Late-onset chorea after cerebral revascularization as a clinical manifestation of moyamoya disease

Enríquez-Ruano, Pilar; Navarro, Cristian Eduardo; Penagos, Natalia; Espitia, Oscar Mauricio

doi:10.1007/s10072-021-05189-z

Cited by 1 publication

(3 citation statements)

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“…e information would also be In one of the six patients, the involuntary movements resolved spontaneously, whereas haloperidol was effective in completely resolving it in the other five cases. [1,4,14] On the other hand, clonazepam was not effective in our first case. It is known that a certain subgroup of adult patients with stroke may develop involuntary movements.…”

Section: Discussioncontrasting

confidence: 55%

“…Thus, the late-onset, first-ever involuntary movement in pediatric moyamoya disease would be quite rare. [ 1 , 4 , 14 ] Clinical data for the four previously reported cases and the two cases reported here are summarized in Table 1 . All of them are pediatric cases.…”

Section: Discussionmentioning

confidence: 99%

“…[ 8 ] However, involuntary movements that did not develop before surgery may first appear postoperatively in very rare cases. [ 1 , 4 , 14 ]…”

Section: Introductionmentioning

confidence: 99%

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Late-onset, first-ever involuntary movement after successful surgical revascularization for pediatric moyamoya disease – Report of two cases

Hasegawa,

Tanaka,

Yamamoto

et al. 2023

Surgical Neurology International

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Background: A small number of children with Moyamoya disease develop involuntary movements as an initial presentation at the onset, which usually resolves after effective surgical revascularization. However, involuntary movements that did not occur at the onset first occur after surgery in very rare cases. In this report, we describe two pediatric cases that developed involuntary movements during the stable postoperative period after successful surgical revascularization. Case Description: A 10-year-old boy developed an ischemic stroke and successfully underwent combined bypass surgery. However, he developed chorea six months later. Another 8-year-old boy developed a transient ischemic attack and successfully underwent combined bypass surgery. However, he developed chorea three years later. In both cases, temporary use of haloperidol was quite effective in resolving the symptoms, and involuntary movements did not recur without any medication during follow-up periods of up to 10 years. Conclusion: Postoperative first-ever involuntary movement is very rare in pediatric moyamoya disease, and the underlying mechanisms are still unclear, but a temporary, reversible imbalance of excitatory and inhibitory circuits in the basal ganglia may trigger the occurrence of these rare symptoms. Careful follow-up would be mandatory.

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Section: Discussioncontrasting

confidence: 55%