Late occurence of generalized spike-wave in two children with Rolandic epilepsy : Drug effect ??
Abstract number :
2.104
Submission category :
4. Clinical Epilepsy / 4C. Clinical Treatments
Year :
2016
Submission ID :
195740
Source :
www.aesnet.org
Presentation date :
12/4/2016 12:00:00 AM
Published date :
Nov 21, 2016, 18:00 PM
Authors :
Seema Adhami, University of Massachusetts Medical School and UMass Memorial Children's Medical Center, Worcester, Massachusetts and Chellamani Harini, Boston Children's Hospital, Boston, MA United States, Westborough, Massachusetts
Rationale: Occurrence of generalized spike-wave (GSW) in Rolandic epilepsy has been described. Worsening of EEG with antiepileptic drugs (AED) like carbamazepine and oxcarbazepine (OXC) is also known to occur. This occurs in close temporal relationship to starting the AED. We describe two cases of Rolandic epilepsy where EEG showed GSW several years after starting OXC. Methods: Natural history of two cases of Rolandic epilepsy and EEG findings are described. Results: Case#1 15 yo healthy male, with unremarkable perinatal and family history (FH), had three nocturnal seizures (sz) between age 10. 5 and 11 yrs. Each characterized by drooling and slurred speech for 2 ?" 5 min. In two sz, at onset, he felt involuntary body movements/odd sensation (described as "squirming"). EEG showed left mid-temporal spikes in drowsiness. OXC was started. After this he reported three episodes of waking up with a strange feeling and excessive drool on pillow, but no definite sz. OXC dose was adjusted. He had a history of mild speech delay and received help for reading comprehension. Course: No further sz. No behavioral problems. No other medication. 2nd EEG (on OXC;24 hr EEG ) @ 11.5 yrs: B/L independent central sharps and rare L parietal sharps, in drowsiness and sleep. 3rd EEG (on OXC;1 hr EEG ) @ 14.3 yrs: Bursts (< 1 sec) of GSW in sleep. No activation on HV/PS. OXC gradually tapered. 4th EEG (off OXC 1 month;1 hr EEG ) @ 14.7 yrs : In sleep, 2 < 1 sec bursts of GSW. Last EEG (no AED for 5.5 months;24 hr EEG) @ 15.2 yrs: Normal Case#2 15yo dev normal girl, no perinatal problems, presented at 7.5 years with first seizure. Woke up at night with "funny" sensation in R hand and transient inability to move hand. Followed by drooling and mouth twitching. Conscious but unable to speak for 1 ?" 2 mins. Speech slurred for another 5 min. EEG showed right central and temporal spikes in drowsiness, rare left temporal spikes. No AED started. Second seizure 22 months later (age 9.2 yrs ). Semiology same as above. Family did not want to start AED. Ten months later (age 10 yrs) convulsive seizure 1 hr after falling asleep. Lasted > 5 min, stopped with rectal diazepam. OXC was started after this. Two further szs 23 months after starting OXC (age 11.8 yrs). Had not taken OXC for 1 day trough level was borderline. EEG after this, awake and drowsy, normal. No further szs. FH: Maternal uncle had sz at age 2 yrs, was on PHT until college. Had sz recurrence at age 35 yrs. Paternal uncle probable szs as a child. Maternal grandfather possible szs as a child. Course: No szs after age 11.8 yrs. No academic/behavioral concerns. EEG two years later (@ 13.5 yrs; on OXC ): Rare bitemporal, independent and synchronous spike-wave in drowsiness and sleep. Most recent EEG (@ 14.6 yrs; on OXC ): Spike-wave at left frontal, parietal and anterior temporal regions. In sleep, runs of generalized spikes and sharp waves. Brain MRI (3T) Normal. OXC was tapered and stopped in early June 2016. EEG will be repeated in a month. Conclusions: OXC has been associated with seizure and EEG worsening. This is reported in close temporal relationship with introduction of the drug and resolves with drug withdrawal. In our patients, there was no clinical worsening, but the EEG pattern changed from focal to GSW years after starting OXC. Though there was no temporal relation of the occurrence of GSW with the introduction of OXC, it is still possible that OXC may have contributed to GSW as withdrawal has resulted in a normal EEG in 1 patient. Post withdrawal EEG is pending in the other patient. However it is also possible that the EEG changes are age related and not induced by OXC. Re-challenge with OXC would be needed to prove drug effect, but this cannot be ethically justified. Our cases suggest that OXC induced EEG changes may be delayed. This does not indicate change in seizure type or epilepsy syndrome. Funding: N/A
Clinical Epilepsy