View Supplementary Video 1

Hereditary spastic paraplegias (HSP) are a group of genetically heterogeneous diseases characterized by progressive degeneration of the corticospinal tracts. They are clinically differentiated into pure forms, in which the manifestations are restricted to the presence of spasticity and paresis of the lower limbs, with eventual involvement of bladder control and hypopalesthesia, and in complex forms, in which other neurological and clinical signs and symptoms are present. Among these additional manifestations are movement disorders (MD), which occur due to the involvement of the motor system, with involvement of the extrapyramidal and cerebellar systems. The genetic and phenotypic heterogeneity of HSPs contributes to make their diagnosis difficult. Recognizing the main clinical manifestations associated with the complex phenotype of HSP is critical for clinical suspicion. Although useful, classifications based on mode of inheritance and clinical distinctions between pure and complex forms remain confusing to the neurologist. Thus, it may be interesting to reconsider the practical approach and the semiological classification of this disorder by focusing on clinical phenotypes. Numerous publications of case reports and case series linking new neurological and non-neurological manifestations to the phenotype of HSP have been published, but MD, except ataxia, have never been the main subject of discussion in a review. Considering the prevalence of movement disorders in patients with HSP, a Systematic Review on the subject is of great relevance. The present study aimed to carry out a Systematic Review of the literature with the purpose of describing the forms of MD described as a manifestation of HSP. We carried out a systematic search in the literature published in the five electronic databases, PubMed, Embase, Scopus, SciElo and LILACS, which were accessed between September/2020 and January/2021. Search limits were set to include only english-language articles published in peer-reviewed journals during any period. The review considered in the end 93 articles describing 150 patients with HSP and MD for analysis. Most of these cases had disease with autosomal recessive inheritance (85.3%), and 19.3% of them had MD as an initial manifestation. Various forms of dystonia, tremor, parkinsonism, dyskinesias, chorea, and myoclonias were described as clinical presentations of HSP, with dystonia being the most frequent problem (35% of cases). There are cases of parkinsonism, dystonia and tremors described with good response to levodopa. Most of the cases presented alterations in neuroimaging exams that have already been associated with HSP. It was not possible to associate any genetic type of HSP to a specific clinical presentation of MD.

Introduction: Migraine infarction is one of the rare complications of migraine with aura, characterized by the persistence of aura symptoms for more than sixty minutes, associated with ischemic brain damage in the appropriate territory, demonstrated by neuroimaging examination. In spite of several cases reported in the literature correlate cerebral infarction with migraine, it is still unclear which magnetic resonance (MRI) pattern is associated with this condition. Cortical laminar necrosis (CLN) is a type of cortical infarction characterized by selective and late necrosis, especially in the third layer of the cerebral cortex, a region where there is greater vulnerability to metabolic stress. On MRI, CLN is characterized by an increase in cortical signal intensity on T1-weighted images with a typical curvaceous gyriform distribution. The goal of this study was to demonstrate, throughout a literature review, a possible association between migraine with aura, migraine infarction and cortical laminar necrosis without neuroimaging examination.Methods: A systematic research was performed in databases at the PubMed and Embase in February 2021. The search terms used for 'Migraine' or 'Migraine with aura' or 'Migrainous Infarction' combined with 'Cortical Laminar Necrosis', using their respective variations of the according as MeSH and Emtree. It was articles published in English, in pair reviewed journals, during any period and submitted to studies relevant to clinical questions, which revealed cases of Laminar Cortical Necrosis associated with migraine, were included. Studies that do not attempt the criteria, was excluded.Results and Discussion: The search for selected databases resulted in 24 articles. Excluding 14 articles were not chosen because they are not CLN, and 1 article published in conference abstracts that we did not choose for this review. Finally, 8 original studies were selected and critically analyzed in this review. There was a prevalence in hundred percent of the cases analyzed with migraine with aura (Table 1), and all of them manifested additional neurological symptoms. In two cases, the etiology was of familiar legacy of genetic origin, which consisted of a familial hemiplegic migraine. MR was performed days after the begining of symptoms, ranging from the 3rd to the 30th day. In all cases, was observed unilateral cortical hypersignal on axial T1-evaluation. Unilateral T1-evaluation cortical hyperintensity with Gadoliun and FLAIR was also identified in most of the cases. Less commonly, alterations to the DWI and ADC sequences are seen. Conclusion: Despite being a rare manifestation, this review demonstrates that CLN can be recognized as an attribute associated with migraine with aura, spread the spectrum of neuroimaging acts correlated with migraine. Our goal was narrow the boundaries between neurovascular disease on imaging and a migraine with aura, helping neurologists to recognize this association. Before this scene of high predominance of migraines, further studies are needed to elucidate its relationship with CLN, as its pathophysiology and group of patients with risk factors, who may benefit from prophylactic treatment.

No abstract