Identifying diagnostic and prognostic factors in cerebral amyloid angiopathy-related inflammation : A systematic analysis of published and seven new cases
© 2023 The Authors. Neuropathology and Applied Neurobiology published by John Wiley & Sons Ltd on behalf of British Neuropathological Society..
AIMS: Cerebral amyloid angiopathy (CAA)-related inflammation (CAA-RI) is a potentially reversible manifestation of CAA, histopathologically characterised by transmural and/or perivascular inflammatory infiltrates. We aimed to identify clinical, radiological and laboratory variables capable of improving or supporting the diagnosis of or predicting/influencing the prognosis of CAA-RI and to retrospectively evaluate different therapeutic approaches.
METHODS: We present clinical and neuroradiological observations in seven unpublished CAA-RI cases, including neuropathological findings in two definite cases. These cases were included in a systematic analysis of probable/definite CAA-RI cases published in the literature up to 31 December 2021. Descriptive and associative analyses were performed, including a set of clinical, radiological and laboratory variables to predict short-term, 6-month and 1-year outcomes and mortality, first on definite and second on an expanded probable/definite CAA-RI cohort.
RESULTS: Data on 205 definite and 100 probable cases were analysed. CAA-RI had a younger symptomatic onset than non-inflammatory CAA, without sex preference. Transmural histology was more likely to be associated with the co-localisation of microbleeds with confluent white matter hyperintensities on magnetic resonance imaging (MRI). Incorporating leptomeningeal enhancement and/or sulcal non-nulling on fluid-attenuated inversion recovery (FLAIR) enhanced the sensitivity of the criteria. Cerebrospinal fluid pleocytosis was associated with a decreased probability of clinical improvement and longer term positive outcomes. Future lobar haemorrhage was associated with adverse outcomes, including mortality. Immunosuppression was associated with short-term improvement, with less clear effects on long-term outcomes. The superiority of high-dose over low-dose corticosteroids was not established.
CONCLUSIONS: This is the largest retrospective associative analysis of published CAA-RI cases and the first to include an expanded probable/definite cohort to identify diagnostic/prognostic markers. We propose points for further crystallisation of the criteria and directions for future prospective studies.
| Medienart: |
E-Artikel |
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| Erscheinungsjahr: |
2024 |
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| Erschienen: |
2024 |
| Enthalten in: |
Zur Gesamtaufnahme - volume:50 |
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| Enthalten in: |
Neuropathology and applied neurobiology - 50(2024), 1 vom: 13. Feb., Seite e12946 |
| Sprache: |
Englisch |
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| Beteiligte Personen: |
Szalardy, Levente [VerfasserIn] |
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| Links: |
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| Anmerkungen: |
Date Completed 19.02.2024 Date Revised 12.03.2024 published: Print Citation Status MEDLINE |
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| doi: |
10.1111/nan.12946 |
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| funding: |
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| Förderinstitution / Projekttitel: |
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NLM365841676 |
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| 100 | 1 | |a Szalardy, Levente |e verfasserin |4 aut | |
| 245 | 1 | 0 | |a Identifying diagnostic and prognostic factors in cerebral amyloid angiopathy-related inflammation |b A systematic analysis of published and seven new cases |
| 264 | 1 | |c 2024 | |
| 336 | |a Text |b txt |2 rdacontent | ||
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| 500 | |a Date Completed 19.02.2024 | ||
| 500 | |a Date Revised 12.03.2024 | ||
| 500 | |a published: Print | ||
| 500 | |a Citation Status MEDLINE | ||
| 520 | |a © 2023 The Authors. Neuropathology and Applied Neurobiology published by John Wiley & Sons Ltd on behalf of British Neuropathological Society. | ||
| 520 | |a AIMS: Cerebral amyloid angiopathy (CAA)-related inflammation (CAA-RI) is a potentially reversible manifestation of CAA, histopathologically characterised by transmural and/or perivascular inflammatory infiltrates. We aimed to identify clinical, radiological and laboratory variables capable of improving or supporting the diagnosis of or predicting/influencing the prognosis of CAA-RI and to retrospectively evaluate different therapeutic approaches | ||
| 520 | |a METHODS: We present clinical and neuroradiological observations in seven unpublished CAA-RI cases, including neuropathological findings in two definite cases. These cases were included in a systematic analysis of probable/definite CAA-RI cases published in the literature up to 31 December 2021. Descriptive and associative analyses were performed, including a set of clinical, radiological and laboratory variables to predict short-term, 6-month and 1-year outcomes and mortality, first on definite and second on an expanded probable/definite CAA-RI cohort | ||
| 520 | |a RESULTS: Data on 205 definite and 100 probable cases were analysed. CAA-RI had a younger symptomatic onset than non-inflammatory CAA, without sex preference. Transmural histology was more likely to be associated with the co-localisation of microbleeds with confluent white matter hyperintensities on magnetic resonance imaging (MRI). Incorporating leptomeningeal enhancement and/or sulcal non-nulling on fluid-attenuated inversion recovery (FLAIR) enhanced the sensitivity of the criteria. Cerebrospinal fluid pleocytosis was associated with a decreased probability of clinical improvement and longer term positive outcomes. Future lobar haemorrhage was associated with adverse outcomes, including mortality. Immunosuppression was associated with short-term improvement, with less clear effects on long-term outcomes. The superiority of high-dose over low-dose corticosteroids was not established | ||
| 520 | |a CONCLUSIONS: This is the largest retrospective associative analysis of published CAA-RI cases and the first to include an expanded probable/definite cohort to identify diagnostic/prognostic markers. We propose points for further crystallisation of the criteria and directions for future prospective studies | ||
| 650 | 4 | |a Case Reports | |
| 650 | 4 | |a Journal Article | |
| 650 | 4 | |a Review | |
| 650 | 4 | |a amyloid-β-related angiitis | |
| 650 | 4 | |a cerebral amyloid angiopathy | |
| 650 | 4 | |a cerebral amyloid angiopathy-related inflammation | |
| 650 | 4 | |a criteria | |
| 650 | 4 | |a diagnosis | |
| 650 | 4 | |a inflammatory cerebral amyloid angiopathy | |
| 650 | 4 | |a outcome | |
| 650 | 4 | |a predictor | |
| 700 | 1 | |a Fakan, Bernadett |e verfasserin |4 aut | |
| 700 | 1 | |a Maszlag-Torok, Rita |e verfasserin |4 aut | |
| 700 | 1 | |a Ferencz, Emil |e verfasserin |4 aut | |
| 700 | 1 | |a Reisz, Zita |e verfasserin |4 aut | |
| 700 | 1 | |a Radics, Bence L |e verfasserin |4 aut | |
| 700 | 1 | |a Csizmadia, Sandor |e verfasserin |4 aut | |
| 700 | 1 | |a Szpisjak, Laszlo |e verfasserin |4 aut | |
| 700 | 1 | |a Annus, Adam |e verfasserin |4 aut | |
| 700 | 1 | |a Zadori, Denes |e verfasserin |4 aut | |
| 700 | 1 | |a Kovacs, Gabor G |e verfasserin |4 aut | |
| 700 | 1 | |a Klivenyi, Peter |e verfasserin |4 aut | |
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