments for Mucopolysaccharidosis Type II (MPS II; Hunter Syndrome).

Authors:  Molly Stapleton; Francyne Kubaski; Robert W Mason; Hiromasa Yabe; Yasuyuki Suzuki; Kenji E Orii; Tadao Orii; Shunji Tomatsu
Journal:  Expert Opin Orphan Drugs       Date:  2017-03-08       Impact factor: 0.694

  10 in total

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