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A case report of Alice in Wonderland Syndrome caused by temporal–occipital glioblastoma in a patient with a long-time history of migraine without aura

A case report of Alice in Wonderland Syndrome caused by temporal–occipital glioblastoma in a patient with a long-time history of migraine without aura A case report of Alice in Wonderland Syndrome caused by temporal–occipital glioblastoma in a patient with a long-time history of migraine without aura
A case report of Alice in Wonderland Syndrome caused by temporal–occipital glioblastoma in a patient with a long-time history of migraine without aura A case report of Alice in Wonderland Syndrome caused by temporal–occipital glioblastoma in a patient with a long-time history of migraine without aura

A 54-year old man was presented to the Emergency Department (ED) with a long-time history of migraine without aura. He developed neurological symptoms gradually. He complained that the desktop icon of his computer screens appeared nearer, moving towards his right visual field. These visible symptoms appeared for 10 minutes, and then he developed sensory aphasia that lasted almost an hour and disappeared shortly. All these symptoms were transient and grew gradually. These symptoms were further followed by pulsating, intense headache accompanied by nausea and photophobia. He was diagnosed with AIWS due to a glioblastoma located in the left temporal– occipital junction, and to date, it is the first case of AIWS caused by glioblastoma.

What will be most likely explanation for the pathogenesis of Alice in Wonderland Syndrome in the present case?

  • Temporal–occipital glioblastoma
  • Migraine with aura
  •  Seizures 

See All

Key take away

Glioblastoma multiforme (GBM) is a highly malignant form of a brain tumour presented with focal neurological symptoms and signs of raised intracranial pressure as a reduced level of consciousness, seizures and headache. 

Medical history

The patient did not show any medical history for related diseases except episodic migraines occurring irregularly in the previous years with a mean of two attacks per month. He had a family history of gliomas. Brain MRI was performed three years before to exclude secondary headaches were normal.

Examination & lab investigations

The patient was admitted to ED, and a neurological exam was performed. Standard electroencephalography (EEG) showed normal results. Unenhanced brain computed tomography (CT), cerebrospinal fluid and laboratory blood examination showed no alterations. The patient was shifted to the Neurology Department. The neurological exam remained unchanged even after one day, and the patient confirmed that headache following the transient symptom was similar to his usual migraine. It was the first time he experienced an aura-like phenomenon. Brain MRI revealed a 2.4 × 2.5 cm cortical-subcortical temporal–occipital lesion in the left hemisphere, positive in diffusion-weighted imaging (DWI), with inhomogeneous gadolinium enhancement. The examination was completed by MR spectroscopy and tractography. The space-occupying lesion was identified as a possible high-grade glioma. 

Management

The patient underwent surgery, and the glioma mass was removed en bloc with a laser. No postoperative complications were reported. Histological examination showed an incidence of glioblastoma with primitive neuroectodermal tumour (PNET) component.

He was advised to start antiepileptic therapy (valproate 1000 mg daily) immediately after the surgery. The patient recovered after a few days, and he was discharged after a few days in good condition without neurological deficits. Outpatients clinic visits were planned to administrate temozolomide and radiotherapy. The patient underwent neurosurgery again after one year for removing a relapse of glioma in the same site. A secondly generalised seizure preceded him with language disturbance without visual alterations. He reported no features of AIWS after the first episode. Levetiracetam (1000 mg daily) was added to valproate. Twenty months after the first AIWS episode, the patient is alive, with no evidence of disease and in good clinical condition.

Discussion

It is the first reported case of AIWS associated with GBM. The patient had a transient visual disorder characterised by kinetopsia (objects appearing moving while they are still) and pelopsia (objects appearing nearer than they are) as initial clinical presentation of a GBM in the left occipital-temporal area. These symptoms were followed by sensory aphasia and headache, indicating the diagnosis of migraine with aura. It was the first episode; therefore, a definite diagnosis of migraine with aura cannot be made. Migraine is the prevalent neurological disease, affecting about 15% of the global population. Among these, 30% of migraineurs experienced transient neurological deficits as migraine aura. The differential diagnosis between migraine aura and its mimics may be challenging because aura features are variable among migraine patients. A brain MRI was performed due to the occurrence of symptoms of migraine with aura. It showed the presence of brain lesion in the temporal-occipital carrefour. This area is altered in most of the cases reporting AIWS following localised damage. The lesions affect the right hemisphere in most of the cases, but abnormalities on the left side have also been reported. The direct effect of intracranial space-occupying lesion involving the temporal-parietal-occipital multimodal area can be described as the most reasonable hypothesis that can trigger AIWS. The patient reported only one episode of AIWS, which did not appear in the disease-free period after removal of GBM nor during the tumour relapse. Another explanation can be the occurrence of a seizure caused by irritation of cortex induced by GBM, but this possibility seems less probable. Apart from negative EEG at the admission, symptoms developed slowly with a gradual symptom-to-symptom progression lasting for about 1 hr.

Moreover, symptoms were followed by a headache of migrainous characteristics in due time. Headache accompanied by an epileptic seizure is a rare event, and it lacks typical features of migraine. The occurrence of cortical spreading depolarization is an intriguing hypothesis. There is no data on the relationship between brain tumours and CSD; the latter can be induced in animal models by an increase of extracellular potassium, a direct consequence of neural damage. Therefore, the neuronal compression and the damage caused by GBM may trigger an aura-mimic mechanism leading to AIWS symptoms. Lesional AIWS seems to be related to a dysfunction in the parietal-temporal-occipital region, so attention should be paid in the area in AIWS cases occurring without signs of systemic alteration (e.g., encephalitis, drug abuse, etc…) 

Learning

A careful diagnostic workup should always be suggested in the first-ever occurrence of migraine aura with symptoms related to the inclusion of multimodal areas and also in patients with a long history of migraine so that the aura mimicking phenomenon or underlying pathologies such as high-grade gliomas could be excluded. 

References

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Source:

Neurocase.2018; 24: 5-6.

Article:

Temporal-occipital glioblastoma presenting with Alice in Wonderland Syndrome in a patient with a long-time history of migraine without aura

Authors:

Mastria, G. et al.

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