A case of recurrent depressive disorder presenting with Alice in Wonderland syndrome: psychopathology and pre- and post-treatment FDG-PET findings

Abstract

BackgroundAlice in Wonderland syndrome (AIWS) is a rare neuropsychiatric syndrome that typically manifests in distortion of extrapersonal visual image, altered perception of one’s body image, and a disturbed sense of the passage of distance and time. Several conditions have been reported to contribute to AIWS, although its biological basis is still unknown. Here, we present the first case demonstrating a clear concurrence of recurrent depressive disorder and AIWS. The clinical manifestations and pre- and post-treatment fluorodeoxyglucose positron-emission tomographic (FDG-PET) images provide insights into the psychopathological and biological basis of AIWS.Case presentationWe describe a 63-year-old Japanese male who developed two distinct episodes of major depression concurrent with AIWS. In addition to typical AIWS perceptual symptoms, he complained of losing the ability to intuitively grasp the seriousness of news and the value of money, which implies disturbance of high-order cognition related to estimating magnitude and worth. Both depression and AIWS remitted after treatment in each episode. Pre-treatment FDG-PET images showed significant hypometabolism in the frontal cortex and hypermetabolism in the occipital and parietal cortex. Post-treatment images showed improvement of these abnormalities.ConclusionsThe clinical co-occurrence of depressive episodes and presentation of AIWS can be interpreted to mean that they have certain functional disturbances in common. In view of incapacity, indifference, devitalization, altered perception of one’s body image, and disturbed sense of time and space, the features of AIWS analogous to those of psychotic depression imply a common psychopathological basis. These high-order brain dysfunctions are possibly associated with the metabolic abnormalities in visual and parietotemporal association cortices that we observed on the pre- and post-treatment FDG-PET images in this case, while the hypometabolism in the frontal cortex is probably associated with depressive symptoms.

Atypical Symptoms in Migraine-Related Alice in Wonderland Syndrome: Expansion of the Phenotype and Reflection on the Pathomechanism

“…AIWS patients have shown regional cortical dysfunction in either occipital-parietal, occipital-temporal or frontal-parietal areas on functional neuroimaging. [5][6][7][8][9] Interestingly, abnormal perfusion or metabolism were identified in the parietal lobes of patients with visual distortion of size or shape of objects, and in the temporal lobe of a patient with color misrecognition. 9 The abnormal parietal and temporal association cortex corresponded to the dorsal and ventral stream of high-level visual processing, respectively.…”

Alice in Wonderland syndrome (AIWS): a research overview

“…A lack of primary studies and a poor representation of low-income countries provide several insights on the research trends in the realm of AIWS. One possible explanation for these trends is the rarity of AIWS in the general population [4] , [33] , [34] , which may have contributed to a low number of studies globally with even a lower number of studies from low-income countries. Another challenge can be a lack of research capacities in those countries that share a major proportion of the global burden of diseases yet have fewer publications on critical health problems [35] – [37] .…”

Alice in Wonderland syndrome (AIWS): A research overview

“…The copyright holder for this preprint this version posted August 15, 2020. ; https://doi.org/10.1101/2020.08.12.20173815 doi: medRxiv preprint trends is the rarity of AIWS in the general population [4,25,26], which may have contributed to a low number of studies globally with even a lower number of studies from low-income countries.…”

“…A lack of primary studies and a poor representation of low-income countries provide several insights on the research trends in the realm of AIWS. One possible explanation for these trends is the rarity of AIWS in the general population [4, 25, 26], which may have contributed to a low number of studies globally with even a lower number of studies from low-income countries. Another challenge can be a lack of research capacities in those countries that share a major proportion of the global burden of diseases yet have fewer publications on critical health problems [27–29].…”