Aripiprazole-Triggered Alice in Wonderland Syndrome Episodes Studied with 99mTc-HMPAO Brain SPECT

Mancini, Valentina; Mastria, Giulio; Frantellizzi, Viviana; Viganò, Alessandro; Petsas, Nikolaos; Sollaku, Saadi; Fanella, Martina; Bonaventura, Carlo Di; Liberatore, Mauro; Piero, Vittorio Di

doi:10.1159/000490902

Cited by 14 publications

(9 citation statements)

References 2 publications

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“…To date, it remains to be elucidated if a similar brain network is implicated in AIWS caused by a non-structural disease (functional AIWS). Overall, available single-case studies in AIWS with functional neuroimaging correlates pointed to a hypoactivation of frontal regions, together with increased activation of associative parietal and occipital cortices (16)(17)(18)(19)(20). However, the heterogeneity of the clinical presentation, the underlying etiologies, and the variety of the methods employed to measure brain activity (i.e., fMRI, PET and SPECT) in these studies prevents from drawing strong conclusions.…”

Section: Introductionmentioning

confidence: 96%

“…Recently, a voxel-based metanalysis including all cases of AIWS associated with a structural lesion (16)(17)(18)(19)(20), showed that lesions in extrastriate visual cortex are those most frequently associated with AIWS with visual symptoms (21). To date, it remains to be elucidated if a similar brain network is implicated in AIWS caused by a non-structural disease (functional AIWS).…”

Section: Introductionmentioning

confidence: 99%

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Neural fingerprints of Alice in Wonderland Syndrome in patients with migraine with aura

Mastria

Mancini

Viganò

et al. 2022

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Background: the Alice in Wonderland syndrome (AIWS) is a transient neurological disturbance characterized by visual and somatosensory misperceptions most frequently associated with migraine. The heterogeneity of the etiologies and techniques applied to investigate the reported cases have so far prevented to draw strong conclusions about the origin of AIWS symptoms. Some lines of evidence suggest that AIWS and migraine might share common pathophysiological mechanisms, therefore we set out to investigate the common and distinct neurophysiological alterations associated with these conditions in a population of migraineurs. Methods: we acquired resting-state fMRI data from 12 migraine patients with AIWS, 12 patients with migraine with typical aura (MA) and 24 age-matched healthy controls (HC). We then compared the interictal thalamic seed-to-voxel and ROI-to-ROI cortico-cortical resting-state functional connectivity between the 3 groups. Results: we found a common pattern of altered thalamic connectivity in MA and AIWS, compared to HC, with more profound and diffuse alterations observed in AIWS. The ROI-to-ROI functional connectivity analysis highlighted an increased connectivity between a lateral occipital region corresponding to area V3 and the posterior part of the superior temporal sulcus (STS) in AIWS, compared to both MA and HC. The posterior STS is a multisensory integration area, while area V3 is considered the starting point of the cortical spreading depression (CSD), the neural correlate of migraine aura. This interictal hyperconnectivity might increase the probability of the CSD to directly diffuse to the posterior STS or trigger a diaschisis phenomenon causing the AIWS symptoms during the ictal phase. Conclusions: taken together, these results suggest that AIWS in migraineurs might be a form of complex migraine aura, characterized by the involvement of associative and multisensory integration areas. The altered connectivity between early visual and multisensory associative areas provides a model for the pathophysiology of AIWS associated with other transient neurological conditions or with a structural etiology.

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Section: Introductionmentioning

confidence: 96%

Section: Introductionmentioning

confidence: 99%

Neural fingerprints of Alice in Wonderland Syndrome in patients with migraine with aura

Mastria

Mancini

Viganò

et al. 2022

Preprint

Self Cite

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“…In this regard, one notable example is Alice in Wonderland Syndrome (AIWS), a syndrome characterised by visual and somatosensory misperceptions (5–7). The association between AIWS and migraine is mostly anecdotal, since it has been investigated only by case reports or small studies (8–15). Moreover, while migraine is the first cause of AIWS in adults (6,16), most of the studies on AIWS have focused on children (6,17,18).…”

Section: Introductionmentioning

confidence: 99%

Prevalence and characteristics of Alice in Wonderland Syndrome in adult migraineurs: Perspectives from a tertiary referral headache unit

et al. 2020

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Background Migraine affects how the brain processes sensory information at multiple levels. The aberrant integration of visual and somatosensory stimuli is thought to underlie Alice in Wonderland Syndrome, a disorder often reported as being associated with migraine. However, there is still a lack of knowledge about the epidemiology of this syndrome in migraineurs and the association between Alice in Wonderland Syndrome episodes and migraine attacks. Therefore, we conducted a prospective cohort study to systematically evaluate the prevalence and the clinical features of Alice in Wonderland Syndrome in a large sample of patients with migraine. Methods All the patients attending for the first time a tertiary-level headache clinic were consecutively screened for Alice in Wonderland Syndrome symptoms by means of an ad hoc questionnaire and detailed clinical interview, over a period of 1.5 years. Patients experiencing Alice in Wonderland Syndrome symptoms were contacted for a follow-up after 8–12 months. Results Two hundred and ten patients were recruited: 40 patients (19%) reported lifetime occurrence of Alice in Wonderland Syndrome, 90% of whom (38/40) had migraine with aura. Thirty-one patients experienced episodes of Alice in Wonderland Syndrome within 1 h from the start of migraine headache. Patients reported either visual or visual and somatosensory symptoms (i.e. somatosensory symptoms never presented alone). We collected the follow-up details of 30 patients with Alice in Wonderland Syndrome, 18 of whom had been prescribed a preventive treatment for migraine. After 8–12 months, 5 of the treated patients reported a decrease, while 13 reported no episodes of Alice in Wonderland Syndrome. Conclusion Alice in Wonderland Syndrome prevalence in migraineurs was found to be higher than expected. Alice in Wonderland Syndrome was mostly associated with migraine with aura and tended to occur close to the migraine attack, suggesting the existence of a common pathophysiological mechanism. Patients treated with migraine preventive treatments had a higher chance of decreasing or even resolving Alice in Wonderland Syndrome episodes.

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“…In this line, a second case 3 , of a 47-year-old woman, who suffered from MA may help clarifying how AIWS and MA are related. The patient had visual MA since her adolescence.…”

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confidence: 95%

Adding pieces to the Alice in wonderland syndrome puzzle: a comment to the paper by Brooks and colleagues

Viganò

Maestria

Mancini

et al. 2020

Arq. Neuro-Psiquiatr.

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Aripiprazole-Triggered Alice in Wonderland Syndrome Episodes Studied with 99mTc-HMPAO Brain SPECT

Cited by 14 publications

References 2 publications

Neural fingerprints of Alice in Wonderland Syndrome in patients with migraine with aura

Neural fingerprints of Alice in Wonderland Syndrome in patients with migraine with aura

Prevalence and characteristics of Alice in Wonderland Syndrome in adult migraineurs: Perspectives from a tertiary referral headache unit

Adding pieces to the Alice in wonderland syndrome puzzle: a comment to the paper by Brooks and colleagues

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