Throughout most recorded history, in diverse societies, people with epilepsy have been viewed as “different” in a negative way (1). Babylonian cuneiform tablets (circa 1067–1046 BC) attribute epilepsy to possession by demons and ghosts (2). Hippocrates (3) wrote the first book on epilepsy, on the sacred disease, around 400 BC. He recognized epilepsy as a brain disorder, repudiating the view that seizures were a curse from the gods or that people with epilepsy had prophetic powers.

However, even Hippocrate’s authority could not remove the superstition and intense stigma surrounding epilepsy. For the next two millennia, epilepsy was considered a curse of the gods, possession, or other supernatural etiologies. The stigma of epilepsy as an evil possession led to social isolation for the people with epilepsy and often their family. Possession, magic, and witchcraft were dominant themes applied to the diagnosis and treatment of epilepsy, during
the Middle Ages. The witch-hunting handbook, Malleus Maleficarum (1487), written by two Dominican friars under papal authority, identified witches by stigmata such as seizures. The book also said that witches could cause epilepsy to develop: “For although greater difficulty may be felt in believing that witches are able to cause leprosy or epilepsy since these diseases generally arise from some long-standing physical predisposition or defect, none the less it has sometimes been found that even these have been caused by witchcraft” (4). The Malleus brought a wave of persecution and torture, and led to the death of between 100,000 and 1,000,000 women.

During the eighteenth century’s Enlightenment, the role of the moon as a cause of epilepsy and insanity (lunacy: from luna, the Roman goddess of the moon) gained wide acceptance, as did the role of masturbation. The use of amulets, rings, charms, holy rituals, incantations, human skull bones, and magical therapies waned during the eighteenth century.

The view of epilepsy as a contagious disorder was long lived. Around 50 AD, Pliny noted that medical authorities after Hippocrates often recommended spitting to “throw back” the contagion (5). Romans sent away family members with epilepsy, to avoid “contamination” (6). Even slaves shunned other slaves with epilepsy; they spat at them and avoided sharing their food or drink. In the early nineteenth century, the first asylums were created to house patients with psychiatric disorders and epilepsy. The fear that epilepsy was contagious was “confirmed,” likely through the production of nonepileptic psychogenic seizures in the psychiatric population, leading to a separation of these two groups. For example, the French alienist Etienne Esquirol (7) feared the spread of epilepsy to patients with mental disorders (7). Asylums provided the first opportunity to systematically study epilepsy in diverse populations.

The association between religiousness and epilepsy, first noted by Hippocrates in 400 BC, was also stressed by the nineteenth century physicians such as Esquirol, Benedict Morel (1860), and Henry Maudsley (1879) (7,8,9).

At the turn of the twentieth century, most of the lay and professional communities believed that people with epilepsy had pathological personality traits and psychopathological disorders, such as aggression, sociopathy, and psychosis. However, from the medical perspective, this was partly an artifact of the main setting from which epilepsy was viewed: chronically institutionalized patients many of whom suffered comorbid disorders (e.g., head trauma, neurosyphilis). The concept of an epileptic personality—a ubiquitous and characteristically negative set of behavioral changes in patients with epilepsy—was already established in antiquity, but continued to evolve slowly and relentlessly. Simultaneously, more humanistic and balanced views evolved during the nineteenth century. On the basis of extensive experience with a private outpatient population with idiopathic epilepsy, Reynolds (10) concluded that epilepsy does not necessarily involve any mental change at all; depletion of spirits and timidity are common in males, and excitability of temper occurs in both sexes. Gowers (11) also recognized that many patients with epilepsy had normal personality and intellect, but that many others developed intraparoxysmal behavioral changes. He suggested that these changes resulted from many factors, but mainly from epilepsy. The early twentieth century brought diverse views concerning people with epilepsy and their behaviors. Kinnier Wilson (12) offered a progressive psychosocial view: “On epileptic temperament inordinate stress has been laid. Life is difficult for these patients, and much that is attributed to temperament can with greater reason be assigned to chronic invalidism and unlucky circumstance.” Sjobring (13) adhered to the negative, pervasive view: “A mental change of a specific nature takes place in individuals suffering from epileptic seizures. They become torpid and circumstantial, sticky and adhesive, effectively tense, and suffer from explosive outburst of rage, anxiety,
and so on.” Kraepelin (14) reported aggressiveness in some of his outpatients with epilepsy: “almost always an intensification of mental irritability occurs.” The adhesive or viscous personality traits in which the patient has difficulty in disengaging from interpersonal exchanges were reviewed by many observers in the European literature under various terminologies, such as the enechetic constitution, ixoid character, and glischroid trait.

Early in the twentieth century, the term epileptic personality was used by psychoanalytical theorists to describe a set of character traits associated with epilepsy that focused on impulsivity, egocentricity, and affective viscosity (15). These features were considered to result from hereditary factors, directly from seizures or treatment, or a reaction to painful social situations associated with epilepsy. Others believed that these particular personality features, in individuals with or without seizures, were a direct expression of epilepsy itself. Two coincidental developments in the middle of the century helped reintroduce the highly controversial concept of personality changes in epilepsy—identifying the role of the limbic system in emotion and behavior, and localizing the onset of many partial seizures to the temporal lobe.

Papez (1937) (16) conceived a circuit of interconnected structures comprising the emotion system: hippocampus–fornix–hypothalamus/mammillary bodies–mammillothalamic tract–anterior thalamic nuclei–thalamocingulate fibers–cingulate cortex—amygdala/hippocampus. This emotion circuit theory was based on anatomical connections, sham rage studies, and lesion studies. Yakovlev (1948) (17) conceived a basolateral circuit modulating emotional behavior, including the amygdala, insula, orbitofrontal cortex, and dorsomedial thalamic nucleus. MacLean (1949 and 1990) (18) combined earlier ideas and conceived the limbic system with all the regions mentioned earlier and the septum and nucleus accumbens.

The specific association of temporal lobe epilepsy (TLE) and psychopathology had its major genesis in the 1951 report by Gibbs (19) and up to 33% of patients with “psychomotor seizures” of temporal lobe origin had interictal behavioral changes. Gastaut et al. (20) in 1954 reiterated common observations on the frequency of emotional viscosity, hyposexuality, hypoactivity, and hypoaggresiveness in patients with epilepsy, and first suggested that the stereotypic symptom complex was the antithesis of behaviors in the Kluver-Bucy syndrome (KBS). KBSver-Bucy syndrome is characterized by oral exploratory behavior, increased sexual appetite, decreased aggressivity, and continuous environmental exploration as a consequence of bilateral anterior temporal destructive lesions (21).

Waxman and Geschwind (22) identi-fied a distinct subset of nonpathological behaviors associated with TLE: deepened emotions, circumstantiality, altered religious and sexual concerns, and hypergraphia. They coined the term interictal behavior syndrome, sometimes referred to as Geschwind syndrome or Gastaut-Geschwind syndrome (23). Bear and Fedio (24) expanded this syndrome to include the 18 traits based on a literature review. They found an increased frequency of all 18 traits in patients with TLE compared with non-neurological controls. The interictal behavioral traits described by Bear and Fedio, together with several others reported in the literature, are summarized in Table 17.1. Several traits associated with this syndrome are described in the subsequent text in more detail.