L-Dopa-responsive Parkinson's syndrome in association with phenylketonuria: In vivo dopamine transporter and D2 receptor findings.
Identifieur interne : 003293 ( PubMed/Curation ); précédent : 003292; suivant : 003294L-Dopa-responsive Parkinson's syndrome in association with phenylketonuria: In vivo dopamine transporter and D2 receptor findings.
Auteurs : Andrew H. Evans [Royaume-Uni] ; Durval C. Costa ; Sveto Gacinovic ; Regina Katzenschlager ; John D. O'Sullivan ; Simon Heales ; Phillip Lee ; Andrew J. LeesSource :
- Movement disorders : official journal of the Movement Disorder Society [ 0885-3185 ] ; 2004.
English descriptors
- KwdEn :
- Adult, Antiparkinson Agents (therapeutic use), Benzamides (diagnostic use), Brain (blood supply), Brain (radionuclide imaging), Dopamine Plasma Membrane Transport Proteins, Female, Humans, Iodine Radioisotopes (diagnostic use), Levodopa (therapeutic use), Membrane Glycoproteins (metabolism), Membrane Transport Proteins (metabolism), Nerve Tissue Proteins (metabolism), Parkinson Disease (complications), Parkinson Disease (drug therapy), Parkinson Disease (metabolism), Phenylketonurias (complications), Pyrrolidines (diagnostic use), Radiopharmaceuticals (diagnostic use), Receptors, Dopamine D2 (metabolism), Tomography, Emission-Computed, Single-Photon, Tropanes (diagnostic use).
- MESH :
- chemical , diagnostic use : Benzamides, Iodine Radioisotopes, Pyrrolidines, Radiopharmaceuticals, Tropanes.
- chemical , metabolism : Membrane Glycoproteins, Membrane Transport Proteins, Nerve Tissue Proteins, Receptors, Dopamine D2.
- chemical , therapeutic use : Antiparkinson Agents, Levodopa.
- blood supply : Brain.
- complications : Parkinson Disease, Phenylketonurias.
- drug therapy : Parkinson Disease.
- metabolism : Parkinson Disease.
- radionuclide imaging : Brain.
- Adult, Dopamine Plasma Membrane Transport Proteins, Female, Humans, Tomography, Emission-Computed, Single-Photon.
Abstract
Reports of parkinsonism in phenylketonuria are exceedingly rare. We report on a patient who had received a delayed diagnosis of phenylketonuria as an infant and subsequently developed levodopa-responsive parkinsonism at the age of 33. Single-photon emission computed tomography (SPECT) using (123)I-FP-CIT ([(123))I]-2 beta-carbomethoxy-3beta-(-4-iodophenyl)-N-(3-fluoropropyl)-nortropane) used to measure dopamine transporter levels on two occasions, 7 and 9 years after the onset of neurological symptoms, were normal. Iodine-123-iodo-lisuride SPECT (IBZM) imaging, however, showed reduced caudate over putamen binding. This combination of imaging findings indicates a possible upregulation of postsynaptic D2 receptors in the context of intact presynaptic dopamine nerve terminal density.
DOI: 10.1002/mds.20146
PubMed: 15390012
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pubmed:15390012Le document en format XML
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<affiliation wicri:level="1"><nlm:affiliation>Reta Lila Weston Institute of Neurological Studies, University College London, United Kingdom.</nlm:affiliation>
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<author><name sortKey="Costa, Durval C" sort="Costa, Durval C" uniqKey="Costa D" first="Durval C" last="Costa">Durval C. Costa</name>
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<author><name sortKey="Katzenschlager, Regina" sort="Katzenschlager, Regina" uniqKey="Katzenschlager R" first="Regina" last="Katzenschlager">Regina Katzenschlager</name>
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<term>Brain (blood supply)</term>
<term>Brain (radionuclide imaging)</term>
<term>Dopamine Plasma Membrane Transport Proteins</term>
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<term>Tomography, Emission-Computed, Single-Photon</term>
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<front><div type="abstract" xml:lang="en">Reports of parkinsonism in phenylketonuria are exceedingly rare. We report on a patient who had received a delayed diagnosis of phenylketonuria as an infant and subsequently developed levodopa-responsive parkinsonism at the age of 33. Single-photon emission computed tomography (SPECT) using (123)I-FP-CIT ([(123))I]-2 beta-carbomethoxy-3beta-(-4-iodophenyl)-N-(3-fluoropropyl)-nortropane) used to measure dopamine transporter levels on two occasions, 7 and 9 years after the onset of neurological symptoms, were normal. Iodine-123-iodo-lisuride SPECT (IBZM) imaging, however, showed reduced caudate over putamen binding. This combination of imaging findings indicates a possible upregulation of postsynaptic D2 receptors in the context of intact presynaptic dopamine nerve terminal density.</div>
</front>
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<Abstract><AbstractText>Reports of parkinsonism in phenylketonuria are exceedingly rare. We report on a patient who had received a delayed diagnosis of phenylketonuria as an infant and subsequently developed levodopa-responsive parkinsonism at the age of 33. Single-photon emission computed tomography (SPECT) using (123)I-FP-CIT ([(123))I]-2 beta-carbomethoxy-3beta-(-4-iodophenyl)-N-(3-fluoropropyl)-nortropane) used to measure dopamine transporter levels on two occasions, 7 and 9 years after the onset of neurological symptoms, were normal. Iodine-123-iodo-lisuride SPECT (IBZM) imaging, however, showed reduced caudate over putamen binding. This combination of imaging findings indicates a possible upregulation of postsynaptic D2 receptors in the context of intact presynaptic dopamine nerve terminal density.</AbstractText>
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