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Cerebral toxoplasmosis in Acquired Immunodeficiency Syndrome (AIDS) patients also provides unifying pathophysiologic hypotheses for Holmes tremor
© Lekoubou et al; licensee BioMed Central Ltd. 2010
Received: 6 January 2010
Accepted: 3 June 2010
Published: 3 June 2010
Holmes tremor is a rare symptomatic movement disorder. Currently suggested pathophysiological mechanisms of the disease are mostly derived from stroke cases. Although rare, cerebral toxoplasmosis may strengthen the pathophysiologic mechanism of disease.
A case of Holmes tremor secondary to cerebral toxoplasmosis in an AIDS patient is presented. A relevant literature search was performed, using pubmed and several entries for Holmes tremor as labelled in the literature. The unifying feature of our case and those of the literature is the involvement of either the cerebello-thalamo-cortical and/or the dentato-rubro-olivary pathways. The abscess or the extension of surrounding edema beyond these two circuits may account for the superimposed dysfunction of the nigrostriatal system in some but not all cases. The short delay observed in our observation and the dramatic response to treatment may indirectly support the secondary neuronal degeneration theory in the mechanism of Holmes tremor.
Cases of cerebral toxoplasmosis in AIDS patients also provide arguments for the role of the thalamo-cortical and/or the dentato-rubro-olivary pathways dysfunction in the pathogenesis of Holmes tremor. Involvement of the nigro-striatal pathway may not be crucial in the development of this syndrome. Our case also brings additional indirect arguments for the role of secondary neuronal degeneration in the mechanism of Holmes tremor.
Holmes tremor is a rare symptomatic movement disorder . It has a predominantly proximal distribution in the limbs and is characterized by its large amplitude, low frequency (less than 4 Hz), and postural and action patterns that worsen during movement and markedly increase in goal directed movements [2, 3]. We report on a case of Holmes tremor in an acquired immune-deficiency syndrome (AIDS) patient with cerebral toxoplasmosis. Because of the location of the lesion in the postero-lateral thalamus, the extension of surrounding edema to the brain stem and the short delay from initial neurological deficit to tremor onset, this case may strengthen the currently suggested pathophysiological mechanisms of the disease.
A 35-year old heterosexual man consulted for fatigue and speech disturbances on the 17th January 2009 at the Yaounde University Hospital Center. One week before, he started complaining of head dullness and slurred speech. His weight had dropped by 22%, from 72 to 56 kg over the past few months during which he also had persisting fever.
Additional file 2: video file after treatment. This file shows a complete resolution of the tremor after treatment. (MPG 6 MB)
Cases of Holmes tremor in patients with AIDS and cerebral toxoplasmosis.
First author, Year of publication
Location of brain abscess
Involvement of the cerebello-thalamo-cortical and/or dento-rubro-olivary pathway
Involvement of the nigro-striatal pathway
- Right posterior thalamus
- Internal capsule
Strecker K, 2006
- Left midbrain
Pezzini A, 2002
- Left frontal superior gyrus,
- Inferior parietal lobule,
- right thalamus,
- right midbrain
Mattos JP, 2002
- Left midbrain
- Left cerebellar hemisphere
Micheli F, 1997
- Left posterior thalamus
- Posterior arm of internal capsule
Koppel S, 1990
- Left midbrain
- Left frontal white matter
In our patient, the delay from initial neurological deficit to the onset of tremor was only eighteen days. In previous reports, this delay (when available) ranged from 1 month to 5 months [4–7]. The role of a secondary degeneration in the mechanisms of tremor has been advocated as it usually arises as a delayed manifestation of the initial lesion . In our observation and in that of Mattos , tremor occurred within one month of initial neurological deficit. Unlike other cases of Holmes tremor including those related to toxoplasmic abscess, in these two cases, there was a dramatic improvement of the tremor while on antitoxoplasmic/steroid treatment. It is likely that in our case, neuronal integrity was restored before degeneration was initiated and very unlikely that the improvement could have occurred spontaneously as reported cases of spontaneous relief occurred within one year of tremor onset .
Our case further illustrates the role of neural pathways namely the cerebello-thalamo-cortical and/or the dentato-rubro-olivary in the pathogenesis of Holmes tremor. Put together, reported cases of Holmes tremor in the setting of AIDS and toxoplasmic abscess suggest that involvement of the nigro-striatal pathway may not be crucial in the development of this syndrome. Our case also brings additional indirect arguments for the role of secondary neuronal degeneration in the mechanism of Holmes tremor.
Written informed consent was obtained from the patient for publication of this case report and any accompanying images/video. A copy of the written consent is available for review by the Editor-in-Chief of this journal.
We deeply thank Dr Stephane Thobois for reviewing the video and providing suggestions for the manuscript.
- Pezzini A, Zavarise P, Palvarini L, Viale P, Oladeji O, Padovani A: Holme's tremor following midbrain toxoplasma abscess: clinical features and treatment of a case. Parkinsonism Relat Disord. 2002, 8: 177-180. 10.1016/S1353-8020(01)00013-X.View ArticlePubMedGoogle Scholar
- Paviour DC, Jäger HR, Wilkinson L, Jahanshashi M, Lees AJ: Holmes tremor: Application of modern Neuroimaging techniques. Mov Disord. 2006, 21 (12): 2260-2262. 10.1002/mds.20981.View ArticlePubMedGoogle Scholar
- Vidailhet M, Jedynak CP, Pollak P, Agid Y: Pathology of Symptomatic Tremors. Mov Disord. 1998, 13 (3): 49-54. 10.1002/mds.870131309. 3PubMedGoogle Scholar
- Mattos JP, Rosso AL, Correa RB, Novis SA: Movement disorders in 28 HIV-infected patients. Arq Neuropsiquiatr. 2002, 60: 525-530.View ArticlePubMedGoogle Scholar
- Koppel BS, Daras : "Rubral" Tremor Due to Midbrain Toxoplasma Abscess. Mov Disord. 1990, 5: 254-256. 10.1002/mds.870050314.View ArticlePubMedGoogle Scholar
- Strecker K, Schneider JP, Sabri O, Wegner F, Bergh FT, Schwarz J, Hesse S: Responsiveness to a dopamine agent in Holmes tremor - case report. Eur J Neurol. 2007, 14: e9-e10. 10.1111/j.1468-1331.2006.01665.x.View ArticlePubMedGoogle Scholar
- Micheli F, Graňana N, Scorticati MC, Giannaula RJ, Reboredo G: Unilateral postural and action tremor resulting from thalamic toxoplasmosis in a patient with Acquired Immunodeficiency Syndrome. Mov Disord. 1997, 12 (6): 1096-1098. 10.1002/mds.870120647.View ArticlePubMedGoogle Scholar
- Gajos A, Bogucki A, Schinwelski M, Sołtan W, Rudzińska M, Budrewicz S, Koszewicz M, Majos A, Górska-Chrząstek M, Bieńkiewicz M, Kuśmierek J, Sławek J: The clinical and neuroimaging studies in Holmes tremor. Acta Neurol Scand. 2010Google Scholar
- Velez M, Cosentino M, Torres L: Levodopa-Responsive Rubral (Holmes') Tremor. Mov Disord. 2002, 17 (4): 741-742. 10.1002/mds.10224.View ArticlePubMedGoogle Scholar
- The pre-publication history for this paper can be accessed here:http://0-www.biomedcentral.com.brum.beds.ac.uk/1471-2377/10/37/prepub
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