Retrograde Amnesia in LGI1 and CASPR2 Limbic Encephalitis: Two Case Reports and a Systematic Literature Review
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2025
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| author | Antonio Malvaso Denise Cerne Sara Bernini Sara Bottiroli Enrico Marchioni Pietro Businaro Stefano Masciocchi Chiara Morandi Silvia Scaranzin Emanuela Maria Mobilia Stefano F. Cappa Luana Benedetti Diego Franciotta Matteo Gastaldi |
| author_facet | Antonio Malvaso Denise Cerne Sara Bernini Sara Bottiroli Enrico Marchioni Pietro Businaro Stefano Masciocchi Chiara Morandi Silvia Scaranzin Emanuela Maria Mobilia Stefano F. Cappa Luana Benedetti Diego Franciotta Matteo Gastaldi Antonio Malvaso Denise Cerne Sara Bernini Sara Bottiroli Enrico Marchioni Pietro Businaro Stefano Masciocchi Chiara Morandi Silvia Scaranzin Emanuela Maria Mobilia Stefano F. Cappa Luana Benedetti Diego Franciotta Matteo Gastaldi |
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| contents | Retrograde Amnesia in LGI1 and CASPR2 Limbic Encephalitis: Two Case Reports and a Systematic Literature Review Antonio Malvaso Denise Cerne Sara Bernini Sara Bottiroli Enrico Marchioni Pietro Businaro Stefano Masciocchi Chiara Morandi Silvia Scaranzin Emanuela Maria Mobilia Stefano F. Cappa Luana Benedetti Diego Franciotta Matteo Gastaldi European Journal of Neurology ABSTRACTBackgroundBoth anterograde and retrograde amnesia can typically co‐occur in limbic autoimmune encephalitis (LAE), including the forms associated with antibodies to CASPR2/LGI1, two protein complexed with the voltage‐gated potassium channel (VGKC). However, isolated retrograde amnesia is very rare, and it has never been described in LAE.MethodsWe report two patients with CASPR2 LAE who showed isolated retrograde amnesia, without other significant cognitive impairments. A systematic literature review was performed in accordance with the PRISMA guidelines on patients with LAE, antibodies to the VGKC complex (including LGI1, CASPR2, or the VGKC), and memory impairment.ResultsWe identified 467 patients from 29 studies. Fourteen/467 had retrograde amnesia (2.9%), which co‐occurred with anterograde amnesia in 12 with VGKC antibodies (7 with LGI1 LAE‐like clinical phenotypes). Our two cases with CASPR2 LAE (2/469, 0.4%) were the only ones with isolated retrograde amnesia, which was actively investigated in only 56/467 patients. Thirteen/14 patients, including the two with isolated retrograde amnesia, had partial or poor cognitive improvement.ConclusionsRetrograde amnesia is rare but likely under‐recognized in VGKC‐complex antibodies LAE and associates with poor recovery. When isolated, it adds to the spectrum of CASPR2 LAE. These findings promote insights into retrograde amnesia pathophysiology, deserving investigation across the whole spectrum of AE. 10.1111/ene.70113 http://creativecommons.org/licenses/by-nc/4.0/ |
| doi_str_mv | 10.1111/ene.70113 |
| format | Artículo Open Access |
| id | wiley_oa_10_1111_ene_70113 |
| institution | Wiley Open Access |
| license_str_mv | http://creativecommons.org/licenses/by-nc/4.0/ |
| publishDate | 2025 |
| publisher | Wiley |
| record_format | wiley_oa |
| spellingShingle | Retrograde Amnesia in LGI1 and CASPR2 Limbic Encephalitis: Two Case Reports and a Systematic Literature Review Antonio Malvaso Denise Cerne Sara Bernini Sara Bottiroli Enrico Marchioni Pietro Businaro Stefano Masciocchi Chiara Morandi Silvia Scaranzin Emanuela Maria Mobilia Stefano F. Cappa Luana Benedetti Diego Franciotta Matteo Gastaldi European Journal of Neurology Retrograde Amnesia in LGI1 and CASPR2 Limbic Encephalitis: Two Case Reports and a Systematic Literature Review Antonio Malvaso Denise Cerne Sara Bernini Sara Bottiroli Enrico Marchioni Pietro Businaro Stefano Masciocchi Chiara Morandi Silvia Scaranzin Emanuela Maria Mobilia Stefano F. Cappa Luana Benedetti Diego Franciotta Matteo Gastaldi European Journal of Neurology ABSTRACTBackgroundBoth anterograde and retrograde amnesia can typically co‐occur in limbic autoimmune encephalitis (LAE), including the forms associated with antibodies to CASPR2/LGI1, two protein complexed with the voltage‐gated potassium channel (VGKC). However, isolated retrograde amnesia is very rare, and it has never been described in LAE.MethodsWe report two patients with CASPR2 LAE who showed isolated retrograde amnesia, without other significant cognitive impairments. A systematic literature review was performed in accordance with the PRISMA guidelines on patients with LAE, antibodies to the VGKC complex (including LGI1, CASPR2, or the VGKC), and memory impairment.ResultsWe identified 467 patients from 29 studies. Fourteen/467 had retrograde amnesia (2.9%), which co‐occurred with anterograde amnesia in 12 with VGKC antibodies (7 with LGI1 LAE‐like clinical phenotypes). Our two cases with CASPR2 LAE (2/469, 0.4%) were the only ones with isolated retrograde amnesia, which was actively investigated in only 56/467 patients. Thirteen/14 patients, including the two with isolated retrograde amnesia, had partial or poor cognitive improvement.ConclusionsRetrograde amnesia is rare but likely under‐recognized in VGKC‐complex antibodies LAE and associates with poor recovery. When isolated, it adds to the spectrum of CASPR2 LAE. These findings promote insights into retrograde amnesia pathophysiology, deserving investigation across the whole spectrum of AE. 10.1111/ene.70113 http://creativecommons.org/licenses/by-nc/4.0/ |
| title | Retrograde Amnesia in LGI1 and CASPR2 Limbic Encephalitis: Two Case Reports and a Systematic Literature Review |
| topic | European Journal of Neurology |
| url | https://onlinelibrary.wiley.com/doi/10.1111/ene.70113 |