Síndrome de Alicia en el país de las maravillas secundario a consumo de montelukast. Presentación de caso / Alice in wonderland syndrome secondary to medication with montelukast. Case presentation

RESUMEN La condición neurológica definida por la aparición de alteraciones en la percepción, usualmente interpretada como fenómenos extraños de metamorfosis y despersonalización, se reconoce como síndrome de Alicia en el país de las maravillas. Se presenta el caso de una paciente femenina de 9 años de edad, con el diagnóstico de síndrome de Alicia en el país de las maravillas secundario a medicación crónica con montelukast. El diagnóstico del síndrome psiquiátrico se realizó teniendo en consideración los antecedentes patológicos personales y el examen físico. El síndrome de Alicia en el país de las maravillas tiene un carácter benigno, sumamente infrecuente, y aunque su etiología no es del todo conocida, su aparición como reacción adversa a medicamentos es una opción que debe ser siempre considerada por el médico actuante (AU).

ABSTRACT The neurological condition defined by the appearance of alterations in perception usually interpreted as strange phenomena of metamorphosis and depersonalization is recognized as Alice in wonderland syndrome. The case of a 9-year-old female patient is presented, with the diagnosis of Alice in Wonderland syndrome secondary to chronic medication with montelukast. The diagnosis of the psychiatric syndrome was made taking into account personal pathological history and physical examination. Alice in Wonderland syndrome has a benign, extremely rare character and although its etiology is not fully known, its appearance, as an adverse reaction to medications, is an option that should always be considered by the acting physician (AU).

Síndrome de Alice no país das maravilhas e reativação de infeção por Epstein-Barr / Alice in wonderland syndrome and reactivation of infection by Epstein-Barr

INTRODUÇÃO: O síndrome de Alice no País das Maravilhas (SAPM) é uma entidade rara que pode ocorre no contexto de várias condições clínicas, sendo a infeção por vírus Epstein-barr (EBV) a mais comum nas causas infeciosas. Apresenta-se um caso de SAPM associado a infeção a EBV alertando para a necessidade de investigação etiológica destes casos. RELATO DE CASO: Criança de 8 anos, com síndrome de Asperger que, no contexto de amigdalite aguda e febre, surgiu com episódios paroxísticos de alguns minutos de metamorfopsias (macro e micropsia), distorção da perceção das vozes e sensação de medo. A ressonância magnética e o eletroencefalograma foram normais, e o exame citoquímico do líquor foi normal mas a polimerase chain reaction (PCR) foi positiva para vírus EBV. As serologias para EBV, repetidas 3 e 10 semanas após a avaliação inicial, confirmaram uma reativação da infeção por este agente. O doente ficou assintomático após 2 semanas e não houve recidivas. CONCLUSÕES: A investigação de metamorfopsias ou síndrome de SAPM é mandatória pois podem indiciar patologia grave, nomeadamente lesão cerebral ou epilepsia focal. Embora a doença seja rara a etiologia infecciosa deve ser excluída mesmo em doentes com perturbação prévia do comportamento.

INTRODUCTION: Alice in wonderland syndrome (AWS) is a rare condition which may occur as a sign of multiple conditions, with the most frequent infectious etiology being Epstein-barr virus (EBV) infection. We present a case of an AWS caused by EBV infection to alert for the need to investigate these cases. CASE REPORT: 8-year-old boy with Asperger syndrome who developed, in the context of tonsillitis and fever, brief paroxystic episodes of metamorfopsias (macro and micropsia), with voice perception distortion and fear. Physical exam was otherwise normal. Brian magnetic resonance and electroencephalogram were normal, liquor cytochemical exam was normal but Epstein-barr virus (EBV) polimerase chain reaction was positive. EBV blood serologies, repeated 3 and 10 weeks after the initial evaluation, confirmed the reactivation of this agent's infection. Symptoms succumbed 2 weeks after its beginning, with no relapses. CONCLUSIONS: Metamorfopsias or AWS impose etiological investigation because they may occur due to severe disease, namely cerebral lesion or focal epilepsy. Although it is a rare disease, infectious causes should be excluded, even in patients with previous disturbed behavior.

Evidence for the Perception of Time Distortion During Episodes of Alice in Wonderland Syndrome.

Alice in Wonderland syndrome (AIWS) is a rare perceptual disorder associated with sensation of one or several visual and/or auditory perceptual distortions including size of body parts, size of external objects, or passage of time (either speeding up or slowing down). Cause for AIWS is yet to be widely agreed, and the implications are widely varied. One of the research difficulties is the brevity of each episode, typically not exceeding few tens of minutes. This article presents a male adult in late 20s who has apparently experienced AIWS episodes since childhood, and infection has been ruled out. Reaction speed tests were conducted during and after AIWS episodes, across a span of 13 months. Statistically significant evidence is present for delayed response time during AIWS episodes when the patient claims to experience a sensation of time distortion: where events seem to move faster and people appear to speak quicker.

The Alice in Worderland Syndrome.

The Alice in Wonderland Syndrome (AWS) was first described more than 60 years ago by Lippman. It refers to episodes during which an individual may variously experience (as did Alice during her time in Wonderland) somatic, visuo-perceptual and/or visuo-spatial hallucinations, as well as feelings of depersonalisation, derealisation and distorted sense of time. Although the prevalence of AWS is unknown, indirect evidence from both retrospective and prospective studies suggests that it is a rare disorder. This paper describes the case of Zoe, a right-handed, native English speaker who was age 45 years when she experienced an episode of AWS. On neuropsychological assessment, Zoe demonstrated notable impairment of attention, learning and recall (particularly for visuo-spatial information) as well as executive dysfunction (viz., impairment of planning, cognitive flexibility and abstraction), consistent with fronto-temporal dysfunction. Detailed profiles of neuropsychological impairment in the context of AWS have not previously been reported, and it is unclear if such impairment is, indeed, a central and characteristic feature of AWS.

Alice in Wonderland Syndrome: A Clinical and Pathophysiological Review.

Alice in Wonderland Syndrome (AIWS) is a perceptual disorder, principally involving visual and somesthetic integration, firstly reported by Todd, on the literary suggestion of the strange experiences described by Lewis Carroll in Alice in Wonderland books. Symptoms may comprise among others aschematia and dysmetropsia. This syndrome has many different etiologies; however EBV infection is the most common cause in children, while migraine affects more commonly adults. Many data support a strict relationship between migraine and AIWS, which could be considered in many patients as an aura or a migraine equivalent, particularly in children. Nevertheless, AIWS seems to have anatomical correlates. According to neuroimaging, temporoparietal-occipital carrefour (TPO-C) is a key region for developing many of AIWS symptoms. The final part of this review aims to find the relationship between AIWS symptoms, presenting a pathophysiological model. In brief, AIWS symptoms depend on an alteration of TPO-C where visual-spatial and somatosensory information are integrated. Alterations in these brain regions may cause the cooccurrence of dysmetropsia and disorders of body schema. In our opinion, the association of other symptoms reported in literature could vary depending on different etiologies and the lack of clear diagnostic criteria.

Hallucinations and illusions in migraine in children and the Alice in Wonderland Syndrome.

DESIGN: A prospective observational study over 1 year. SETTING: A District General Hospital, and Child and Adolescent Mental Health Department. PATIENTS: Children aged 8-18 years living in the catchment area of a district hospital service with any type of unexplained hallucinations or illusions associated with or without an established diagnosis of migraine. RESULTS: The study identified nine children with a combination of migraine and a variety of hallucinations and illusions, including illusions of size, time, colour, body shape, movement and visual and auditory hallucination. An average of 10 symptoms (range 7-15) were reported. INTERVENTIONS: None. MAIN OUTCOME MEASURE: None. CONCLUSIONS: It is important to recognise these symptoms to enable appropriate history taking and diagnosis. These symptoms are common and currently seem to go unrecognised and may pose diagnostic difficulties if onset is before typical migraine headaches occur.

Alice in Wonderland syndrome as aura of migraine.

Alice in Wonderland syndrome (AIWS), named for Lewis Carroll's titular character, is a disorder characterized by transient episodes of visual hallucinations and perceptual distortions, during which objects or body parts are perceived as altered in various ways (metamorphopsia), including enlargement (macropsia) or reduction (micropsia) in the perceived size of a form. Migraine aura is a transient neurological symptom that most commonly involves the visual fields and occurs before the headache phase. Aura symptoms include the perception of flashing lights that begin in the center of vision and expand in jagged patterns out into the periphery. Symptoms may be somatosensory, such as numbness and tingling in the lips or fingers. They may also involve a profound alteration of the perception of space and time (the "Alice in Wonderland" syndrome). In this article, we present a child had Alice in Wonderland syndrome as aura of migraine.

The study of time perception in migraineurs.

OBJECTIVE: The study aimed to explore the impairment of time perception in migraineurs. BACKGROUND: Headache is the most common pain syndrome in middle-aged adults, and migraine is highly prevalent and severely disabling. Although the mechanisms of and the therapies for migraines have long been explored, less is known about the functional impairments associated with them, especially the impairment in time perception, that is, the ability to estimate the passage of time. METHODS: In this study, we used a temporal reproduction task to assess the estimation of the duration of visual stimulus in 27 migraine patients. The stimulus was delivered at different intervals over the milliseconds and seconds range. RESULTS: In the setting of an interstimulus interval for 1 second and an interstimulus interval for 5 seconds in the 600-millisecond-duration reproduction task, the migraineurs showed impairment in time perception, and in that they significantly overestimated the duration, as compared with the healthy subjects. When compared with the healthy controls for the 3-second and 5-second duration reproduction task, migraineurs in the setting of an interstimulus interval for 1 second and an interstimulus interval for 5 seconds did not show impairment in time perception. CONCLUSIONS: This study indicates that not only is time perception impaired in migraineurs, but that this impairment is exhibited for durations in the milliseconds range, and not the seconds range.