To report a rare case of a patient presenting with unilateral symptoms whose work-up revealed partial stiff person syndrome (SPS).

Our patient is a 62 year old female with no prior medical problem who presented to the hospital with right arm and leg weakness as well as a body locked sensation where she felt muscle stiffening and could not move.  She also described random, large amplitude involuntary movements either in her right arm or right leg. No symptoms in her left side.  She was admitted for suspected stroke in November 2018 and had extensive workup including MRI brain with contrast which did not show any area of enhancement nor acute DWI. No cause for her symptoms was found, and after discharge continued to have worsened right sided ataxia and hyperreflexia and increased muscle tone.  Further work-up ensued, and it was not until April 2019 that she was found to have an elevated GAD (>250 IU/mL) and HbA1C 6.3 with positive islet cell antibody.  Lumbar puncture results showed positive GAD65 Ab Assay (3.09 nmol/L) and the presence of oligoclonal bands.