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Acute, Localized Paroxysmal Pain: An Uncommon Ictal Manifestation.

ABSTRACT: Pain is a recognized, but rare manifestation of epileptic seizures. Pain associated with seizures can be severe and disabling. Ictal pain is usually associated with paresthesia, thermal sensations, or somatognostic disturbance. If localized, paroxysmal pain is the sole manifestation, seizure may be often overlooked as a cause for pain, leading to unnecessary investigations and erroneous treatment, resulting in the prolongation of suffering. We hereby report the case of a 6-year-old boy, whose initial manifestation was multiple episodes of localized, intense, disabling paroxysms of pain over multiple body sites. He was diagnosed to have focal unaware seizures, and was started on antiepileptic drugs, leading to complete resolution of symptoms.

SUBMITTER: Sheetal S

PROVIDER: S-EPMC7847120 | biostudies-literature | 2020 Jul-Sep

REPOSITORIES: biostudies-literature

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Acute, Localized Paroxysmal Pain: An Uncommon Ictal Manifestation.

Sheetal Sasikumar S Amith Kumar Sasikumar S

Journal of pediatric neurosciences 20200701 3

Pain is a recognized, but rare manifestation of epileptic seizures. Pain associated with seizures can be severe and disabling. Ictal pain is usually associated with paresthesia, thermal sensations, or somatognostic disturbance. If localized, paroxysmal pain is the sole manifestation, seizure may be often overlooked as a cause for pain, leading to unnecessary investigations and erroneous treatment, resulting in the prolongation of suffering. We hereby report the case of a 6-year-old boy, whose in ...[more]

PMID: 33531955

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Project description:ObjectiveAutoantibodies targeting voltage-gated potassium channel (VGKC) complexes cause a spectrum of neuronal hyperexcitability disorders. We investigated pain as a manifestation of VGKC-complex autoimmunity.MethodsWe reviewed the prevalence and characteristics of pain in VGKC-complex-immunoglobulin G (IgG)-seropositive patients in 25 months of comprehensive service testing for neural autoantibodies, subtyped positive sera for LGI1-IgG and CASPR2-IgG specificities, and reviewed pain prevalence in autoimmune control patients.ResultsVGKC-complex-IgG was identified in 1,992 patients of 54,853 tested (4%). Of 316 evaluated neurologically at Mayo Clinic, 159 (50%) had pain, in isolation (28%) or with accompanying neurologic manifestations (72%), and not attributable to alternative cause. Pain was subacute in onset, chronic in course, neuropathic, nociceptive, regional, or diffuse and sometimes attributed to fibromyalgia (6%) or psychogenic cause (13%). Most patients had normal peripheral nervous system function, measured by neuropathy impairment scores and nerve conduction. Evidence of neuronal hyperexcitability (hyperhidrosis, quantitative heat-pain hyperalgesia, or electromyographic excitability) was 25-fold more common in pain patients. Pain management required multiple medications in 70% (narcotics, 30%); 13 of 16 patients reported pain relief with immunotherapy. Pain was significantly associated with CASPR2-IgG-positivity (16% positive with pain, 7% without pain; p = 0.014) but not with LGI1-IgG. Less than 10% of 167 patients with neural autoantibodies other than VGKC-complex-IgG reported pain.ConclusionsChronic idiopathic pain is a syndromic manifestation of VGKC-complex autoimmunity. Hyperexcitability of nociceptive pathways is implicated. CASPR2-IgG significantly associates with pain, but in most patients the antigenic VGKC-complex molecule remains to be determined. VGKC-complex autoimmunity represents an important new direction for pain research and therapy.

Dataset Information

Acute, Localized Paroxysmal Pain: An Uncommon Ictal Manifestation.

Publications

Acute, Localized Paroxysmal Pain: An Uncommon Ictal Manifestation.

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