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Frontotemporal dementia as a comorbidity to idiopathic normal pressure hydrocephalus (iNPH): a short review of literature and an unusual case.

Abstract Behavioural variant frontotemporal dementia (bvFTD) and idiopathic normal pressure hydrocephalus (iNPH) are neurodegenerative diseases that can present with similar symptoms. These include decline in executive functions, psychomotor slowness, and behavioural and personality changes. Ventricular enlargement is a key radiological finding in iNPH that may also be present in bvFTD caused by the C9ORF72 expansion mutation. Due to this, bvFTD has been hypothesized as a potential comorbidity to iNPH but bvFTD patients have never been identified in studies focusing in clinical comorbidities with iNPH. Here we describe a patient with the C9ORF72 expansion-associated bvFTD who also showed enlarged ventricles on brain imaging. The main clinical symptoms were severe gait disturbances and psychiatric problems with mild cognitive decline. Cerebrospinal fluid removal increased the patient's walking speed, so a ventriculoperitoneal shunt was placed. After insertion of the shunt, there was a significant improvement in walking speed as well as mild improvement in cognitive function but not in neuropsychiatric symptoms relating to bvFTD. Comorbid iNPH should be considered in bvFTD patients who have enlarged ventricles and severely impaired gait.
PMID
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Authors

Mayor MeshTerms
Keywords

C9ORF72 repeat expansion

CSF shunt

Dementia

Idiopathic normal pressure hydrocephalus

Neurodegeneration

Journal Title fluids and barriers of the cns
Publication Year Start




PMID- 28420385
OWN - NLM
STAT- MEDLINE
DA  - 20170419
DCOM- 20170424
LR  - 20170424
IS  - 2045-8118 (Electronic)
IS  - 2045-8118 (Linking)
VI  - 14
IP  - 1
DP  - 2017 Apr 19
TI  - Frontotemporal dementia as a comorbidity to idiopathic normal pressure
      hydrocephalus (iNPH): a short review of literature and an unusual case.
PG  - 10
LID - 10.1186/s12987-017-0060-7 [doi]
AB  - Behavioural variant frontotemporal dementia (bvFTD) and idiopathic normal
      pressure hydrocephalus (iNPH) are neurodegenerative diseases that can present
      with similar symptoms. These include decline in executive functions, psychomotor 
      slowness, and behavioural and personality changes. Ventricular enlargement is a
      key radiological finding in iNPH that may also be present in bvFTD caused by the 
      C9ORF72 expansion mutation. Due to this, bvFTD has been hypothesized as a
      potential comorbidity to iNPH but bvFTD patients have never been identified in
      studies focusing in clinical comorbidities with iNPH. Here we describe a patient 
      with the C9ORF72 expansion-associated bvFTD who also showed enlarged ventricles
      on brain imaging. The main clinical symptoms were severe gait disturbances and
      psychiatric problems with mild cognitive decline. Cerebrospinal fluid removal
      increased the patient's walking speed, so a ventriculoperitoneal shunt was
      placed. After insertion of the shunt, there was a significant improvement in
      walking speed as well as mild improvement in cognitive function but not in
      neuropsychiatric symptoms relating to bvFTD. Comorbid iNPH should be considered
      in bvFTD patients who have enlarged ventricles and severely impaired gait.
FAU - Korhonen, V E
AU  - Korhonen VE
AD  - Department of Neurosurgery, Kuopio University Hospital, P.O. Box 100, 70029 KYS, 
      Kuopio, Finland.
AD  - University of Eastern Finland, P.O. Box 100, 70029 KYS, Kuopio, Finland.
FAU - Solje, E
AU  - Solje E
AD  - Institute of Clinical Medicine-Neurology, University of Eastern Finland, P.O. Box
      1627, 70211, Kuopio, Finland.
FAU - Suhonen, N M
AU  - Suhonen NM
AD  - Medical Research Center, Oulu University Hospital, P.O. Box 20, 90029, Oulu,
      Finland.
AD  - Unit of Clinical Neuroscience, Neurology, University of Oulu, P.O. Box 5000,
      90014, Oulu, Finland.
FAU - Rauramaa, T
AU  - Rauramaa T
AD  - Institute of Clinical Medicine-Pathology, School of Medicine, University of
      Eastern, Kuopio, Finland.
AD  - Department of Pathology, Kuopio University Hospital, P.O. Box 162, 70211, Kuopio,
      Finland.
FAU - Vanninen, R
AU  - Vanninen R
AD  - Department of Radiology, Kuopio University Hospital, P.O. Box 100, 70029 KYS,
      Kuopio, Finland.
FAU - Remes, A M
AU  - Remes AM
AD  - Institute of Clinical Medicine-Neurology, University of Eastern Finland, P.O. Box
      1627, 70211, Kuopio, Finland.
AD  - Medical Research Center, Oulu University Hospital, P.O. Box 20, 90029, Oulu,
      Finland.
AD  - Unit of Clinical Neuroscience, Neurology, University of Oulu, P.O. Box 5000,
      90014, Oulu, Finland.
FAU - Leinonen, V
AU  - Leinonen V
AD  - Department of Neurosurgery, Kuopio University Hospital, P.O. Box 100, 70029 KYS, 
      Kuopio, Finland. [email protected]
AD  - University of Eastern Finland, P.O. Box 100, 70029 KYS, Kuopio, Finland.
      [email protected]
LA  - eng
PT  - Case Reports
PT  - Journal Article
PT  - Review
DEP - 20170419
PL  - England
TA  - Fluids Barriers CNS
JT  - Fluids and barriers of the CNS
JID - 101553157
RN  - 0 (C9orf72 protein, human)
RN  - 0 (Proteins)
SB  - IM
MH  - Brain/diagnostic imaging/pathology
MH  - Cerebrospinal Fluid Shunts
MH  - Comorbidity
MH  - Female
MH  - Frontotemporal Dementia/*complications/diagnostic imaging/physiopathology/surgery
MH  - Humans
MH  - Hydrocephalus, Normal Pressure/*complications/diagnostic
      imaging/physiopathology/surgery
MH  - Middle Aged
MH  - Proteins/genetics
MH  - Trinucleotide Repeat Expansion
PMC - PMC5395836
OTO - NOTNLM
OT  - *C9ORF72 repeat expansion
OT  - *CSF shunt
OT  - *Dementia
OT  - *Idiopathic normal pressure hydrocephalus
OT  - *Neurodegeneration
EDAT- 2017/04/20 06:00
MHDA- 2017/04/25 06:00
CRDT- 2017/04/20 06:00
PHST- 2017/02/07 [received]
PHST- 2017/04/05 [accepted]
AID - 10.1186/s12987-017-0060-7 [doi]
AID - 10.1186/s12987-017-0060-7 [pii]
PST - epublish
SO  - Fluids Barriers CNS. 2017 Apr 19;14(1):10. doi: 10.1186/s12987-017-0060-7.

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