Article: Young-onset Parkinsonism in a Hong Kong Chinese man with adult-onset Hallervorden-Spatz syndrome
| Title | Young-onset Parkinsonism in a Hong Kong Chinese man with adult-onset Hallervorden-Spatz syndrome |
|---|---|
| Authors | Mak, CM2 Sheng, B2 Lee, HHC2 Lau, KK2 Chan, WT2 Lam, CW1 Chan, YW2 |
| Issue Date | 2011 |
| Publisher | Informa Healthcare. The Journal's web site is located at http://www.tandf.co.uk/journals/titles/00207454.asp |
| Citation | International Journal Of Neuroscience, 2011, v. 121 n. 4, p. 224-227 [How to Cite?] DOI: http://dx.doi.org/10.3109/00207454.2010.542843 |
| Abstract | Neurodegeneration with brain iron accumulation (NBIA) is a heterogeneous group of disorders varied in genetic etiologies, clinical presentations, and radiological features. NBIA is an iron homeostasis disorder with progressive iron accumulation in the central nervous systems and is clinically characterized by extrapyramidal movement abnormalities, retinal pigmentary changes, and cognitive impairment. Panthothenate kinase-associated neurodegeneration (Hallervorden-Spatz disease) is the commonest disorder of NBIA with a prevalence of one-three per million. Clinically, it is classified into early-onset childhood, atypical late-onset, and adult-onset type. Adult-onset type is rarer. We report the first case of adult-onset panthothenate kinase-associated neurodegeneration in Hong Kong in a 28-year-old Chinese man who presented with pure young-onset parkinsonism. Magnetic resonance imaging (MRI) of the brain showed the presence of eye-of-the-tiger sign. Two compound heterozygous mutations PANK2 NM-153638.2: c.445G > T; NP-705902.2: p.E149X and PANK2 NM-153638.2: c.1133A > G; NP-705902.2: p.D378G were detected. Parkinsonism per se is a very heterogeneous phenotypic group. In view of the readily available genetic analysis of PANK2, panthothenate kinase-associated neurodegeneration should be considered in adult patients with young-onset parkinsonism with or without the eye-of-the-tiger sign. The exact diagnosis offers a different management approach and genetic counseling. NBIA is likely under- or misdiagnosed in Hong Kong Chinese. © 2011 Informa Healthcare USA, Inc. |
| ISSN | 0020-7454 2011 Impact Factor: 0.967 2011 SCImago Journal Rankings: 0.075 |
| DOI | http://dx.doi.org/10.3109/00207454.2010.542843 |
| ISI Accession Number ID | WOS:000289033700009 |
| References | References in Scopus |
| dc.contributor.author | Mak, CM |
|---|---|
| dc.contributor.author | Sheng, B |
| dc.contributor.author | Lee, HHC |
| dc.contributor.author | Lau, KK |
| dc.contributor.author | Chan, WT |
| dc.contributor.author | Lam, CW |
| dc.contributor.author | Chan, YW |
| dc.date.accessioned | 2011-09-23T06:01:39Z |
| dc.date.available | 2011-09-23T06:01:39Z |
| dc.date.issued | 2011 |
| dc.description.abstract | Neurodegeneration with brain iron accumulation (NBIA) is a heterogeneous group of disorders varied in genetic etiologies, clinical presentations, and radiological features. NBIA is an iron homeostasis disorder with progressive iron accumulation in the central nervous systems and is clinically characterized by extrapyramidal movement abnormalities, retinal pigmentary changes, and cognitive impairment. Panthothenate kinase-associated neurodegeneration (Hallervorden-Spatz disease) is the commonest disorder of NBIA with a prevalence of one-three per million. Clinically, it is classified into early-onset childhood, atypical late-onset, and adult-onset type. Adult-onset type is rarer. We report the first case of adult-onset panthothenate kinase-associated neurodegeneration in Hong Kong in a 28-year-old Chinese man who presented with pure young-onset parkinsonism. Magnetic resonance imaging (MRI) of the brain showed the presence of eye-of-the-tiger sign. Two compound heterozygous mutations PANK2 NM-153638.2: c.445G > T; NP-705902.2: p.E149X and PANK2 NM-153638.2: c.1133A > G; NP-705902.2: p.D378G were detected. Parkinsonism per se is a very heterogeneous phenotypic group. In view of the readily available genetic analysis of PANK2, panthothenate kinase-associated neurodegeneration should be considered in adult patients with young-onset parkinsonism with or without the eye-of-the-tiger sign. The exact diagnosis offers a different management approach and genetic counseling. NBIA is likely under- or misdiagnosed in Hong Kong Chinese. © 2011 Informa Healthcare USA, Inc. |
| dc.description.nature | Link_to_subscribed_fulltext |
| dc.identifier.citation | International Journal Of Neuroscience, 2011, v. 121 n. 4, p. 224-227 [How to Cite?] DOI: http://dx.doi.org/10.3109/00207454.2010.542843 |
| dc.identifier.doi | http://dx.doi.org/10.3109/00207454.2010.542843 |
| dc.identifier.epage | 227 |
| dc.identifier.hkuros | 192462 |
| dc.identifier.isi | WOS:000289033700009 |
| dc.identifier.issn | 0020-7454 2011 Impact Factor: 0.967 2011 SCImago Journal Rankings: 0.075 |
| dc.identifier.issue | 4 |
| dc.identifier.pmid | 21198414 |
| dc.identifier.scopus | eid_2-s2.0-79951926412 |
| dc.identifier.spage | 224 |
| dc.identifier.uri | http://hdl.handle.net/10722/139927 |
| dc.identifier.volume | 121 |
| dc.language | eng |
| dc.publisher | Informa Healthcare. The Journal's web site is located at http://www.tandf.co.uk/journals/titles/00207454.asp |
| dc.publisher.place | United Kingdom |
| dc.relation.ispartof | International Journal of Neuroscience |
| dc.relation.references | References in Scopus |
| dc.subject.mesh | Adult |
| dc.subject.mesh | Asian Continental Ancestry Group - ethnology - genetics |
| dc.subject.mesh | Genetic Predisposition to Disease - ethnology - genetics |
| dc.subject.mesh | Hong Kong - ethnology |
| dc.subject.mesh | Humans |
| dc.subject.mesh | Male |
| dc.subject.mesh | Mutation - genetics |
| dc.subject.mesh | Pantothenate Kinase-Associated Neurodegeneration - diagnosis - ethnology |
| dc.subject.mesh | Parkinsonian Disorders - diagnosis - ethnology |
| dc.subject.mesh | Phenotype |
| dc.subject.mesh | Phosphotransferases (Alcohol Group Acceptor) - genetics |
| dc.title | Young-onset Parkinsonism in a Hong Kong Chinese man with adult-onset Hallervorden-Spatz syndrome |
| dc.type | Article |
Author Affiliations
- The University of Hong Kong Li Ka Shing Faculty of Medicine
- Princess Margaret Hospital Hong Kong

