A Case of Crying Seizures

To the Editor: Ictal crying (dacrystic seizure) is a rare, paroxysmal phenomenon. Dacrystic seizures may be simple or complex¹ and occur with preserved consciousness. They are most commonly reported in patients with brain lesions, but can also occur in patients with normal brain imaging.^1–7 Luciano et al.¹ identified 11 published cases with dacrystic seizures and reported on seven adult patients with video-EEG documented ictal crying. More recently, six more cases of adult patients^2,3,6 and infants^5,7 were reported.

The lesion topography remains imprecise, but in the majority of cases the ictal activity predominates in the temporal lobe of the nondominant hemisphere.^1–5

We report a case of a patient with ictal and postictal crying associated with fear and anguish, as the cardinal manifestations of ictal activity in the right fronto-polar region, and its implications for differential diagnosis and treatment.

Case Report

Our 37-year-old right-handed female patient, with no history of learning disability or birth complications, had suffered from a single episode of grand-mal 8 years before. She was treated with carbamazepine that was suspended 2 years after, due to alopecia and leucopenia, with no further similar episodes. She was observed in a psychiatric consultation due to episodes of anxiety, anguish, and fear, especially while driving, associated with an urge to cry, but with no feeling of sadness or negative emotional affect. She denied feeling depressed, describing herself as an optimistic and positive person. Mirtazapine (15 mg/day) was started, with no response and intolerance (sedation). Electroencephalography (EEG) was normal during vigilance and after attempted induction with injection of saline solution; during cognitive stimulation (Stroop Test) she developed an ictal and postictal single partial dacrystic crisis. The ictal activity occurred in the right fronto-polar region, showing no propagation. Neurological examination and brain MRI were normal. Mirtazapine was suspended and lamotrigine introduced, up to 200 mg/day, and she has been seizure-free for 1 year.

Discussion

Ictal and postictal crying is an apparently rare phenomenon, associated with marked heterogeneity in clinical, neurophysiological, and imagiological manifestations, most frequently associated to right temporal foci and feelings of fear and sadness.^1,3–5

Although dacrystic crisis usually occurs in patients with cerebral tumors, vascular malformations, hippocampal sclerosis, or cerebral infarction, a small percentage of patients have been reported to have no anatomical alterations,^1,6 as was the case in our patient.

In the healthy brain, each hemisphere controls the mood, inhibiting the contralateral structure; however, it is not known whether this inhibition is reciprocal or independent and whether individual exceptions exist.³ Crying seems to be topographically initiated in the temporal gyrus and right temporal hippocampus, supporting the lateralization hypothesis of negative affects to the nondominant hemisphere.¹ Moreover, the treatment of depression with ECT is more efficacious when applied on the right side.³

In our patient, ictal crying was probably due to the cortical stimulation of the nondominant hemisphere, resulting in paroxysmal activity with negative affect and crying.

The Stroop Test, which measures the capacity to provide adequate responses to incongruent stimuli, activates the anterior cingulate;⁸ this region, localized in the prefrontal cortex, is involved in mental processes and emotional responses. The initiation of a dacrystic crisis during cognitive stimulation evidences the anatomical location of the focus, since hypoactivity in the anterior dorsal cingulate is involved in depressive conditions.⁹

The underestimation of crying as an ictal manifestation, or the fact that it may be obscured by other ictal manifestations, suggests that dacrystic epilepsy may be more common than reported in the literature.¹

This case supports the theories that propose lateralization of feelings, with the dominant hemisphere to negative feelings.^1,3–5 It also illustrates the difficulty of recognizing subtle ictal manifestations, and alerts to neurological pathologies that may manifest with psychiatric semiology without impairment of consciousness, automatisms, or changes in imagiologic or electroencephalographic exams, with diagnostic and therapeutic implications. In our patient, the correct diagnosis was based on the stereotyped constellation of symptoms, supported by epileptic activity detected during an ictal-EEG, as well as by episode resolution with antiepileptic treatment. Treatment of dacrystic seizures varies, but antiepileptic drugs seem to be the most adequate,^1,2,5,7 and in our patient managed ictal manifestations successfully.

This case was presented at the 5th National Congress of the Portuguese Society of Psychiatry, Oporto (Portugal), 2009. The authors declare no potential conflicts of interest relative to the contents of this article. Dr. Sofia Brissos is consultant for Janssen Pharmaceutica-Cilag Portugal. Dr. Vasco Videira Dias is consultant for Angelini Pharmaceuticals and has received educational grants from Sanofi-Aventis and Lundbeck.