Complex Striate-frontal Projection and Specific Frontal Gyrus Dysfunctions Concern with the Delusional Misidentification Syndrome: A Case Report and Literature Review

Overview

Journal Sci Prog

Publisher Sage Publications

Specialty Science

Date 2025 Feb 20

PMID 39973080

Authors

Kosuke Matsuzono

Yoshiyuki Onuki

Kyoko Otsuka

Honoka Hiki

Yuhei Anan

Takafumi Mashiko

Reiji Koide

Naoto Kunii

Kensuke Kawai

Shigeru Fujimoto

Affiliations

Soon will be listed here.

Abstract

Delusional misidentification, a rare syndrome in which a patient displays persistent delusional misidentification of individuals or objects, occurs in several types of dementia. However, the pathology of delusional misidentification is still unclear, and there was no data pertaining to striate-frontal projection. Here, we report a case of delusional misidentification following frontotemporal dementia in which complex striate-frontal and some specific frontal gyrus dysfunction were observed. In our presented case, delusional misidentification progressed following frontal atrophy. Believing that her actual daughter had been replaced by her niece, her symptoms of delusional misidentification and frontal atrophy progressed in the short term, and social arrangement was necessary three months after the onset. There were no abnormal neurological findings including parkinsonism and general cognitive function test scores were preserved. Validated by dopamine transporter single-photon emission computed tomography, right unilateral striatal uptake decreased significantly without parkinsonism or Parkinson's disease. In addition, of specific concern, functional magnetic resonance images showed left opercular inferior frontal gyrus and right superior frontal gyrus dysfunctions. Our case study highlights complex striate-frontal projection and specific frontal gyrus dysfunctions associated with the pathology of delusional misidentification syndrome.

References

Rascovsky K, Hodges J, Knopman D, Mendez M, Kramer J, Neuhaus J . Sensitivity of revised diagnostic criteria for the behavioural variant of frontotemporal dementia. Brain. 2011; 134(Pt 9):2456-77. PMC: 3170532. DOI: 10.1093/brain/awr179. View

Haber S, Kim K, Mailly P, Calzavara R . Reward-related cortical inputs define a large striatal region in primates that interface with associative cortical connections, providing a substrate for incentive-based learning. J Neurosci. 2006; 26(32):8368-76. PMC: 6673798. DOI: 10.1523/JNEUROSCI.0271-06.2006. View

Dubois B, Slachevsky A, Litvan I, Pillon B . The FAB: a Frontal Assessment Battery at bedside. Neurology. 2000; 55(11):1621-6. DOI: 10.1212/wnl.55.11.1621. View

Varvat J, Thomas-Anterion C, Decousus M, Perret-Liaudet A, Laurent B . [Atypical Lewy body disease revealed by burning mouth syndrome and a pseudo-psychiatric syndrome]. Rev Neurol (Paris). 2010; 166(5):547-9. DOI: 10.1016/j.neurol.2009.10.018. View

Gagnier J, Kienle G, Altman D, Moher D, Sox H, Riley D . The CARE guidelines: consensus-based clinical case reporting guideline development. Headache. 2013; 53(10):1541-7. DOI: 10.1111/head.12246. View

De Leon J, Stanilla J, Cloud B, Simpson G . Misidentification syndromes in schizophrenia: case reviews with implications for classification and prevalence. Aust N Z J Psychiatry. 1995; 29(1):63-8. DOI: 10.3109/00048679509075893. View

Cipriani G, Vedovello M, Ulivi M, Lucetti C, Di Fiorino A, Nuti A . Delusional misidentification syndromes and dementia: a border zone between neurology and psychiatry. Am J Alzheimers Dis Other Demen. 2013; 28(7):671-8. PMC: 10852797. DOI: 10.1177/1533317513506103. View

Darby R, Laganiere S, Pascual-Leone A, Prasad S, Fox M . Finding the imposter: brain connectivity of lesions causing delusional misidentifications. Brain. 2017; 140(2):497-507. PMC: 5278302. DOI: 10.1093/brain/aww288. View

Thiel C, Studte S, Hildebrandt H, Huster R, Weerda R . When a loved one feels unfamiliar: a case study on the neural basis of Capgras delusion. Cortex. 2014; 52:75-85. DOI: 10.1016/j.cortex.2013.11.011. View

10.

Nambu A, Tokuno H, Takada M . Functional significance of the cortico-subthalamo-pallidal 'hyperdirect' pathway. Neurosci Res. 2002; 43(2):111-7. DOI: 10.1016/s0168-0102(02)00027-5. View

11.

Karakasi M, Markopoulou M, Alexandri M, Douzenis A, Pavlidis P . In fear of the most loved ones. A comprehensive review on Capgras misidentification phenomenon and case report involving attempted murder under Capgras syndrome in a relapse of a schizophrenia spectrum disorder. J Forensic Leg Med. 2019; 66:8-24. DOI: 10.1016/j.jflm.2019.05.019. View

12.

Palermo G, Giannoni S, Bellini G, Siciliano G, Ceravolo R . Dopamine Transporter Imaging, Current Status of a Potential Biomarker: A Comprehensive Review. Int J Mol Sci. 2021; 22(20). PMC: 8538800. DOI: 10.3390/ijms222011234. View

13.

Van Essen D, Smith S, Barch D, Behrens T, Yacoub E, Ugurbil K . The WU-Minn Human Connectome Project: an overview. Neuroimage. 2013; 80:62-79. PMC: 3724347. DOI: 10.1016/j.neuroimage.2013.05.041. View

14.

Friston K, Buechel C, Fink G, Morris J, Rolls E, Dolan R . Psychophysiological and modulatory interactions in neuroimaging. Neuroimage. 1997; 6(3):218-29. DOI: 10.1006/nimg.1997.0291. View

15.

Benson N, Hulac D, Kranzler J . Independent examination of the Wechsler Adult Intelligence Scale-Fourth Edition (WAIS-IV): what does the WAIS-IV measure?. Psychol Assess. 2010; 22(1):121-30. DOI: 10.1037/a0017767. View

16.

Josephs K . Capgras syndrome and its relationship to neurodegenerative disease. Arch Neurol. 2007; 64(12):1762-6. DOI: 10.1001/archneur.64.12.1762. View

17.

Kim K, Lee D, Jhoo J, Youn J, Suh Y, Jun Y . Diagnostic accuracy of mini-mental status examination and revised hasegawa dementia scale for Alzheimer's disease. Dement Geriatr Cogn Disord. 2005; 19(5-6):324-30. DOI: 10.1159/000084558. View

18.

Hartman-Maeir A, Katz N . Validity of the Behavioral Inattention Test (BIT): relationships with functional tasks. Am J Occup Ther. 1995; 49(6):507-16. DOI: 10.5014/ajot.49.6.507. View

19.

Devinsky O . Delusional misidentifications and duplications: right brain lesions, left brain delusions. Neurology. 2009; 72(1):80-7. DOI: 10.1212/01.wnl.0000338625.47892.74. View

20.

Wada Y, Yamamoto T . Selective impairment of facial recognition due to a haematoma restricted to the right fusiform and lateral occipital region. J Neurol Neurosurg Psychiatry. 2001; 71(2):254-7. PMC: 1737516. DOI: 10.1136/jnnp.71.2.254. View