Febrile Infection-Related Epilepsy Syndrome (FIRES), a Possible Cause of Super-Refractory Status Epilepticus: A Case Report
DOI:
https://doi.org/10.3889/oamjms.2024.11895Keywords:
Antiepileptic drugs, Fever, Immunoglobulin, Ketogenic diet, New-onset super refractory status epilepticus, Febrile infection-related epilepsy syndrome, Febrile seizuresAbstract
BACKGROUND: Febrile infection-related epilepsy syndrome (FIRES) is a subset of NORSE that requires a febrile infection between 24 h and 2 weeks before the onset of refractory status epilepticus (SE), with or without fever at the onset of SE, and with no restriction to the age of the patient. The literature on FIRES is scarce.
CASE PRESENTATION: This article reports a case of a patient of 8 years old without relevant family, psychosocial, prenatal or perinatal, or pathological history and normal neurodevelopment. The child consulted on a pediatric emergency with convulsive SE preceded 8 days before by otitis with acute fever managed with cephalosporin. Subsequently, seizures appeared in the following 48 h, characterized by generalized tonic-clonic seizures lasting 5–6 min. The seizures became more frequent until consciousness deteriorated, and motor SE appeared. In consequence, a coma was initiated, and the patient was hospitalized for emergency pediatric reanimation, where multifocal clonic and myoclonic seizures continued. A lumbar puncture with polymerase chain reaction was done twice and returned negative. Two magnetic resonance imaging scans were performed: the first was normal and the second, after 20 days in the hospital, showed cortical damage with SE. Different anticonvulsant treatments were used for 4 weeks: Valproic acid, clobazam, ketamine, levetiracetam for 24 days, and relay to, brivaracetam used in the treatment of epilepsy to control focal seizures; propofol, midazolam, and some antiepileptic drugs were used simultaneously and at maximum recommended doses, achieving a poor initial response, but eventually leading to total crisis control. The patient receives immunoglobulin for 2 days 1 g/kg/J. A ketogenic diet aimed at therapy and known antiepileptic properties, rich in lipids, and low in sugars, causing ketonemia in non-surgical drug-resistant epilepsies, was used for our patient with an efficacy that proved to be remarkable on the frequency of undergoing seizures, his tolerance was good. The diet can be maintained for 20 days without incident, in particular, with no episode of hypoglycemia. Electroconvulsive therapy (formerly called sismotherapy, electronarcosis, or shock therapy) is a therapeutic tool used in several psychiatric illnesses was also used with different frequencies for our patients twice a week with no favorable response. The evolution was marked by the aggravation of the patient following diffuse cerebral edema resulting in cardiac arrest and non-response to resuscitation measures, the patient declared at 07 AM on February 27, 2023.
CONCLUSION: FIRES is a rare epilepsy syndrome of unclear aetiology in which children, usually of school age, suddenly develop very frequent seizures after a mild febrile illness. Seizures in FIRES are typically difficult to treat, and the prognosis is poor.
Downloads
Metrics
Plum Analytics Artifact Widget Block
References
Mikaeloff Y, Jambaque´ I, Hertz-Pannier L, Zanfirescu A, Adamsbaum C, Ploin P, et al. Devastating epileptic encephalopathy in school-aged children (DESC): A pseudo encephalitis. Epilepsy Res. 2006;69(1):67-79. https://doi.org/10.1016/j.eplepsyres.2006.01.002 PMid:16469483 DOI: https://doi.org/10.1016/j.eplepsyres.2006.01.002
Awaya Y, Fukuyama Y. Epilepsy sequelae of acute encephalitis: Encephalopathy (3rd report). Jpan J Psychiatry Neurol. 1986;40:385-7.
Sahin M, Menache CC, Holmes GL, Riviello JJ. Outcome of severe refractory status epilepticus in children. Epilepsia. 2001;42(11):1461-7. https://doi.org/10.1046/j.1528-1157.2001.21301.x PMid:11879350 DOI: https://doi.org/10.1046/j.1528-1157.2001.21301.x
Nabbout R. Autoimmune and inflammatory epilepsies. Epilepsia. 2012;53(Suppl 4):58-62. https://doi.org/10.1111/j.1528-1167.2012.03614.x PMid:22946722 DOI: https://doi.org/10.1111/j.1528-1167.2012.03614.x
BrittonJ.Autoimmuneepilepsy.HandbClinNeurol.2016;133:219-45. https://doi.org/10.1016/B978-0-444-63432-0.00013-X PMid:27112680 DOI: https://doi.org/10.1016/B978-0-444-63432-0.00013-X
Greco A, Rizzo MI, De Virgilio A, Conte M, Gallo A, Attanasio G, et al. Autoimmune epilepsy. Autoimmun Rev. 2016;15(3):221-5. https://doi.org/10.1016/j.autrev.2015.11.007 PMid:26626229 DOI: https://doi.org/10.1016/j.autrev.2015.11.007
Vezzani A, Rüegg S. The pivotal role of immunity and inflammatory processes in epilepsy is increasingly recognized: Introduction. Epilepsia. 2011;52(Suppl 3):1-4. https://doi.org/10.1111/j.1528-1167.2011.03028.x PMid:21542838 DOI: https://doi.org/10.1111/j.1528-1167.2011.03028.x
Nabbout R, Vezzani A, Dulac O, Chiron C. Acute encephalopathy with inflammation-mediated status epilepticus. Neurology. 2011;10(1):99-108. https://doi.org/10.1016/ S1474-4422(10)70214-3 PMid:21163447 DOI: https://doi.org/10.1016/S1474-4422(10)70214-3
Illingworth MA, Hanrahan D, Anderson CE, O’Kane K, Anderson J, Casey M, et al. Elevated VGKC-complex antibodies in a boy with fever-induced refractory epileptic encephalopathy in school-age children (FIRES). Dev Med Child Neurol. 2011;53(11):1053-7. https://doi.org/10.1111/j.1469-8749.2011.04008.x PMid:21592118 DOI: https://doi.org/10.1111/j.1469-8749.2011.04008.x
Van Baalen A, Häusler M, Plecko-Startining B, Strautmanis J, Vlaho S, Gebhardt B, et al. Febrile infection-related epilepsy syndrome without detectable autoantibodies and response to immunotherapy: A case series and discussion of epileptogenesis in FIRES. Neuropediatrics. 2012;43(4):209-16. https://doi.org/10.1055/s-0032-1323848 PMid:22911482 DOI: https://doi.org/10.1055/s-0032-1323848
Singh RK, Joshi SM, Potter DM, Leber SM, Carlson MD, Shellhaas RA. Cognitive outcomes in febrile infection-related epilepsy syndrome treated with the ketogenic diet. Pediatrics. 2014;134(5):e1431-5. https://doi.org/10.1542/peds.2013-3106 PMid:25332495 DOI: https://doi.org/10.1542/peds.2013-3106
Specchio N, Fusco L, Vigevano F. Acute-onset epilepsy triggered by fever mimicking FIRES (febrile infection-related epilepsy syndrome): The role of protocadherin 19 (PCDH19) gene mutation. Epilepsia. 2011;52(11):e172-5. https://doi.org/10.1111/j.1528-1167.2011.03193.x PMid:21777234 DOI: https://doi.org/10.1111/j.1528-1167.2011.03193.x
Appenzeller S, Helbig I, Stephani U, Häusler M, Kluger G, Bungeroth M, et al. Febrile infection-related epilepsy syndrome (FIRES) is not caused by SCN1A, POLG, PCDH19 mutations or rare copy number variations. Dev Med Child Neurol. 2012;54(12):1144-8. https://doi.org/10.1111/j.1469-8749.2012.04435.x PMid:23066759 DOI: https://doi.org/10.1111/j.1469-8749.2012.04435.x
Carranza-Rojo D, Simon-Harvey A, Iona X, Dibbens LM, Damiano JA, Arsov T, et al. Febrile infection-related epilepsy syndrome is not caused by SCN1A mutations. Epilepsy Res. 2012;100(1-2):194-8. https://doi.org/10.1016/j.eplepsyres201202.007 PMid:22386634 DOI: https://doi.org/10.1016/j.eplepsyres.2012.02.007
Pardo CA, Nabbout R, Galanopoulou AS. Mechanisms of epileptogenesis in pediatric epileptic syndromes: Rasmussen encephalitis, infantile spasms, and febrile infection- related epilepsy syndrome (FIRES). Neurotherapeutics. 2014;11(2):297-310. https://doi.org/10.1007/s13311-014-0265-2 PMid:24639375 DOI: https://doi.org/10.1007/s13311-014-0265-2
Kröll-Seger J, Bien CG, Huppertz HJ. Non-paraneoplastic limbic encephalitis associated with antibodies to potassium channels leading to bilateral hippocampal sclerosis in a pre-pubertal girl. Epileptic Disord. 2009;11(1):54-9. https://doi.org/10.1684/ epd.2009.0237 PMid:19251579 DOI: https://doi.org/10.1684/epd.2009.0237
Kramer U, Chi CS, Lin KL, Specchio N, Sahin M, Olson H, et al. Febrile infection-related epilepsy syndrome (FIRES): Pathogenesis, treatment, and outcome: A multicenter study on 77 children. Epilepsia. 2011;52(11):1956-65. https://doi.org/10.1111/j.1528-1167.2011.03250.x PMid:21883180 DOI: https://doi.org/10.1111/j.1528-1167.2011.03250.x
Howell KB, Katanyuwong K, Mackay MT, Bailey CA, Scheffer IE, Freeman JL, et al. Long-term follow-up of febrile infection-related epilepsy syndrome. Epilepsia. 2012;53(1):101-10. https://doi.org/10.1111/j.1528-1167.2011.03350.x PMid:22191582 DOI: https://doi.org/10.1111/j.1528-1167.2011.03350.x
Varadkar S, Cross H. Rasmussen syndrome and other inflammatory epilepsies. Semin Neurol. 2015;35(3):259-68. https://doi.org/10.1055/s-0035-1552921 PMid:26060905 DOI: https://doi.org/10.1055/s-0035-1552921
Van Baalen A, Häusler M, Boor R, Rohr A, Sperner J, Kurlemann G, et al. Febrile infection-related epilepsy syndrome (FIRES): A nonencephalitic encephalopathy in childhood. Epilepsia. 2010;51(7):1323-8. https://doi. org/10.1111/j.1528-1167.2010.02535.x PMid:20345937 DOI: https://doi.org/10.1111/j.1528-1167.2010.02535.x
Sakuma H, Awaya Y, Shiomi M, Yamanouchi H, Takahashi Y, Saito Y, et al. Acute encephalitis with refractory, repetitive partial seizures (AERRPS): A peculiar form of childhood encephalitis. Acta Neurol Scand. 2010;121(4):251-6. https://doi.org/10.1111/j.1600-0404.2009.01198.x PMid:20028339 DOI: https://doi.org/10.1111/j.1600-0404.2009.01198.x
Sharma VK, Hegde AU, Nabi F. Refractory status epilepticus: Febrile illness related epileptic syndrome (FIRES). Int J Epilepsy. 2015;2(2):90-3. DOI: https://doi.org/10.1016/j.ijep.2015.07.001
Van Baalen A, Vezzani A, Häusler M, Kluger G. Febrile infection- related epilepsy syndrome: Clinical review and hypotheses of epileptogenesis. Neuropediatrics. 2017;48(1):5-18. http://doi.org/10.1055/s-0036-1597271 PMid:27919115 DOI: https://doi.org/10.1055/s-0036-1597271
Suleiman J, Brilot F, Lang B, Vincent A, Dale RC. Autoimmune epilepsy in children: Case series and proposed guidelines for identification. Epilepsia. 2013;54(6):1036-45. https://doi.org/10.1111/epi.12142 PMid:23551014 DOI: https://doi.org/10.1111/epi.12142
Rivas-Coppola MS, Shah N, Choudhri AF, Morgan R, Wheless JW. Chronological evolution of magnetic resonance imaging findings in children with febrile infection-related epilepsy syndrome. Pediatr Neurol. 2015;55:22-9. https://doi.org/10.1016/j.pediatrneurol.2015.09.003 PMid:26597039 DOI: https://doi.org/10.1016/j.pediatrneurol.2015.09.003
Caraballo RH, Reyes G, Avaria MF, Buompadre MC, González M, Fortini S, et al. Febrile infection-related epilepsy syndrome: A study of 12 patients. Seizure. 2013;22(7):553-9. https://doi.org/10.1016/j.seizure.2013.04.005 PMid:23643626 DOI: https://doi.org/10.1016/j.seizure.2013.04.005
Specchio N, Fusco L, Claps D, Vigevano F. Epileptic encephalopathy in children possibly related to immune- mediated pathogenesis. Brain Dev. 2010;32(1):51-6. https://doi.org/10.1016/j.braindev.2009.09.017 PMid:19850427 DOI: https://doi.org/10.1016/j.braindev.2009.09.017
Cherian PJ, Nickels KC. Febrile infection-related epilepsy syndromes and their treatment. J Pediatr Epilepsy. 2014;3(2014):67-74.
Ueda R, Saito Y, Ohno K, Maruta K, Matsunami K, Saiki Y. Effect of levetiracetam in acute encephalitis with refractory, repetitive partial seizures during acute and chronic phase. Brain Dev. 2015;37(5):471-7. https://doi.org/10.1016/j.braindev.2014.08.003 PMid:25174548 DOI: https://doi.org/10.1016/j.braindev.2014.08.003
Kramer U, Chi CS, Lin KL, Specchio N, Sahin M, Olson H, et al. Febrile infection-related epilepsy syndrome (FIRES): Does duration of anesthesia affect outcome? Epilepsia. 2011;52(Suppl 8):28-30. https://doi.org/10.1111/j.1528-1167.2011.03230.x PMid:21967356 DOI: https://doi.org/10.1111/j.1528-1167.2011.03230.x
Sort R, Born AP, Pedersen KN, Fonsmark L, Uldall P. Ketogenic diet in 3 cases of childhood refractory status epilepticus. Eur J Paediatr Neurol. 2013;17(6):531-6. https://doi.org/10.1016/j.ejpn.2013.05.001 PMid:23751291 DOI: https://doi.org/10.1016/j.ejpn.2013.05.001
Nabbout R, Mazzuca M, Hubert P, Peudennier S, Allaire C, Flurin V, et al. Efficacy of ketogenic diet in severe refractory status epilepticus initiating fever induced refractory epileptic encephalopathy in school age children (FIRES). Epilepsia. 2010;51(10):2033-7. https://doi.org/10.1111/j.1528-1167.2010.02703.x PMid:20813015 DOI: https://doi.org/10.1111/j.1528-1167.2010.02703.x
Ferlisi M, Hocker S, Grade M, Trinka E, Shorvon S. Preliminary results of the global audit of treatment of refractory status epilepticus. Epilepsy Behav. 2015;49:318-24. https://doi.org/10.1016/j.yebeh.2015.04.010 PMid:25952268 DOI: https://doi.org/10.1016/j.yebeh.2015.04.010
Downloads
Additional Files
Published
How to Cite
Issue
Section
Categories
License
Copyright (c) 2024 Houssam Eddine Sahraoui, Kaoutar Danaoui, Abdelaziz Sihami, Youssef Mouaffak, Said Younous (Author)
This work is licensed under a Creative Commons Attribution-NonCommercial 4.0 International License.
http://creativecommons.org/licenses/by-nc/4.0