PMC10172006_01
Timeline of the clinical course. sEMG, surface electromyography; DATSCAN, dopamine
transporter scan. Other medications included a group of drugs that did not change during
treatment: lithium carbonate 100 mg, lorazepam 1 mg, flunitrazepam 1.5 mg, fluvoxamine
100 mg, clonazepam 1 mg, and brotizolam 0.25 mg (daily doses). Dates are given for
events that can be confirmed after the first visit to our University Hospital. Black
arrowheads on Home Exercise timeline indicate outpatient rehabilitation after
discharge.
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Astellas Pharma, Japan), clonazepam (Landsen , Sumitomo Pharma, Japan), and brotizolam (Lendormin , Boehringer Ingelheim, Germany). Approximately 6 months after increasing the daily dose of ARP from 3 mg to 6 mg, he complained of trouble walking on planar floors and soon after of difficulties when pushing off the ground (Fig. 1). The patient visited the Department of Neurosurgery of our hospital on March 25, 2019 (Fig. 1). At the start of rehabilitation on April 18, 2019 (Fig. 1), the muscle strength of the bilateral iliopsoas quadriceps, tibialis anterior, and gastrocnemius was almost 4 with manual muscle testing. TSD remained, the patient improved to the point where he was able to receive home care (Fig. 1). This improvement was attributed to a short but continuous rehabilitation program that avoided TSD.
Postsurgical brain MRI scans included axial FLAIR sequences. (A) Brain MRI scan
showing the low-intensity area in the right sphenoid ridge, and the arrow indicates the
location of the tumor after removal. (B) Bidirectional arrows indicate asymmetry in the
shape of the basal ganglia. (C) 123I-FP-CIT SPECT shows visually distorted
tracer accumulation in right posterior putamen despite functionally normal binding
capacity [SBR: right (R)=5.10, left (L)=5.47, average (Ave)=5.29, asymmetry index
(AI)=7.10%]. These findings indicate that there is no loss of dopaminergic nerve
terminals because the quantitative SBR is normal without a decrease in dopamine
transporter density in the striatum.
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The brain tumor (BT) was removed by neurosurgery on April 17, 2019 (Fig. 2A,B). Symbia S, Siemens Medical Solutions, USA) on May 9, 2019, showed that dopamine transporter binding capacity was functionally normal (specific binding ratio, SBR: right=5.10, left=5.47; Fig. 2C). Surface electromyography (EMG; Trigno Avanti Platform; Delsys, Natick, MA, USA) showed the findings of dystonia in his LE (Fig. 4).
TSD during forward walking after brain tumor surgery and after cessation of ARP.
Forward gait transition observed from both frontal (1, 2, 3, 4) and posterior (5, 6, 7,
8) views, on the second postoperative day (A) and after cessation of ARP administration
(B). The forward walking transition is shown in the order 1 to 4 and 5 to 8. Bilateral
lower extremity dystonia is visible during forward gait, with both knees in extension
during the swing phase of the gait cycle, and poor leg kicking. Bilateral dorsiflexion
and toe extension are also observed, and the patient appears to have difficulty pushing
off the ground or walking on tiptoe. Although not recorded, dystonia did not appear
during backward walking or bicycle pedaling. Two months after ARP was discontinued
following discharge from the hospital, the abnormal limb position associated with
dystonia seen in (A) improved during walking (B). Although the gait appeared to be
spastic, a diagnosis of dystonia was made based on findings from neurological
examination and surface electromyography (Fig.
4).
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Department. During the stance phase, he showed difficulties of right ankle inversion, plantar flexion, and dorsiflexion (Fig. 3A). TSD during forward walking remained but showed improvement (Fig. 3B). After discharge, his dystonia still appeared crus predominant in both LE immediately after the start of forward walking.
Dynamic multichannel recordings of surface EMG during forward walking. Surface EMG
results reflected either phasic or tonic co-contraction of agonist and antagonist
muscles in the lower extremities. These were seen only during ambulation and not while
sitting at rest, consistent with (but not diagnostic of) TSD. Bilateral asymmetric
dystonic muscle activity was observed in the tibialis anterior and gastrocnemius
muscles. Different traces (from top to bottom) refer to (A) right tibialis anterior, (B)
right gastrocnemius, (C) left tibialis anterior, and (D) left gastrocnemius.
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Symbia S, Siemens Medical Solutions, USA) on May 9, 2019, showed that dopamine transporter binding capacity was functionally normal (specific binding ratio, SBR: right=5.10, left=5.47; Fig. 2C). Surface electromyography (EMG; Trigno Avanti Platform; Delsys, Natick, MA, USA) showed the findings of dystonia in his LE (Fig. 4).