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Detection of Subclinical Cases Using ¹⁸F-Dopa and ¹⁸Fluorodeoxyglucose Positron Emission Tomography

Paola Piccini, MD; Justo de Yebenez, MD; Andrew J. Lees, MD; Roberto Ceravolo, MD; Nora Turjanski, MD; Peter Pramstaller, MD; David J. Brooks, MD

Arch Neurol. 2001;58:1846-1851.

Background  Progressive supranuclear palsy (PSP) is generally considered to be a sporadic disease; however, occasional cases of familial PSP have been described. The rarity of reports of familial PSP may be attributed in part to an inability to detect subclinical disease in affected relatives who subsequently die before symptoms clinically develop.

Objective  To study regional cerebral dopaminergic function and glucose metabolism in members of 2 large kindreds with familial PSP to identify subclinical cases.

Methods  Three clinically affected members from the 2 PSP kindreds were scanned with both ¹⁸F-dopa and ¹⁸fluorodeoxyglucose (¹⁸FDG) positron emission tomography (PET). Fifteen asymptomatic first-degree relatives were scanned with ¹⁸F-dopa PET; 10 of them also underwent a second PET study with ¹⁸FDG.

Results  All 3 clinically affected PSP patients showed a significant reduction in caudate and putamen ¹⁸F-dopa uptake along with a significant reduction in striatal, lateral, and medial premotor area and dorsal prefrontal cortex glucose metabolism. In 4 of the 15 asymptomatic relatives, caudate and putamen ¹⁸F-dopa uptake was 2.5 SDs lower than the normal mean. These 4 subjects and a fifth asymptomatic relative with normal ¹⁸F-dopa uptake showed a significant reduction of cortical and striatal glucose metabolism in a pattern similar to that of their affected relatives.

Conclusion  ¹⁸F-dopa and ¹⁸FDG PET allowed us to identify 5 cases with subclinical metabolic dysfunction among 15 subjects (33%) at risk for PSP, suggesting that this approach is useful for characterizing the pattern of aggregation in PSP kindreds.

From the Medical Research Council Clinical Sciences Centre, Imperial College School of Medicine, Hammersmith Hospital, London, England (Drs Piccini, Ceravolo, Turjanski, and Brooks); Department of Neurology, Fundacion Jimenez Diaz, Universidad Autonoma de Madrid, Madrid, Spain (Dr de Yebenez); Institute of Neurology, Queen Square, London (Drs Lees and Brooks); and the Department of Neurology, Regional General Hospital, Bolzano, Italy (Dr Pramstaller).

Corresponding author and reprints: Paola Piccini, MRC Clinical Science Center, Cyclotron Unit, Hammersmith Hospital, DuCane Road, W12 ONN London, England (e-mail: paola.piccini{at}csc.mrc.ac.uk).

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