Thrombotic microangiopathy in a patient with anti-signal recognition particle antibody-positive immune-mediated necrotizing myopathy.

anti-SRP antibody immune-mediated necrotizing myopathy intravenous immunoglobulin renal biopsy renal failure tacrolimus thrombotic microangiopathy

Journal

International journal of rheumatic diseases
ISSN: 1756-185X
Titre abrégé: Int J Rheum Dis
Pays: England
ID NLM: 101474930

Informations de publication

Date de publication:
12 Oct 2023
Historique:
revised: 20 08 2023
received: 10 07 2023
accepted: 04 10 2023
medline: 13 10 2023
pubmed: 13 10 2023
entrez: 13 10 2023
Statut: aheadofprint

Résumé

We describe the case of a 61-year-old woman with anti-signal recognition particle (SRP) antibody-positive immune-mediated necrotizing myopathy (IMNM) who exhibited biopsy-confirmed thrombotic microangiopathy (TMA). The patient developed proximal-dominant muscle weakness and was diagnosed with anti-SRP antibody-positive IMNM based on muscle biopsy results and serological examination. A high-dose corticosteroid prescription was initiated, followed by intravenous methylprednisolone and intravenous immunoglobulin therapy (IVIg). The patient showed IVIg-induced hemolytic anemia with preserved ADAMTS13 activity. Transient oral tacrolimus administration was initiated. Approximately 8 weeks after admission, the serum creatinine levels gradually increased. Renal histological examination revealed TMA, including ischemic changes in the renal tubules, stenosis, and occlusion of the interlobular arteries with fibrinoid necrosis of the afferent arteriolar walls. The arteriolar walls demonstrated an accumulation of C1q and C3c. Myofiber damage in patients with IMNM accounts for the activation of the classical pathway of the complement cascade in the sarcolemma due to antibody deposition. Additionally, a membrane attack complex is observed on capillaries in the muscle tissues of patients with anti-SRP antibody-positive IMNM. Although drug-induced pathomechanisms, such as IVIg and tacrolimus, can trigger the development of TMA, we suggest that the presence of serum anti-SRP antibodies would be implicated in complement-associated kidney vascular damage.

Identifiants

pubmed: 37828793
doi: 10.1111/1756-185X.14942
doi:

Types de publication

Case Reports

Langues

eng

Sous-ensembles de citation

IM

Informations de copyright

© 2023 Asia Pacific League of Associations for Rheumatology and John Wiley & Sons Australia, Ltd.

Références

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Auteurs

Kenji Sakai (K)

Department of Neurology, Joetsu General Hospital, Joetsu, Japan.

Megumi Takahashi (M)

Department of Nephrology, Joetsu General Hospital, Joetsu, Japan.

Yumi Ito (Y)

Division of Clinical Nephrology and Rheumatology, Niigata University Graduate School of Medical and Dental Sciences, Niigata, Japan.

Shota Yamada (S)

Department of Neurology, Joetsu General Hospital, Joetsu, Japan.

Toru Ito (T)

Department of Nephrology, Joetsu General Hospital, Joetsu, Japan.

Yo Higuchi (Y)

Department of Neurology, Joetsu General Hospital, Joetsu, Japan.

Shigemi Kameda (S)

Department of Nephrology, Joetsu General Hospital, Joetsu, Japan.

Classifications MeSH