Literature DB >> 23649721

Diagnosis of Pompe disease: muscle biopsy vs blood-based assays.

John Vissing1, Zoltan Lukacs, Volker Straub.   

Abstract

The diagnosis of Pompe disease (acid maltase deficiency, glycogen storage disease type II) in children and adults can be challenging because of the heterogeneous clinical presentation and considerable overlap of signs and symptoms found in other neuromuscular diseases. This review evaluates some of the methods used in the diagnosis and differential diagnosis of late-onset Pompe disease. Muscle biopsy is commonly used as an early diagnostic tool in the evaluation of muscle disease. However, experience has shown that relying solely on visualizing a periodic acid-Schiff-positive vacuolar myopathy to identify late-onset Pompe disease often leads to false-negative results and subsequent delays in identification and treatment of the disorder. Serum creatine kinase level can be normal or only mildly elevated in late-onset Pompe disease and is not very helpful alone to suggest the diagnosis, but in combination with proximal and axial weakness it may raise the suspicion for Pompe disease. A simple blood-based assay to measure the level of α-glucosidase activity is the optimal initial test for confirming or excluding Pompe disease. A timely and accurate diagnosis of late-onset Pompe disease likely will improve patient outcomes as care standards including enzyme replacement therapy can be applied and complications can be anticipated. Increased awareness of the clinical phenotype of Pompe disease is therefore warranted to expedite diagnostic screening for this condition with blood-based enzymatic assays.

Entities:  

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Year:  2013        PMID: 23649721     DOI: 10.1001/2013.jamaneurol.486

Source DB:  PubMed          Journal:  JAMA Neurol        ISSN: 2168-6149            Impact factor:   18.302


  15 in total

Review 1.  Diagnostic tools in late onset Pompe disease (LOPD).

Authors:  Olimpia Musumeci; Antonio Toscano
Journal:  Ann Transl Med       Date:  2019-07

Review 2.  Pompe Disease: From Basic Science to Therapy.

Authors:  Lara Kohler; Rosa Puertollano; Nina Raben
Journal:  Neurotherapeutics       Date:  2018-10       Impact factor: 7.620

3.  Sensitivity of whole exome sequencing in detecting infantile- and late-onset Pompe disease.

Authors:  Mari Mori; Gloria Haskell; Zoheb Kazi; Xiaolin Zhu; Stephanie M DeArmey; Jennifer L Goldstein; Deeksha Bali; Catherine Rehder; Elizabeth T Cirulli; Priya S Kishnani
Journal:  Mol Genet Metab       Date:  2017-10-17       Impact factor: 4.797

Review 4.  Pompe disease: literature review and case series.

Authors:  Majed Dasouki; Omar Jawdat; Osama Almadhoun; Mamatha Pasnoor; April L McVey; Ahmad Abuzinadah; Laura Herbelin; Richard J Barohn; Mazen M Dimachkie
Journal:  Neurol Clin       Date:  2014-08       Impact factor: 3.806

5.  Effect of Cadmium Ion on alpha-Glucosidase: An Inhibition Kinetics and Molecular Dynamics Simulation Integration Study.

Authors:  Tao Luo; Jinhyuk Lee; Zhi-Rong Lü; Hang Mu; Li-Mei Yue; Yong-Doo Park; Zhuo-Ming Ye
Journal:  Protein J       Date:  2016-06       Impact factor: 2.371

6.  Clinical and GAA gene mutation analysis in mainland Chinese patients with late-onset Pompe disease: identifying c.2238G > C as the most common mutation.

Authors:  Xiao Liu; Zhaoxia Wang; Weina Jin; He Lv; Wei Zhang; Chengli Que; Yu Huang; Yun Yuan
Journal:  BMC Med Genet       Date:  2014-12-20       Impact factor: 2.103

7.  Clinical features of Pompe disease.

Authors:  Fiore Manganelli; Lucia Ruggiero
Journal:  Acta Myol       Date:  2013-10

8.  The value of muscle biopsies in Pompe disease: identifying lipofuscin inclusions in juvenile- and adult-onset patients.

Authors:  Erin J Feeney; Stephanie Austin; Yin-Hsiu Chien; Hanna Mandel; Benedikt Schoser; Sean Prater; Wuh-Liang Hwu; Evelyn Ralston; Priya S Kishnani; Nina Raben
Journal:  Acta Neuropathol Commun       Date:  2014-01-02       Impact factor: 7.801

9.  Early is better? A new algorithm for early diagnosis in late onset Pompe disease (LOPD).

Authors:  Antonio Toscano; Federica Montagnese; Olimpia Musumeci
Journal:  Acta Myol       Date:  2013-10

10.  Glycogenosome accumulation in the arrector pili muscle in Pompe disease.

Authors:  Istvan Katona; Joachim Weis; Frank Hanisch
Journal:  Orphanet J Rare Dis       Date:  2014-02-05       Impact factor: 4.123

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