Pathological coagulation parameters in as many as 54 patients with autoimmune acquired factor XIII deficiency due to anti-factor XIII autoantibodies.

autoantibody coagulation factor deficiency coagulation factor inhibitor factor XIII haemorrhagic diathesis

Journal

Haemophilia : the official journal of the World Federation of Hemophilia
ISSN: 1365-2516
Titre abrégé: Haemophilia
Pays: England
ID NLM: 9442916

Informations de publication

Date de publication:
May 2021
Historique:
revised: 04 03 2021
received: 26 01 2021
accepted: 06 03 2021
pubmed: 14 4 2021
medline: 21 9 2021
entrez: 13 4 2021
Statut: ppublish

Résumé

Autoimmune factor XIII (FXIII) deficiency (AiF13D) due to anti-FXIII autoantibodies is an extremely rare, life-threatening bleeding disorder that mostly occurs in the elderly. The number of patients diagnosed with AiF13D has been increasing in Japan, probably because of the nationwide survey on AiF13D supported by the Japanese Ministry of Health, Labour and Welfare. To explore the pathologic characteristics of coagulation parameters in AiF13D. AiF13D-suspected cases were consulted, and underwent unified/integrated coagulation screening and were definitively diagnosed as AiF13D separately. AiF13D patients had lower FXIII antigen levels than non-AiF13D patients, but their values overlapped. Among a series of 22-item screening tests and their resulting parameters, the 'FXIII inhibitory potential' yielded by a 1:1 mixing test of the patient's and healthy control's plasma and its 'residual FXIII activity' in 54 AiF13D cases were most distinguishable from 139 non-AiF13D cases, followed by FXIII activity per se and FXIII-specific activity. While the cross-linked α AiF13D appears to induce a hypocoagulopathy combined with a hyper-fibrinolytic state secondary to severe FXIII deficiency caused by anti-FXIII autoantibodies, and the consequent bleeding further modifies its pathological conditions. In addition, the 1:1 mixing test of FXIII activity was confirmed to be a reliable screening method for AiF13D, especially when its derivative parameter, such as the 'FXIII inhibitory potential' or 'FXIII inhibitory potential ratio', is employed.

Identifiants

pubmed: 33847063
doi: 10.1111/hae.14298
doi:

Substances chimiques

Autoantibodies 0
Factor XIII 9013-56-3

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

454-462

Subventions

Organisme : Japanese Ministry of Health, Labor, and Welfare (MHLW)
Organisme : Japanese Ministry of Education, Culture, Sports, Science and Technology (MEXT)

Informations de copyright

© 2021 John Wiley & Sons Ltd.

Références

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Auteurs

Akitada Ichinose (A)

Department of Molecular Patho-Biochemistry and Patho-Biology, Yamagata University School of Medicine, Yamagata, Japan.
The Japanese Collaborative Research Group (JCRG) on Autoimmune Acquired Coagulation Factor Deficiencies supported by the Japanese Ministry of Health, Labor and Welfare (MHLW), Yamagata, Japan.

Tsukasa Osaki (T)

Department of Molecular Patho-Biochemistry and Patho-Biology, Yamagata University School of Medicine, Yamagata, Japan.
The Japanese Collaborative Research Group (JCRG) on Autoimmune Acquired Coagulation Factor Deficiencies supported by the Japanese Ministry of Health, Labor and Welfare (MHLW), Yamagata, Japan.
Department of Public Health and Hygiene, Yamagata University Graduate School of Medical Science, Yamagata, Japan.

Masayoshi Souri (M)

Department of Molecular Patho-Biochemistry and Patho-Biology, Yamagata University School of Medicine, Yamagata, Japan.
The Japanese Collaborative Research Group (JCRG) on Autoimmune Acquired Coagulation Factor Deficiencies supported by the Japanese Ministry of Health, Labor and Welfare (MHLW), Yamagata, Japan.
Department of Public Health and Hygiene, Yamagata University Graduate School of Medical Science, Yamagata, Japan.

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