Limb-shaking TIA in Moyamoya angiopathy.


Journal

Clinical neurology and neurosurgery
ISSN: 1872-6968
Titre abrégé: Clin Neurol Neurosurg
Pays: Netherlands
ID NLM: 7502039

Informations de publication

Date de publication:
08 2021
Historique:
received: 07 05 2021
revised: 06 06 2021
accepted: 18 06 2021
pubmed: 6 7 2021
medline: 22 1 2022
entrez: 5 7 2021
Statut: ppublish

Résumé

Limb shaking TIA(LS-TIA) is an rare manifestation seen in high grade steno-occlusive carotid disease. LS-TIA is rarely recognized as a presenting manifestation of MMA and has never been previously studied prospectively in MMA cohort. Of 104 cases of MMA collected in our Indian center in last 4 years, 5 new patients had LS-TIA. MMA was diagnosed by MR angiography and LS-TIA was confirmed by three different neurologist by detailed scrutiny of history, neurological examination and EEG-recording (to rule out seizure). Seven previously reported cases of LS-TIA in MMA were retrieved by literature search in PubMed using keywords "Moyamoya" AND "Limb shaking TIA", "Moyamoya" AND "Limb shaking". Subsequently all data were analyzed and compared using descriptive statistics. Analysis of our 5 cases and those 7 found in the literature showed mean age at the time of diagnosis of MMA was 12.2 ± 8.9(Mean ± S.D.) years in our cases and 32.7 ± 13.6 years in previous cases. The commonest precipitating factors included prolonged crying and hyperventilation(3 of 5 our cases each) and exercise (3 of 7 previous cases). CT-perfusion demonstrated frontal white matter hypoperfusion in 2 of our 5 cases, corroborative with fronto-parietal hypoperfusion in previous cases. Medical management showed decreased symptoms in 4 of our 5 cases in a follow-up of 15.8 ± 11.6 months, while all previous patients had underwent revascularization surgery with resolution of symptoms. LS-TIA in MMA may not be so uncommon, especially in children. Early recognition is imperative to avoid future catastrophe. Steno-occlusive posterior circulation lesions might be an independent predictor of development of LS-TIA in MMA. In a resource-limited setting, careful avoidance of triggers and anti-platelet therapy can lead to decreased symptomatology of LS-TIA in MMA.

Sections du résumé

BACKGROUND
Limb shaking TIA(LS-TIA) is an rare manifestation seen in high grade steno-occlusive carotid disease. LS-TIA is rarely recognized as a presenting manifestation of MMA and has never been previously studied prospectively in MMA cohort.
METHODS
Of 104 cases of MMA collected in our Indian center in last 4 years, 5 new patients had LS-TIA. MMA was diagnosed by MR angiography and LS-TIA was confirmed by three different neurologist by detailed scrutiny of history, neurological examination and EEG-recording (to rule out seizure). Seven previously reported cases of LS-TIA in MMA were retrieved by literature search in PubMed using keywords "Moyamoya" AND "Limb shaking TIA", "Moyamoya" AND "Limb shaking". Subsequently all data were analyzed and compared using descriptive statistics.
RESULTS
Analysis of our 5 cases and those 7 found in the literature showed mean age at the time of diagnosis of MMA was 12.2 ± 8.9(Mean ± S.D.) years in our cases and 32.7 ± 13.6 years in previous cases. The commonest precipitating factors included prolonged crying and hyperventilation(3 of 5 our cases each) and exercise (3 of 7 previous cases). CT-perfusion demonstrated frontal white matter hypoperfusion in 2 of our 5 cases, corroborative with fronto-parietal hypoperfusion in previous cases. Medical management showed decreased symptoms in 4 of our 5 cases in a follow-up of 15.8 ± 11.6 months, while all previous patients had underwent revascularization surgery with resolution of symptoms.
CONCLUSION
LS-TIA in MMA may not be so uncommon, especially in children. Early recognition is imperative to avoid future catastrophe. Steno-occlusive posterior circulation lesions might be an independent predictor of development of LS-TIA in MMA. In a resource-limited setting, careful avoidance of triggers and anti-platelet therapy can lead to decreased symptomatology of LS-TIA in MMA.

Identifiants

pubmed: 34225004
pii: S0303-8467(21)00312-7
doi: 10.1016/j.clineuro.2021.106783
pii:
doi:

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

106783

Informations de copyright

Copyright © 2021 Elsevier B.V. All rights reserved.

Auteurs

Shambaditya Das (S)

Department of Neurology, Institute of Post Graduate Medical Education & Research, Bangur Institute of Neurosciences, Kolkata, India. Electronic address: drshambadityadas@gmail.com.

Ritwik Ghosh (R)

Department of General Medicine, Burdwan Medical College and Hospital, Burdwan, India. Electronic address: ritwikmed2014@gmail.com.

Souvik Dubey (S)

Department of Neurology, Institute of Post Graduate Medical Education & Research, Bangur Institute of Neurosciences, Kolkata, India. Electronic address: drsouvik79@gmail.com.

Alak Pandit (A)

Department of Neurology, Institute of Post Graduate Medical Education & Research, Bangur Institute of Neurosciences, Kolkata, India. Electronic address: dralakpandit@gmail.com.

Biman Kanti Ray (BK)

Department of Neurology, Institute of Post Graduate Medical Education & Research, Bangur Institute of Neurosciences, Kolkata, India. Electronic address: biman.kanti@rediffmail.com.

Markus Kraemer (M)

Department of Neurology, Alfried Krupp Von Bohlen Und Halbach, Hospital, Alfried-Krupp-Str. 21, 45117 Essen, Germany; Heinrich Heine University of Duesseldorf, Medical Faculity, Duesseldorf, Germany. Electronic address: markus.kraemer@krupp-krankenhaus.de.

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