Skip to main content
Top
Published in: Journal of Medical Case Reports 1/2016

Open Access 01-12-2016 | Case report

Cholangiocarcinoma associated with limbic encephalitis and early cerebral abnormalities detected by 2-deoxy-2-[fluorine-18]fluoro-d-glucose integrated with computed tomography-positron emission tomography: a case report

Authors: Sergio L. Schmidt, Juliana J. Schmidt, Julio C. Tolentino, Carlos G. Ferreira, Sergio A. de Almeida, Regina P. Alvarenga, Eunice N. Simoes, Guilherme J. Schmidt, Nathalie H. S. Canedo, Leila Chimelli

Published in: Journal of Medical Case Reports | Issue 1/2016

Login to get access

Abstract

Background

Limbic encephalitis was originally described as a rare clinical neuropathological entity involving seizures and neuropsychological disturbances. In this report, we describe cerebral patterns visualized by positron emission tomography in a patient with limbic encephalitis and cholangiocarcinoma. To our knowledge, there is no other description in the literature of cerebral positron emission tomography findings in the setting of limbic encephalitis and subsequent diagnosis of cholangiocarcinoma.

Case presentation

We describe a case of a 77-year-old Caucasian man who exhibited persistent cognitive changes 2 years before his death. A cerebral scan obtained at that time by 2-deoxy-2-[fluorine-18]fluoro-d-glucose integrated with computed tomography-positron emission tomography showed low radiotracer uptake in the frontal and temporal lobes. Cerebrospinal fluid analysis indicated the presence of voltage-gated potassium channel antibodies. Three months before the patient’s death, a lymph node biopsy indicated a cholangiocarcinoma, and a new cerebral scan obtained by 2-deoxy-2-[fluorine-18]fluoro-d-glucose integrated with computed tomography-positron emission tomography showed an increment in the severity of metabolic deficit in the frontal and parietal lobes, as well as hypometabolism involving the temporal lobes. Two months before the patient’s death, cerebral metastases were detected on a contrast-enhanced computed tomographic scan. Postmortem examination revealed a cholangiocarcinoma with multiple metastases including the lungs and lymph nodes. The patient’s brain weighed 1300 g, and mild cortical atrophy, ex vacuo dilation of the ventricles, and mild focal thickening of the cerebellar leptomeninges, which were infiltrated by neoplastic epithelial cells, were observed.

Conclusions

These findings support the need for continued vigilance in malignancy surveillance in patients with limbic encephalitis and early cerebral positron emission tomographic scan abnormalities. The difficulty in early diagnosis of small tumors, such as a cholangiocarcinoma, is discussed in the context of the clinical utility of early cerebral hypometabolism detected by 2-deoxy-2-[fluorine-18]fluoro-d-glucose integrated with computed tomography-positron emission tomography in patients with rapidly progressive dementia.
Literature
1.
go back to reference Ohshita T, Hideshi H, Maruyama H, Kohriyama T, Arimura K, Matsumoto M, et al. Voltage-gated potassium channel antibodies associated limbic encephalitis in a patient with invasive thymoma. J Neurol Sci. 2006;250:167–9. Ohshita T, Hideshi H, Maruyama H, Kohriyama T, Arimura K, Matsumoto M, et al. Voltage-gated potassium channel antibodies associated limbic encephalitis in a patient with invasive thymoma. J Neurol Sci. 2006;250:167–9.
3.
go back to reference Alcantara M, Bennani O, Verdure P, Leprêtre S, Tilly H, Jardin F. Voltage-gated potassium channel antibody paraneoplastic limbic encephalitis associated with acute myeloid leukemia. Case Rep Oncol. 2013;6:289–92.CrossRefPubMedPubMedCentral Alcantara M, Bennani O, Verdure P, Leprêtre S, Tilly H, Jardin F. Voltage-gated potassium channel antibody paraneoplastic limbic encephalitis associated with acute myeloid leukemia. Case Rep Oncol. 2013;6:289–92.CrossRefPubMedPubMedCentral
6.
go back to reference Rocamora R, Becerra JL, Fossas P, Gomez M, Vivanco-Hidalgo RM, Mauri JA, et al. Pilomotor seizures: an autonomic semiology of limbic encephalitis? Seizure. 2014;23:670–3.CrossRefPubMed Rocamora R, Becerra JL, Fossas P, Gomez M, Vivanco-Hidalgo RM, Mauri JA, et al. Pilomotor seizures: an autonomic semiology of limbic encephalitis? Seizure. 2014;23:670–3.CrossRefPubMed
7.
go back to reference Graus F, Titulaer MJ, Balu R, Benseler S, Bien CG, Cellucci T, et al. A clinical approach to diagnosis of autoimmune encephalitis. Lancet Neurol. 2016;15:391–404.CrossRefPubMed Graus F, Titulaer MJ, Balu R, Benseler S, Bien CG, Cellucci T, et al. A clinical approach to diagnosis of autoimmune encephalitis. Lancet Neurol. 2016;15:391–404.CrossRefPubMed
8.
go back to reference Lai M, Huijbers MG, Lancaster E, Graus F, Bataller L, Balice-Gordon R, et al. Investigation of LGI1 as the antigen in limbic encephalitis previously attributed to potassium channels: a case series. Lancet Neurol. 2010;9:776–85.CrossRefPubMedPubMedCentral Lai M, Huijbers MG, Lancaster E, Graus F, Bataller L, Balice-Gordon R, et al. Investigation of LGI1 as the antigen in limbic encephalitis previously attributed to potassium channels: a case series. Lancet Neurol. 2010;9:776–85.CrossRefPubMedPubMedCentral
9.
go back to reference van Sonderen A, Schreurs MW, de Bruijn MA, Boukhrissi S, Nagtzaam MM, Hulsenboom ES, et al. The relevance of VGKC positivity in the absence of LGI1 and Caspr2 antibodies. Neurology. 2016;18:1692–9.CrossRef van Sonderen A, Schreurs MW, de Bruijn MA, Boukhrissi S, Nagtzaam MM, Hulsenboom ES, et al. The relevance of VGKC positivity in the absence of LGI1 and Caspr2 antibodies. Neurology. 2016;18:1692–9.CrossRef
10.
11.
go back to reference Scheid R, Lincke T, Voltz R, Von Cramon DY, Sabri O. Serial 18F-fluoro-2-deoxy-d-glucose positron emission tomography and magnetic resonance imaging of paraneoplastic limbic encephalitis. Arch Neurol. 2004;61:1785–9.CrossRefPubMed Scheid R, Lincke T, Voltz R, Von Cramon DY, Sabri O. Serial 18F-fluoro-2-deoxy-d-glucose positron emission tomography and magnetic resonance imaging of paraneoplastic limbic encephalitis. Arch Neurol. 2004;61:1785–9.CrossRefPubMed
12.
go back to reference Chatzikonstantinou A, Szabo K, Ottomeyer C, Kern R, Hennerici M. Successive affection of bilateral temporomesial structures in a case of non-paraneoplastic limbic encephalitis demonstrated by serial MRI and FDG-PET. J Neurol. 2009;256:1753–5.CrossRefPubMed Chatzikonstantinou A, Szabo K, Ottomeyer C, Kern R, Hennerici M. Successive affection of bilateral temporomesial structures in a case of non-paraneoplastic limbic encephalitis demonstrated by serial MRI and FDG-PET. J Neurol. 2009;256:1753–5.CrossRefPubMed
13.
go back to reference Baumgartner A, Rauer S, Mader I, Meyer PT. Cerebral FDG-PET and MRI findings in autoimmune limbic encephalitis: correlation with autoantibody types. J Neurol. 2013;260:2744–53.CrossRefPubMed Baumgartner A, Rauer S, Mader I, Meyer PT. Cerebral FDG-PET and MRI findings in autoimmune limbic encephalitis: correlation with autoantibody types. J Neurol. 2013;260:2744–53.CrossRefPubMed
14.
go back to reference Hoffmann LA, Jarius S, Pellkofer HL, Schueller M, Krumbholz M, Koenig F, et al. Anti-Ma and anti-Ta associated paraneoplastic neurological syndromes: 22 newly diagnosed patients and review of previous cases. J Neurol Neurosurg Psychiatry. 2008;79:767–73.CrossRefPubMed Hoffmann LA, Jarius S, Pellkofer HL, Schueller M, Krumbholz M, Koenig F, et al. Anti-Ma and anti-Ta associated paraneoplastic neurological syndromes: 22 newly diagnosed patients and review of previous cases. J Neurol Neurosurg Psychiatry. 2008;79:767–73.CrossRefPubMed
15.
go back to reference Blanc F, Ruppert E, Kleitz C, Valenti MP, Cretin B, Humbel RL, et al. Acute limbic encephalitis and glutamic acid decarboxylase antibodies: a reality? J Neurol Sci. 2009;287:69–71.CrossRefPubMed Blanc F, Ruppert E, Kleitz C, Valenti MP, Cretin B, Humbel RL, et al. Acute limbic encephalitis and glutamic acid decarboxylase antibodies: a reality? J Neurol Sci. 2009;287:69–71.CrossRefPubMed
16.
go back to reference Hughes EG, Peng X, Gleichman AJ, Lai M, Zhou L, Tsou R, et al. Cellular and synaptic mechanisms of anti-NMDA receptor encephalitis. J Neurosci. 2010;30:5866–75.CrossRefPubMedPubMedCentral Hughes EG, Peng X, Gleichman AJ, Lai M, Zhou L, Tsou R, et al. Cellular and synaptic mechanisms of anti-NMDA receptor encephalitis. J Neurosci. 2010;30:5866–75.CrossRefPubMedPubMedCentral
Metadata
Title
Cholangiocarcinoma associated with limbic encephalitis and early cerebral abnormalities detected by 2-deoxy-2-[fluorine-18]fluoro-d-glucose integrated with computed tomography-positron emission tomography: a case report
Authors
Sergio L. Schmidt
Juliana J. Schmidt
Julio C. Tolentino
Carlos G. Ferreira
Sergio A. de Almeida
Regina P. Alvarenga
Eunice N. Simoes
Guilherme J. Schmidt
Nathalie H. S. Canedo
Leila Chimelli
Publication date
01-12-2016
Publisher
BioMed Central
Published in
Journal of Medical Case Reports / Issue 1/2016
Electronic ISSN: 1752-1947
DOI
https://doi.org/10.1186/s13256-016-0989-1

Other articles of this Issue 1/2016

Journal of Medical Case Reports 1/2016 Go to the issue