Tripeptidyl Peptidase 1 (TPP1) Deficiency in a 36-Year-Old Patient with Cerebellar-Extrapyramidal Syndrome and Dilated Cardiomyopathy.

autosomal recessive spinocerebellar ataxia type 7 (SCAR7) cerebellar ataxia ceroid lipofuscinosis 2 (CLN2) dilated cardiomyopathy tripeptidyl peptidase 1 (TPP1)

Journal

Life (Basel, Switzerland)
ISSN: 2075-1729
Titre abrégé: Life (Basel)
Pays: Switzerland
ID NLM: 101580444

Informations de publication

Date de publication:
21 Dec 2021
Historique:
received: 18 09 2021
revised: 17 12 2021
accepted: 18 12 2021
entrez: 21 1 2022
pubmed: 22 1 2022
medline: 22 1 2022
Statut: epublish

Résumé

We report on a 36-year-old man with cerebellar-extrapyramidal syndrome and severe heart failure because of dilated cardiomyopathy of unknown origin. Dysarthria and cardiac arrhythmia began at early childhood (4 years of age). Brain MRI (28 years of age) demonstrated severe cerebellar atrophy. At the age 32, he presented with dysarthria, ataxia, dystonia, and tremor of the right hand, bilateral slowed neural conduction in the visual pathways, and decreased mental acuity. At the age of 33 years, the patient underwent cardiac transplantation because of severe dilated cardiomyopathy. In the

Identifiants

pubmed: 35054396
pii: life12010003
doi: 10.3390/life12010003
pmc: PMC8779458
pii:
doi:

Types de publication

Case Reports

Langues

eng

Subventions

Organisme : National Science Center
ID : 2011/01/B/NZ4/03455

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Auteurs

Agnieszka Ługowska (A)

Department of Genetics, Institute of Psychiatry and Neurology, Al. Sobieskiego 9, 02-957 Warsaw, Poland.

Joanna K Purzycka-Olewiecka (JK)

Department of Genetics, Institute of Psychiatry and Neurology, Al. Sobieskiego 9, 02-957 Warsaw, Poland.

Rafał Płoski (R)

Department of Medical Genetics, Medical University of Warsaw, ul. A. Pawińskiego 3c, 02-106 Warsaw, Poland.

Grażyna Truszkowska (G)

Molecular Biology Laboratory, Department of Medical Biology, National Institute of Cardiology, ul. Alpejska 42, 04-628 Warsaw, Poland.

Maciej Pronicki (M)

Department of Pathology, The Children's Memorial Health Institute, al. Dzieci Polskich 20, 04-730 Warsaw, Poland.

Paulina Felczak (P)

Department of Neuropathology, Institute of Psychiatry and Neurology, Al. Sobieskiego 9, 02-957 Warsaw, Poland.

Mateusz Śpiewak (M)

Magnetic Resonance Unit, Department of Radiology, National Institute of Cardiology, ul. Alpejska 42, 04-628 Warsaw, Poland.

Aleksandra Podlecka-Piętowska (A)

Department of Neurology, Medical University of Warsaw, ul. Banacha 1a, 02-097 Warsaw, Poland.

Martyna Sitek (M)

Department of Neurology, Medical University of Warsaw, ul. Banacha 1a, 02-097 Warsaw, Poland.

Zofia T Bilińska (ZT)

Unit for Screening Studies in Inherited Cardiovascular Diseases, National Institute of Cardiology, ul. Alpejska 42, 04-628 Warsaw, Poland.

Przemysław Leszek (P)

Department of Heart Failure and Transplantology, National Institute of Cardiology, ul. Alpejska 42, 04-628 Warsaw, Poland.

Małgorzata Bednarska-Makaruk (M)

Department of Genetics, Institute of Psychiatry and Neurology, Al. Sobieskiego 9, 02-957 Warsaw, Poland.

Classifications MeSH