SHORT REPORT

A case of manganese induced parkinsonism in hereditary haemorrhagic telangiectasia

K Yoshikawa, M Matsumoto, M Hamanaka and M Nakagawa

Research Institute for Neurological Diseases and Geriatrics, Kyoto Prefectural University of Medicine, 465 Kawaramachi-Hirokoji, Kamigyo-ku, Kyoto 602-8566, Japan

Correspondence to:
Correspondence to:
Dr Kenji Yoshikawa;
kyoshika{at}koto.kpu-m.ac.jp

A 44 year old right handed woman complained of difficulty in moving. She and her relatives had skin telangiectasia or recurrent epistaxis. On neurological examination, she had a mask-like facies and bradykinesia in both extremities. Laboratory examinations showed iron deficiency anaemia and mild liver dysfunction with raised serum manganese. On T1 weighted cranial magnetic resonance imaging there were hyperintense areas in the globus pallidus bilaterally, suggesting manganese deposition. Abdominal angiography confirmed multiple portal-systemic shunts in the liver, and a needle biopsy of the liver showed diffuse dilatation of the sinusoids with fatty change. Levodopa did not improve the bradykinesia. This appears to be a case of hereditary haemorrhagic telangiectasia with manganese induced parkinsonism, which may be a new type of neurological disorder in such patients.

Keywords: hereditary haemorrhagic telangiectasia; parkinsonism; manganese

CiteULike    Complore    Connotea    Del.icio.us    Digg    Reddit    Technorati    What's this?

This article has been cited by other articles:

• Kumar, N., Boeve, B. F., Cowl, C. T., Ellison, J. W., Kamath, P. S., Swanson, K. L. (2008). HYPERMANGANESEMIA, HEREDITARY HEMORRHAGIC TELANGIECTASIA, BRAIN ABSCESS: THE HEPATIC CONNECTION. Neurology 71: 1118-1119 [Full Text]  
• da Rocha, A. J., Braga, F. T., da Silva, C. J., Maia, A. C. M. Jr, Mourao, G. S., Gagliardi, R. J. (2004). Reversal of Parkinsonism and Portosystemic Encephalopathy Following Embolization of a Congenital Intrahepatic Venous Shunt: Brain MR Imaging and 1H Spectroscopic Findings. Am. J. Neuroradiol. 25: 1247-1250 [Abstract] [Full Text]  
• de la Fuente-Fernandez, R, Yoshikawa, K, Nakagawa, M (2004). Portal-systemic shunts, manganese, and parkinsonism * Authors' reply. J. Neurol. Neurosurg. Psychiatry 75: 1081-1082 [Full Text]