Limits...
Adult-onset temporal lobe epilepsy, cognitive decline, multi-antiepileptic drug hypersensitivity, and Hashimoto's encephalopathy: Two case studies.

Sadan O, Seyman E, Ash EL, Kipervasser S, Neufeld MY - Epilepsy Behav Case Rep (2013)

Bottom Line: Hashimoto's encephalopathy is defined by the coexistence of encephalopathy and antithyroid antibodies.We report two cases of adult-onset temporal lobe epilepsy with subacute cognitive decline, high titers of antithyroid antibodies, multi-antiepileptic drug hypersensitivity, and good response to immunomodulatory treatment.The relevance of multidrug hypersensitivity in the setting of adult-onset epilepsy and the importance of searching for autoimmune causes for epilepsy are discussed.

View Article: PubMed Central - PubMed

Affiliation: Department of Neurology, Tel-Aviv Sourasky Medical Center, Tel-Aviv, Israel.

ABSTRACT
Hashimoto's encephalopathy is defined by the coexistence of encephalopathy and antithyroid antibodies. We report two cases of adult-onset temporal lobe epilepsy with subacute cognitive decline, high titers of antithyroid antibodies, multi-antiepileptic drug hypersensitivity, and good response to immunomodulatory treatment. The relevance of multidrug hypersensitivity in the setting of adult-onset epilepsy and the importance of searching for autoimmune causes for epilepsy are discussed.

No MeSH data available.


Related in: MedlinePlus

Timeline of Case 1. Clock drawing as a sample of the cognitive assessment before plasma exchange (A), after plasma exchange (notice the relative worsening, attributed to the side effects of the treatment) (B), and two (C) and five (D) months following plasma exchange, with increasingly better performance. The clock was fully accurate only on the last test. (E) This representative right temporal encephalographic seizure was recorded during a night's sleep. LMT — lamotrigine, CBZ — carbamazepine, VPA — valproate, LEV — levetiracetam, GBP — gabapentin, CLB — clobazam, PRD — prednisone, PE — plasma exchange, MoCA — Montreal Cognitive Assessment.
© Copyright Policy - CC BY-NC-ND
Related In: Results  -  Collection

License
getmorefigures.php?uid=PMC4150645&req=5

f0005: Timeline of Case 1. Clock drawing as a sample of the cognitive assessment before plasma exchange (A), after plasma exchange (notice the relative worsening, attributed to the side effects of the treatment) (B), and two (C) and five (D) months following plasma exchange, with increasingly better performance. The clock was fully accurate only on the last test. (E) This representative right temporal encephalographic seizure was recorded during a night's sleep. LMT — lamotrigine, CBZ — carbamazepine, VPA — valproate, LEV — levetiracetam, GBP — gabapentin, CLB — clobazam, PRD — prednisone, PE — plasma exchange, MoCA — Montreal Cognitive Assessment.

Mentions: A few months later, the patient was readmitted to our department because of cognitive changes, insomnia, and agitation. A brain magnetic resonance imaging (MRI) scan demonstrated asymmetry of the hippocampi without abnormal signal or any other pathological finding. Video-EEG monitoring revealed multiple right temporal electroencephalographic seizures during the night. Serology results for HIV, hepatitis B and C, VDRL, TPHA, and HHV6 were all negative. Thyroid function was normal. A routine rheumatologic workup and testing for antiphospholipid antibodies were negative. Blood samples were sent to test neoplastic markers as well as the following paraneoplastic antibodies: anti-Yo (anti-Purkinje cell cytoplasmic antibody, associated with ovarian and breast cancer) and anti-Hu (antineuronal nuclear antibody type I, associated with small-cell lung cancer). The result of a full gynecological examination including ultrasonography and mammography was normal. She also underwent colonoscopy, which revealed a single polyp whose histology was consistent with high-grade dysplastic adenoma, as well as chest-abdominal and pelvic CT, the results of which were normal. The paraneoplastic laboratory results turned out to be normal. Thus, the only positive finding was extremely high titers of antithyroglobulin (5318 U/ml) and of antithyroid peroxidase (TPO, > 3000 U/ml), corresponding to the diagnosis of Hashimoto's encephalitis. Since steroid therapy had been previously administered for the diagnosis of severe drug eruption and found to be ineffective for the clinical presentation of Hashimoto's encephalitis as cognitive changes were added to the clinical picture, she was treated with plasma exchange (5 courses over 10 days). Although no clinical improvement was noted at first, EEG monitoring demonstrated significant improvement after 14 days, with only one EEG-verified electroencephalographic seizure per night, compared to 3–4 electroencephalographic seizures per night before the plasma exchange (Fig. 1E). Cognitive assessment (Montreal Cognitive Assessment [MoCA]) performed before and after plasma exchange revealed a mild cognitive impairment (24/30, mainly attention deficit and memory) that improved by two points after receiving treatment (Figs. 1A–D). She was discharged with the recommendation of continued ambulatory immunomodulatory treatment with plasma exchange. There was no further improvement in the MoCA score at the 1-month follow-up, but the patient reported that she felt that her cognitive function improved. At that time, she complained of side effects related to the AED treatment (mainly fatigue). She was offered intravenous immunoglobulin (IVIg) treatment which she refused. One month later, she returned for another scheduled outpatient visit, after having stopped all antiepileptic therapy, and felt that she had returned to her regular levels of function, an observation supported by her spouse. She refused to undergo another EEG examination and blood tests. Further follow-up demonstrated clinical and electroencephalographic relapses every few months, with elevated antithyroid antibodies at similar levels, which responded to an additional course of plasma exchange with improvement in her clinical state. Our suggestion was therefore to continue treatment with plasma exchange on a regular basis, and because of the good response, there was no need for more aggressive immunomodulatory treatment.


Adult-onset temporal lobe epilepsy, cognitive decline, multi-antiepileptic drug hypersensitivity, and Hashimoto's encephalopathy: Two case studies.

Sadan O, Seyman E, Ash EL, Kipervasser S, Neufeld MY - Epilepsy Behav Case Rep (2013)

Timeline of Case 1. Clock drawing as a sample of the cognitive assessment before plasma exchange (A), after plasma exchange (notice the relative worsening, attributed to the side effects of the treatment) (B), and two (C) and five (D) months following plasma exchange, with increasingly better performance. The clock was fully accurate only on the last test. (E) This representative right temporal encephalographic seizure was recorded during a night's sleep. LMT — lamotrigine, CBZ — carbamazepine, VPA — valproate, LEV — levetiracetam, GBP — gabapentin, CLB — clobazam, PRD — prednisone, PE — plasma exchange, MoCA — Montreal Cognitive Assessment.
© Copyright Policy - CC BY-NC-ND
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC4150645&req=5

f0005: Timeline of Case 1. Clock drawing as a sample of the cognitive assessment before plasma exchange (A), after plasma exchange (notice the relative worsening, attributed to the side effects of the treatment) (B), and two (C) and five (D) months following plasma exchange, with increasingly better performance. The clock was fully accurate only on the last test. (E) This representative right temporal encephalographic seizure was recorded during a night's sleep. LMT — lamotrigine, CBZ — carbamazepine, VPA — valproate, LEV — levetiracetam, GBP — gabapentin, CLB — clobazam, PRD — prednisone, PE — plasma exchange, MoCA — Montreal Cognitive Assessment.
Mentions: A few months later, the patient was readmitted to our department because of cognitive changes, insomnia, and agitation. A brain magnetic resonance imaging (MRI) scan demonstrated asymmetry of the hippocampi without abnormal signal or any other pathological finding. Video-EEG monitoring revealed multiple right temporal electroencephalographic seizures during the night. Serology results for HIV, hepatitis B and C, VDRL, TPHA, and HHV6 were all negative. Thyroid function was normal. A routine rheumatologic workup and testing for antiphospholipid antibodies were negative. Blood samples were sent to test neoplastic markers as well as the following paraneoplastic antibodies: anti-Yo (anti-Purkinje cell cytoplasmic antibody, associated with ovarian and breast cancer) and anti-Hu (antineuronal nuclear antibody type I, associated with small-cell lung cancer). The result of a full gynecological examination including ultrasonography and mammography was normal. She also underwent colonoscopy, which revealed a single polyp whose histology was consistent with high-grade dysplastic adenoma, as well as chest-abdominal and pelvic CT, the results of which were normal. The paraneoplastic laboratory results turned out to be normal. Thus, the only positive finding was extremely high titers of antithyroglobulin (5318 U/ml) and of antithyroid peroxidase (TPO, > 3000 U/ml), corresponding to the diagnosis of Hashimoto's encephalitis. Since steroid therapy had been previously administered for the diagnosis of severe drug eruption and found to be ineffective for the clinical presentation of Hashimoto's encephalitis as cognitive changes were added to the clinical picture, she was treated with plasma exchange (5 courses over 10 days). Although no clinical improvement was noted at first, EEG monitoring demonstrated significant improvement after 14 days, with only one EEG-verified electroencephalographic seizure per night, compared to 3–4 electroencephalographic seizures per night before the plasma exchange (Fig. 1E). Cognitive assessment (Montreal Cognitive Assessment [MoCA]) performed before and after plasma exchange revealed a mild cognitive impairment (24/30, mainly attention deficit and memory) that improved by two points after receiving treatment (Figs. 1A–D). She was discharged with the recommendation of continued ambulatory immunomodulatory treatment with plasma exchange. There was no further improvement in the MoCA score at the 1-month follow-up, but the patient reported that she felt that her cognitive function improved. At that time, she complained of side effects related to the AED treatment (mainly fatigue). She was offered intravenous immunoglobulin (IVIg) treatment which she refused. One month later, she returned for another scheduled outpatient visit, after having stopped all antiepileptic therapy, and felt that she had returned to her regular levels of function, an observation supported by her spouse. She refused to undergo another EEG examination and blood tests. Further follow-up demonstrated clinical and electroencephalographic relapses every few months, with elevated antithyroid antibodies at similar levels, which responded to an additional course of plasma exchange with improvement in her clinical state. Our suggestion was therefore to continue treatment with plasma exchange on a regular basis, and because of the good response, there was no need for more aggressive immunomodulatory treatment.

Bottom Line: Hashimoto's encephalopathy is defined by the coexistence of encephalopathy and antithyroid antibodies.We report two cases of adult-onset temporal lobe epilepsy with subacute cognitive decline, high titers of antithyroid antibodies, multi-antiepileptic drug hypersensitivity, and good response to immunomodulatory treatment.The relevance of multidrug hypersensitivity in the setting of adult-onset epilepsy and the importance of searching for autoimmune causes for epilepsy are discussed.

View Article: PubMed Central - PubMed

Affiliation: Department of Neurology, Tel-Aviv Sourasky Medical Center, Tel-Aviv, Israel.

ABSTRACT
Hashimoto's encephalopathy is defined by the coexistence of encephalopathy and antithyroid antibodies. We report two cases of adult-onset temporal lobe epilepsy with subacute cognitive decline, high titers of antithyroid antibodies, multi-antiepileptic drug hypersensitivity, and good response to immunomodulatory treatment. The relevance of multidrug hypersensitivity in the setting of adult-onset epilepsy and the importance of searching for autoimmune causes for epilepsy are discussed.

No MeSH data available.


Related in: MedlinePlus