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Progressive apraxic agraphia with micrographia presenting as corticobasal syndrome showing extensive Pittsburgh compound B uptake.

Abstract
A 65-year-old woman developed progressive apraxic agraphia, characterized by poorly formed graphemes, a kanji (Japanese morphograms) recall impairment, relatively preserved oral spelling of kanji characters, and incorrect stroke sequences on writing accompanied by micrographia over a 3-year period. She also showed minor degrees of rigidity, limb-kinetic apraxia, and ideomotor apraxia of the left hand. Although asymmetric rigidity and limb-kinetic apraxia strongly suggested corticobasal degeneration, (11)C-Pittsburgh compound B positron emission tomography (PiB-PET) showed the predominantly right-sided accumulation of amyloid β in the cortices and striatum. (18)F-fluoro-deoxy-glucose PET and single photon emission computed tomography with a (99m)Tc-ethylcysteinate dimer (ECD-SPECT) also revealed predominantly right-sided hypometabolism and hypoperfusion in the primary sensorimotor cortex, posterior cingulate gyrus, temporoparietal cortices, frontal cortices, thalamus, and basal ganglia, a pattern characteristic of both corticobasal degeneration and Alzheimer's disease. The findings suggest that progressive apraxic agraphia with micrographia presenting as corticobasal syndrome can show an Alzheimer's disease pathology. It is also suggested that ideomotor apraxia of the left hand can occur without a callosal lesion, and is caused by hypometabolism or hypoperfusion in the right frontal and parietal cortices, as revealed by PET and SPECT.
AuthorsYasuhisa Sakurai, Kenji Ishii, Masahiro Sonoo, Yuko Saito, Shigeo Murayama, Atsushi Iwata, Kensuke Hamada, Izumi Sugimoto, Shoji Tsuji, Toru Mannen
JournalJournal of neurology (J Neurol) Vol. 260 Issue 8 Pg. 1982-91 (Aug 2013) ISSN: 1432-1459 [Electronic] Germany
PMID23589191 (Publication Type: Journal Article, Research Support, Non-U.S. Gov't)
Chemical References
  • 2-(4'-(methylamino)phenyl)-6-hydroxybenzothiazole
  • Aniline Compounds
  • Apolipoproteins E
  • Organotechnetium Compounds
  • Radiopharmaceuticals
  • Thiazoles
  • technetium Tc 99m bicisate
  • Cysteine
Topics
  • Aged
  • Agraphia (diagnostic imaging, physiopathology)
  • Aniline Compounds
  • Apolipoproteins E (cerebrospinal fluid)
  • Apraxias (diagnostic imaging, physiopathology)
  • Basal Ganglia Diseases (diagnostic imaging, physiopathology)
  • Brain (pathology)
  • Cerebral Cortex (diagnostic imaging, physiopathology)
  • Cysteine (analogs & derivatives)
  • Disease Progression
  • Executive Function
  • Female
  • Handwriting
  • Humans
  • Image Processing, Computer-Assisted
  • Intelligence Tests
  • Neurodegenerative Diseases (diagnostic imaging, physiopathology)
  • Neurologic Examination
  • Neuropsychological Tests
  • Organotechnetium Compounds
  • Positron-Emission Tomography
  • Radiopharmaceuticals
  • Reading
  • Thiazoles
  • Tomography, Emission-Computed, Single-Photon

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