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Letter to the Editor
Burned-out hippocampus syndrome: myth or reality? A case report
Síndrome de hipocampo quemado, mito o realidad. Reporte de caso
I.D. Freire Carliera,
Corresponding author
ifreire66@gmail.com

Corresponding author.
, S.A. Andrade Rondónb, F.A. Silva Siegerb, I.A. Freire Figueroac, E.A. Barroso Da Silvac
a Servicio de Neurocirugía, Instituto Neurológico, Hospital Internacional de Colombia (HIC), Piedecuesta, Colombia
b Servicio de Neurología, Instituto Neurológico, Hospital Internacional de Colombia (HIC), Piedecuesta, Colombia
c Universidad de La Sabana, Cundinamarca, Colombia
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an urge to run away&#44; anxiety&#44; disconnection from his setting&#44; and automatisms&#44; with post-ictal confusion&#59; seizure duration was 2&#160;minutes&#44; with a frequency of 2-3 episodes per month&#46; The patient was receiving polytherapy with lacosamide&#44; lamotrigine&#44; phenobarbital&#44; and carbamazepine&#46; Brain MRI revealed right HS &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>A&#41;&#46; Video EEG with surface electrodes &#40;vEEG-s&#41;&#44; lasting 96&#160;hours&#44; revealed bilateral temporal interictal epileptiform activity&#44; predominantly in the right hemisphere&#44; and 8 identical ictal events with initially unilateral rhythmic theta waves in the temporal region&#59; onset was left-sided on 6 occasions and right-sided on 2&#46; Neuropsychological evaluation revealed mild to moderate attentional and language impairment&#44; very low intelligence quotient score&#44; and moderate verbal and visuospatial memory impairment&#46; Ictal and interictal SPECT studies were inconclusive&#46; Physical examination revealed anxiety&#44; bradypsychia&#44; and poor short-term memory&#44; with no other abnormalities&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">We bilaterally implanted foramen ovale electrodes &#40;FOE&#59; 1&#160;&#215;&#160;5&#41; &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>B&#41; and performed complementary vEEG-s &#40;reference montage&#41;&#44; of 48 hours&#8217; duration&#46; The study detected interictal activity similar to that recorded in previous EEG studies&#44; with 3 electrically identical stereotyped seizures&#58; fast&#44; low-amplitude activity in contacts 1 and 2 of the right FOE&#44; of 7 seconds&#8217; duration&#44; with secondary propagation to contacts 1 and 2 of the left FOE&#44; where amplitude clearly increased&#44; and subsequently to electrodes F8&#44; T4&#44; and T6&#44; with progressive rhythmic theta activity &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; These findings confirmed the localisation of the epileptogenic zone in the right hippocampus&#46; In February 2016&#44; we performed a standard anterior temporal lobectomy with right amygdalohippocampectomy&#59; the patient continued receiving carbamazepine in monotherapy&#46; A routine EEG performed in March 2018 showed no epileptiform activity&#44; and antiepileptic treatment was suspended&#46; The patient has remained seizure free &#40;Engel class 1A&#41; to date&#44; and has integrated well into his previous work as a farmer&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">The aetiology of TLE includes such factors as perinatal hypoxia&#44; febrile seizures&#44; brain trauma&#44; and neurological infections&#44;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> and is frequently bilateral<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a>&#59; vEEG-s may show unilateral or synchronised or independent bilateral interictal activity&#59; ictal activity may spread to the contralateral lobe in 30&#37; of cases &#40;only 3&#37;-7&#46;5&#37; in unilateral TLE-HS&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> These bilateral discharges may be interpreted as multifocal epilepsy&#44; with surgical treatment potentially being ruled out as a result&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Intracranial electrodes show ictal onset in the affected hippocampus with rapid propagation to the contralateral hippocampus&#59; this incongruity indicates false lateralisation&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> Mintzer et al&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> present 5 cases in a sample of 109 patients with TLE who underwent implantation of intracranial electrodes &#40;4&#46;6&#37;&#41;&#44; with unilateral hippocampal atrophy in MRI studies and ictal EEG-s recordings showing seizure onset in the contralateral temporal lobe and few or no seizures with ipsilateral onset&#59; this combination of findings is referred to as burned-out hippocampus syndrome&#46; Williamson et al&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> report burned-out hippocampus syndrome in 5 of 67 patients &#40;7&#46;5&#37;&#41;&#46; Our research group has identified one case among a total of 13 patients &#40;7&#46;7&#37;&#41;&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">It has been suggested that the false lateralisation may be explained by severe neuronal loss in the damaged hippocampus&#44; which would render the structure unable to recruit sufficient neocortical neurons&#44; activating the contralateral hippocampus and neocortex via the dorsal hippocampal commissure&#44; registering on EEG-s as ictal discharges contralateral to the atrophied hippocampus&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;2&#44;4&#44;7</span></a> Activity is propagated simultaneously to the structurally damaged neocortex&#44; which is unable to radially propagate activity originating in the hippocampus to the surface&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">One predictor of false lateralisation is the presence of interictal activity predominantly ipsilateral to the hippocampal atrophy&#44;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> which&#44; as it originates in the irritative zone &#40;greater size&#41;&#44; is able to recruit greater numbers of neurons and to spread to the ipsilateral neocortex&#59; in ictal recordings&#44; high-frequency&#44; low-voltage activity &#40;smaller than that originating in the irritative zone&#41; is of insufficient volume to generate an electric field visible on vEEG-s&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">Diagnostic work-up in cases of suspected burned-out hippocampus syndrome must seek to rule out multifocal onset and confirm false lateralisation with depth electrodes&#44; basal temporal subdural grid electrodes&#44; or such semi-invasive techniques as FOEs&#46;<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2&#8211;4</span></a> Ictal&#47;interictal SPECT and PET have been proposed as non-invasive methods that may help to resolve this conflict&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> In our patient&#44; we opted for FOE implantation&#44; a cost-effective technique given its low cost&#44; reduced complexity&#44; good safety&#44; and optimal accuracy&#46;<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">8&#8211;10</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">Up to 80&#37; of patients with burned-out hippocampus syndrome achieve seizure freedom with surgical treatment&#46;<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4&#44;11&#44;12</span></a> The best outcomes are subject to confirmation of a single epileptogenic focus&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Further research is needed to establish whether patients with suspected burned-out hippocampus syndrome should be treated surgically without prior invasive neurophysiological evaluation&#46; The use of FOE may be a safe&#44; effective alternative in presurgical decision-making&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Funding</span><p id="par0045" class="elsevierStylePara elsevierViewall">The authors have received no funding for this study&#46;</p></span></span>"
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        "nota" => "<p class="elsevierStyleNotepara" id="npar0005">This study was performed within the Epilepsy Surgery Programme at the Neurological Institute of Hospital Internacional de Colombia&#44; Piedecuesta&#44; Santander&#44; Colombia&#46;</p>"
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        "nota" => "<p class="elsevierStyleNotepara" id="npar0010">Please cite this article as&#58; Freire Carlier ID&#44; Andrade Rond&#243;n&#44; SA&#44; Silva Sieger FA&#44; Freire Figueroa IA&#44; Barroso Da Silva EA&#46; S&#237;ndrome de hipocampo quemado&#44; mito o realidad&#46; Reporte de caso&#46; Neurolog&#237;a&#46; 2021&#59;36&#58;558&#8211;561&#46;</p>"
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          "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A&#41; Preoperative brain MRI scan &#40;1&#46;5&#160;T&#41;&#46; Left&#58; coronal T2-FLAIR sequence showing hyperintensity in the right hippocampus&#46; Right&#58; coronal T2-weighted sequence showing the reduced volume and altered architecture of the right hippocampus&#46; B&#41; Implantation of foramen ovale electrodes &#40;FOE&#41;&#46; Top left&#58; antero-posterior fluoroscopy image showing the left FOE&#46; Top right&#58; lateral radiography showing the implanted electrodes&#46; Middle left&#58; infratemporal extracranial position&#46; Middle right&#58; passage through the foramen ovale&#46; Bottom&#58; intracranial localisation in the mesial part of the temporal lobe &#40;ambiens cistern&#41;&#46;</p>"
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          "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Video EEG study&#58; bilateral foramen ovale electrodes &#40;FOE&#41; and complementary surface electrodes&#58; international 10-20 system&#44; left-to-right reference montage&#46; A&#41; Interictal&#58; fast activity with bilateral &#40;predominantly right-sided&#41; spikes of medium voltage&#46; B&#41; Ictal&#58; onset of fast&#44; low-amplitude activity in FOE contacts 1 and 2&#44; lasting 7 seconds&#44; followed by propagation to the contralateral hippocampus&#44; generating greater amplitude&#44; and across the right temporal lobe&#46;</p>"
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                      "titulo" => "A randomized&#44; controlled trial of surgery for temporal-lobe epilepsy"
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                            0 => "S&#46; Wiebe"
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                        "fecha" => "2001"
                        "volumen" => "345"
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                            "url" => "https://www.ncbi.nlm.nih.gov/pubmed/11484687"
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        "identificador" => "xack555139"
        "titulo" => "Acknowledgements"
        "texto" => "<p id="par0050" class="elsevierStylePara elsevierViewall">We are especially grateful to Dr Daniel Sanjuan Orta&#44; epileptologist and clinical neurophysiologist&#44; and head of the clinical research department at Instituto Nacional de Neurolog&#237;a y Neurocirug&#237;a&#44; Mexico&#44; and to Dr Nhora Patricia Ruiz Alfonzo&#44; neurologist and epileptologist at Neurol&#243;gicas Internacional&#44; Piedecuesta&#44; Colombia&#44; for their kind and valued collaboration in the review and analysis of this article&#46;</p>"
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Article information
ISSN: 21735808
Original language: English
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