Autoimmune cerebellar hypermetabolism: Report of three cases and literature overview

•Autoimmune encephalitis is a classical cause of cerebellar damage in adults.•Cerebellar hypermetabolism on 18F-FDG PET-CT is not specific.•It has been reported in various diseases, including autoimmune encephalitis.•This hypermetabolism could be a diagnostic aid in autoimmune encephalitis.•The verm...

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Published in:Revue neurologique Vol. 178; no. 4; pp. 337 - 346
Main Authors: Brunet de Courssou, J.-B., Castilla-Lievre, M.A., Maillot, J., Brechemier, M.-L., Ohlmann, C., Sallansonnet-Froment, M., Tafani, C., Psimaras, D., Ricard, D., Bompaire, F., Taifas, I.
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Language:English
Published: France Elsevier Masson SAS 01.04.2022
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ISSN:0035-3787
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Abstract •Autoimmune encephalitis is a classical cause of cerebellar damage in adults.•Cerebellar hypermetabolism on 18F-FDG PET-CT is not specific.•It has been reported in various diseases, including autoimmune encephalitis.•This hypermetabolism could be a diagnostic aid in autoimmune encephalitis.•The vermian predominance of the hypermetabolism may be of interest. We report three cases of vermian cerebellar hypermetabolism in patients with autoimmune encephalitis. One of our patients was positive for anti-Ma2 antibodies and one for anti-Zic4 antibodies while the remaining patient did not present any known antibodies. The seronegative patient deteriorated after immune checkpoint inhibitor treatment for a pulmonary adenocarcinoma and improved with immunosuppressive drugs, which is in favour of an underlying autoimmune mechanism. They all presented with subacute neurological symptoms. Brain magnetic resonance imaging was normal except in one patient, where hyperintensities were present on FLAIR sequence around the third ventricle and the cerebral aqueduct. 18F-FDG brain positron emission tomography with computed tomography (18F-FDG PET-CT) demonstrated an unusual vermian cerebellar hypermetabolism in the three cases. While cerebellar hypermetabolism on 18F-FDG PET-CT has been described in various neurological diseases, such vermian – and more broadly cerebellar – hypermetabolism was seldom described in previous studies on autoimmune encephalitis. When differential diagnoses have been ruled out, this pattern may be of interest for the positive diagnosis of autoimmune encephalitis in difficult diagnostic cases.
AbstractList We report three cases of vermian cerebellar hypermetabolism in patients with autoimmune encephalitis. One of our patients was positive for anti-Ma2 antibodies and one for anti-Zic4 antibodies while the remaining patient did not present any known antibodies. The seronegative patient deteriorated after immune checkpoint inhibitor treatment for a pulmonary adenocarcinoma and improved with immunosuppressive drugs, which is in favour of an underlying autoimmune mechanism. They all presented with subacute neurological symptoms. Brain magnetic resonance imaging was normal except in one patient, where hyperintensities were present on FLAIR sequence around the third ventricle and the cerebral aqueduct. 18F-FDG brain positron emission tomography with computed tomography (18F-FDG PET-CT) demonstrated an unusual vermian cerebellar hypermetabolism in the three cases. While cerebellar hypermetabolism on 18F-FDG PET-CT has been described in various neurological diseases, such vermian - and more broadly cerebellar - hypermetabolism was seldom described in previous studies on autoimmune encephalitis. When differential diagnoses have been ruled out, this pattern may be of interest for the positive diagnosis of autoimmune encephalitis in difficult diagnostic cases.We report three cases of vermian cerebellar hypermetabolism in patients with autoimmune encephalitis. One of our patients was positive for anti-Ma2 antibodies and one for anti-Zic4 antibodies while the remaining patient did not present any known antibodies. The seronegative patient deteriorated after immune checkpoint inhibitor treatment for a pulmonary adenocarcinoma and improved with immunosuppressive drugs, which is in favour of an underlying autoimmune mechanism. They all presented with subacute neurological symptoms. Brain magnetic resonance imaging was normal except in one patient, where hyperintensities were present on FLAIR sequence around the third ventricle and the cerebral aqueduct. 18F-FDG brain positron emission tomography with computed tomography (18F-FDG PET-CT) demonstrated an unusual vermian cerebellar hypermetabolism in the three cases. While cerebellar hypermetabolism on 18F-FDG PET-CT has been described in various neurological diseases, such vermian - and more broadly cerebellar - hypermetabolism was seldom described in previous studies on autoimmune encephalitis. When differential diagnoses have been ruled out, this pattern may be of interest for the positive diagnosis of autoimmune encephalitis in difficult diagnostic cases.
We report three cases of vermian cerebellar hypermetabolism in patients with autoimmune encephalitis. One of our patients was positive for anti-Ma2 antibodies and one for anti-Zic4 antibodies while the remaining patient did not present any known antibodies. The seronegative patient deteriorated after immune checkpoint inhibitor treatment for a pulmonary adenocarcinoma and improved with immunosuppressive drugs, which is in favour of an underlying autoimmune mechanism. They all presented with subacute neurological symptoms. Brain magnetic resonance imaging was normal except in one patient, where hyperintensities were present on FLAIR sequence around the third ventricle and the cerebral aqueduct. F-FDG brain positron emission tomography with computed tomography ( F-FDG PET-CT) demonstrated an unusual vermian cerebellar hypermetabolism in the three cases. While cerebellar hypermetabolism on F-FDG PET-CT has been described in various neurological diseases, such vermian - and more broadly cerebellar - hypermetabolism was seldom described in previous studies on autoimmune encephalitis. When differential diagnoses have been ruled out, this pattern may be of interest for the positive diagnosis of autoimmune encephalitis in difficult diagnostic cases.
•Autoimmune encephalitis is a classical cause of cerebellar damage in adults.•Cerebellar hypermetabolism on 18F-FDG PET-CT is not specific.•It has been reported in various diseases, including autoimmune encephalitis.•This hypermetabolism could be a diagnostic aid in autoimmune encephalitis.•The vermian predominance of the hypermetabolism may be of interest. We report three cases of vermian cerebellar hypermetabolism in patients with autoimmune encephalitis. One of our patients was positive for anti-Ma2 antibodies and one for anti-Zic4 antibodies while the remaining patient did not present any known antibodies. The seronegative patient deteriorated after immune checkpoint inhibitor treatment for a pulmonary adenocarcinoma and improved with immunosuppressive drugs, which is in favour of an underlying autoimmune mechanism. They all presented with subacute neurological symptoms. Brain magnetic resonance imaging was normal except in one patient, where hyperintensities were present on FLAIR sequence around the third ventricle and the cerebral aqueduct. 18F-FDG brain positron emission tomography with computed tomography (18F-FDG PET-CT) demonstrated an unusual vermian cerebellar hypermetabolism in the three cases. While cerebellar hypermetabolism on 18F-FDG PET-CT has been described in various neurological diseases, such vermian – and more broadly cerebellar – hypermetabolism was seldom described in previous studies on autoimmune encephalitis. When differential diagnoses have been ruled out, this pattern may be of interest for the positive diagnosis of autoimmune encephalitis in difficult diagnostic cases.
Author Maillot, J.
Psimaras, D.
Ricard, D.
Castilla-Lievre, M.A.
Taifas, I.
Brechemier, M.-L.
Ohlmann, C.
Brunet de Courssou, J.-B.
Tafani, C.
Sallansonnet-Froment, M.
Bompaire, F.
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  orcidid: 0000-0001-6039-963X
  surname: Taifas
  fullname: Taifas, I.
  organization: Neurology department, Percy Military hospital, 92190 Clamart, France
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Keywords Cerebellum
Paraneoplastic
PET-scanner
Immune checkpoint inhibitor
Zic4
Hypermetabolism
Autoimmune encephalitis
Ma2
Language English
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Snippet •Autoimmune encephalitis is a classical cause of cerebellar damage in adults.•Cerebellar hypermetabolism on 18F-FDG PET-CT is not specific.•It has been...
We report three cases of vermian cerebellar hypermetabolism in patients with autoimmune encephalitis. One of our patients was positive for anti-Ma2 antibodies...
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SubjectTerms Autoimmune encephalitis
Cerebellum
Encephalitis - diagnostic imaging
Fluorodeoxyglucose F18
Hashimoto Disease
Humans
Hypermetabolism
Immune checkpoint inhibitor
Life Sciences
Ma2
Magnetic Resonance Imaging
Paraneoplastic
PET-scanner
Positron Emission Tomography Computed Tomography
Positron-Emission Tomography
Zic4
Title Autoimmune cerebellar hypermetabolism: Report of three cases and literature overview
URI https://dx.doi.org/10.1016/j.neurol.2021.07.018
https://www.ncbi.nlm.nih.gov/pubmed/34657731
https://www.proquest.com/docview/2583318801
https://hal.science/hal-04504634
Volume 178
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