PMID- 37828793
OWN - NLM
STAT- MEDLINE
DCOM- 20240131
LR  - 20240131
IS  - 1756-185X (Electronic)
IS  - 1756-1841 (Linking)
VI  - 27
IP  - 1
DP  - 2024 Jan
TI  - Thrombotic microangiopathy in a patient with anti-signal recognition particle 
      antibody-positive immune-mediated necrotizing myopathy.
PG  - e14942
LID - 10.1111/1756-185X.14942 [doi]
AB  - 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.
CI  - (c) 2023 Asia Pacific League of Associations for Rheumatology and John Wiley & Sons 
      Australia, Ltd.
FAU - Sakai, Kenji
AU  - Sakai K
AUID- ORCID: 0000-0002-8865-4097
AD  - Department of Neurology, Joetsu General Hospital, Joetsu, Japan.
FAU - Takahashi, Megumi
AU  - Takahashi M
AD  - Department of Nephrology, Joetsu General Hospital, Joetsu, Japan.
FAU - Ito, Yumi
AU  - Ito Y
AD  - Division of Clinical Nephrology and Rheumatology, Niigata University Graduate 
      School of Medical and Dental Sciences, Niigata, Japan.
FAU - Yamada, Shota
AU  - Yamada S
AD  - Department of Neurology, Joetsu General Hospital, Joetsu, Japan.
FAU - Ito, Toru
AU  - Ito T
AD  - Department of Nephrology, Joetsu General Hospital, Joetsu, Japan.
FAU - Higuchi, Yo
AU  - Higuchi Y
AD  - Department of Neurology, Joetsu General Hospital, Joetsu, Japan.
FAU - Kameda, Shigemi
AU  - Kameda S
AD  - Department of Nephrology, Joetsu General Hospital, Joetsu, Japan.
LA  - eng
PT  - Case Reports
DEP - 20231012
PL  - England
TA  - Int J Rheum Dis
JT  - International journal of rheumatic diseases
JID - 101474930
RN  - 0 (Immunoglobulins, Intravenous)
RN  - 0 (Signal Recognition Particle)
RN  - WM0HAQ4WNM (Tacrolimus)
RN  - 0 (Autoantibodies)
SB  - IM
MH  - Female
MH  - Humans
MH  - Middle Aged
MH  - Immunoglobulins, Intravenous/therapeutic use
MH  - Muscle, Skeletal/pathology
MH  - Signal Recognition Particle
MH  - Tacrolimus
MH  - Autoantibodies
MH  - *Myositis/chemically induced/diagnosis/drug therapy
MH  - *Autoimmune Diseases
MH  - *Thrombotic Microangiopathies/chemically induced/diagnosis/drug therapy
OTO - NOTNLM
OT  - anti-SRP antibody
OT  - immune-mediated necrotizing myopathy
OT  - intravenous immunoglobulin
OT  - renal biopsy
OT  - renal failure
OT  - tacrolimus
OT  - thrombotic microangiopathy
EDAT- 2023/10/13 06:45
MHDA- 2024/01/31 06:42
CRDT- 2023/10/13 03:00
PHST- 2023/08/20 00:00 [revised]
PHST- 2023/07/10 00:00 [received]
PHST- 2023/10/04 00:00 [accepted]
PHST- 2024/01/31 06:42 [medline]
PHST- 2023/10/13 06:45 [pubmed]
PHST- 2023/10/13 03:00 [entrez]
AID - 10.1111/1756-185X.14942 [doi]
PST - ppublish
SO  - Int J Rheum Dis. 2024 Jan;27(1):e14942. doi: 10.1111/1756-185X.14942. Epub 2023 
      Oct 12.