In Vivo Measurement of Tau Depositions in Anti-IgLON5 Disease Using [18F]PI-2620 PET

Hendrik Theis; Gérard N. Bischof; Norbert Brüggemann; Justina Dargvainiene; Alexander Drzezga; Thomas Grüter; Jan Lewerenz; Frank Leypoldt; Bernd Neumaier; Klaus Peter Wandinger; Ilya Ayzenberg; Thilo Van Eimeren

doi:10.1212/WNL.0000000000207870

In Vivo Measurement of Tau Depositions in Anti-IgLON5 Disease Using [18F]PI-2620 PET

Hendrik Theis, Gérard N. Bischof, Norbert Brüggemann, Justina Dargvainiene, Alexander Drzezga, Thomas Grüter, Jan Lewerenz, Frank Leypoldt, Bernd Neumaier, Klaus Peter Wandinger, Ilya Ayzenberg, Thilo Van Eimeren^*

^*Korrespondierende/r Autor/-in für diese Arbeit

16 Zitate (Scopus)

Abstract

Objectives: Anti-IgLON5 disease is a recently discovered neurologic disorder combining autoimmunity and neurodegeneration. Core manifestations include sleep disorders, bulbar symptoms, gait abnormalities and cognitive dysfunction, but other presentations have been reported. Hallmarks are autoantibodies targeting the neuronal surface protein IgLON5, a strong human leukocyte antigen system Class II association, and brainstem and hypothalamus-dominant tau deposits. The purpose of this cohort study was to visualize tau deposition in vivo with the second-generation tau-PET tracer.MethodsA cohort of 4 patients with anti-IgLON5 disease underwent a dynamic PET scan with [18F]PI-2620. One patient received a follow-up scan. Z-deviation maps and a 2-sample t test in comparison with healthy controls (n = 10) were performed. Antibody titers, neurofilament light chain, and disease duration were correlated with brainstem binding potentials.

Results: Patients demonstrated increased [18F]PI2620 tau binding potentials in the pons, dorsal medulla, and cerebellum. The longitudinal scan after 28 months showed an increase of tracer uptake in the medulla despite immunotherapy. Higher antibody titers and neurofilament light chain correlated with higher tracer retention.

Discussion: The results indicate that tau depositions in anti-IgLON5 disease can be visualized with [18F]PI-2620 and might correlate with the extent of disease. For validation, a larger longitudinal study is necessary.

Originalsprache	Englisch
Zeitschrift	Neurology
Jahrgang	101
Ausgabenummer	22
Seiten (von - bis)	E2325-E2330
ISSN	0028-3878
DOIs	https://doi.org/10.1212/WNL.0000000000207870
Publikationsstatus	Veröffentlicht - 28.11.2023

Fördermittel

This study was funded by the Deutsche Forschungsgemeinschaft (DFG, German Research Foundation) (Project No. 413543196). GENERATE is supported by the German Ministry of Education and Research (BMBF, 01GM1908 and 01GM2208). H. Theis was supported by the program for rotation positions/Faculty of Medicine/University of Cologne and by the Cologne Clinician Scientist Program (CCSP)/Faculty of Medicine/University of Cologne (Project No. 413543196). The production of the tracer and the work of G.N. Bischof and A. Drzezga were funded by the Deutsche Forschungsgemeinschaft - Project-ID 431549029 - SFB 1451. The Article Processing Charge was funded by the authors.

UN SDGs

Dieser Output leistet einen Beitrag zu folgendem(n) Ziel(en) für nachhaltige Entwicklung

SDG 3 – Gesundheit und Wohlergehen
SDG 10 – Weniger Ungleichheiten

Strategische Forschungsbereiche und Zentren

Forschungsschwerpunkt: Gehirn, Hormone, Verhalten - Center for Brain, Behavior and Metabolism (CBBM)

DFG-Fachsystematik

2.23-07 Klinische Neurologie, Neurochirurgie und Neuroradiologie
2.23-05 Experimentelle Modelle zum Verständnis von Erkrankungen des Nervensystems

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10.1212/WNL.0000000000207870

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title = "In Vivo Measurement of Tau Depositions in Anti-IgLON5 Disease Using [18F]PI-2620 PET",

abstract = "Objectives: Anti-IgLON5 disease is a recently discovered neurologic disorder combining autoimmunity and neurodegeneration. Core manifestations include sleep disorders, bulbar symptoms, gait abnormalities and cognitive dysfunction, but other presentations have been reported. Hallmarks are autoantibodies targeting the neuronal surface protein IgLON5, a strong human leukocyte antigen system Class II association, and brainstem and hypothalamus-dominant tau deposits. The purpose of this cohort study was to visualize tau deposition in vivo with the second-generation tau-PET tracer.MethodsA cohort of 4 patients with anti-IgLON5 disease underwent a dynamic PET scan with [18F]PI-2620. One patient received a follow-up scan. Z-deviation maps and a 2-sample t test in comparison with healthy controls (n = 10) were performed. Antibody titers, neurofilament light chain, and disease duration were correlated with brainstem binding potentials.Results: Patients demonstrated increased [18F]PI2620 tau binding potentials in the pons, dorsal medulla, and cerebellum. The longitudinal scan after 28 months showed an increase of tracer uptake in the medulla despite immunotherapy. Higher antibody titers and neurofilament light chain correlated with higher tracer retention.Discussion: The results indicate that tau depositions in anti-IgLON5 disease can be visualized with [18F]PI-2620 and might correlate with the extent of disease. For validation, a larger longitudinal study is necessary.",

author = "Hendrik Theis and Bischof, \{G{\'e}rard N.\} and Norbert Br{\"u}ggemann and Justina Dargvainiene and Alexander Drzezga and Thomas Gr{\"u}ter and Jan Lewerenz and Frank Leypoldt and Bernd Neumaier and Wandinger, \{Klaus Peter\} and Ilya Ayzenberg and \{Van Eimeren\}, Thilo",

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year = "2023",

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doi = "10.1212/WNL.0000000000207870",

language = "English",

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T1 - In Vivo Measurement of Tau Depositions in Anti-IgLON5 Disease Using [18F]PI-2620 PET

AU - Theis, Hendrik

AU - Bischof, Gérard N.

AU - Brüggemann, Norbert

AU - Dargvainiene, Justina

AU - Drzezga, Alexander

AU - Grüter, Thomas

AU - Lewerenz, Jan

AU - Leypoldt, Frank

AU - Neumaier, Bernd

AU - Wandinger, Klaus Peter

AU - Ayzenberg, Ilya

AU - Van Eimeren, Thilo

PY - 2023/11/28

Y1 - 2023/11/28

N2 - Objectives: Anti-IgLON5 disease is a recently discovered neurologic disorder combining autoimmunity and neurodegeneration. Core manifestations include sleep disorders, bulbar symptoms, gait abnormalities and cognitive dysfunction, but other presentations have been reported. Hallmarks are autoantibodies targeting the neuronal surface protein IgLON5, a strong human leukocyte antigen system Class II association, and brainstem and hypothalamus-dominant tau deposits. The purpose of this cohort study was to visualize tau deposition in vivo with the second-generation tau-PET tracer.MethodsA cohort of 4 patients with anti-IgLON5 disease underwent a dynamic PET scan with [18F]PI-2620. One patient received a follow-up scan. Z-deviation maps and a 2-sample t test in comparison with healthy controls (n = 10) were performed. Antibody titers, neurofilament light chain, and disease duration were correlated with brainstem binding potentials.Results: Patients demonstrated increased [18F]PI2620 tau binding potentials in the pons, dorsal medulla, and cerebellum. The longitudinal scan after 28 months showed an increase of tracer uptake in the medulla despite immunotherapy. Higher antibody titers and neurofilament light chain correlated with higher tracer retention.Discussion: The results indicate that tau depositions in anti-IgLON5 disease can be visualized with [18F]PI-2620 and might correlate with the extent of disease. For validation, a larger longitudinal study is necessary.

AB - Objectives: Anti-IgLON5 disease is a recently discovered neurologic disorder combining autoimmunity and neurodegeneration. Core manifestations include sleep disorders, bulbar symptoms, gait abnormalities and cognitive dysfunction, but other presentations have been reported. Hallmarks are autoantibodies targeting the neuronal surface protein IgLON5, a strong human leukocyte antigen system Class II association, and brainstem and hypothalamus-dominant tau deposits. The purpose of this cohort study was to visualize tau deposition in vivo with the second-generation tau-PET tracer.MethodsA cohort of 4 patients with anti-IgLON5 disease underwent a dynamic PET scan with [18F]PI-2620. One patient received a follow-up scan. Z-deviation maps and a 2-sample t test in comparison with healthy controls (n = 10) were performed. Antibody titers, neurofilament light chain, and disease duration were correlated with brainstem binding potentials.Results: Patients demonstrated increased [18F]PI2620 tau binding potentials in the pons, dorsal medulla, and cerebellum. The longitudinal scan after 28 months showed an increase of tracer uptake in the medulla despite immunotherapy. Higher antibody titers and neurofilament light chain correlated with higher tracer retention.Discussion: The results indicate that tau depositions in anti-IgLON5 disease can be visualized with [18F]PI-2620 and might correlate with the extent of disease. For validation, a larger longitudinal study is necessary.

UR - https://www.scopus.com/pages/publications/85178495006

U2 - 10.1212/WNL.0000000000207870

DO - 10.1212/WNL.0000000000207870

M3 - Journal articles

C2 - 37879939

AN - SCOPUS:85178495006

SN - 0028-3878

VL - 101

SP - E2325-E2330

JO - Neurology

JF - Neurology

IS - 22

ER -

In Vivo Measurement of Tau Depositions in Anti-IgLON5 Disease Using [18F]PI-2620 PET

Abstract

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Strategische Forschungsbereiche und Zentren

DFG-Fachsystematik

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