PMID- 29552370
OWN - NLM
STAT- PubMed-not-MEDLINE
LR  - 20201001
IS  - 2090-6889 (Print)
IS  - 2090-6897 (Electronic)
IS  - 2090-6897 (Linking)
VI  - 2018
DP  - 2018
TI  - Effect of Adalimumab on Refractory Arthritis in Juvenile Idiopathic Inflammatory 
      Myopathy with Anti-MDA5 Autoantibody.
PG  - 2164312
LID - 10.1155/2018/2164312 [doi]
LID - 2164312
AB  - A 10-year-old girl manifested persistent fever, skin rash, leg pain, fatigue, and 
      joint pain. Based on muscle weakness, elevated muscle-derived enzymes, magnetic 
      resonance imaging, and skin biopsy results, the diagnosis was juvenile idiopathic 
      inflammatory myopathies (JIIM). Chest CT was normal; the anti-melanoma 
      differentiation-associated protein-5 (anti-MDA5) autoantibody was positive. 
      Initial manifestations subsided after prednisolone (PSL) and methotrexate 
      treatment. After the PSL dosage was decreased, the patient presented with 
      metacarpophalangeal (MCP) joint pain and swelling in both index fingers, synovial 
      fluid, and signals on power Doppler ultrasound. The arthritis was refractory to 
      cyclosporine and tacrolimus. Radiography showed progressive MCP joint space 
      narrowing and joint erosion. Adalimumab was initiated 14 months after disease 
      onset. There was a mildly increased matrix metalloproteinase-3 (MMP3) level, an 
      erythrocyte sedimentation ratio (ESR), and a normal CRP level. Adalimumab 
      resulted in decreased MCP joint pain and swelling. PSL was discontinued 10 months 
      after adalimumab initiation; after 9 more months of adalimumab, there were no 
      significant ultrasonography findings. MMP3 and ESR levels normalized during 
      treatment. Radiography after 2 years of adalimumab showed further progressive MCP 
      joint space narrowing restricting dorsiflexion. This report clarified that 
      anti-MDA5-positive JIIM joint manifestations were due to active synovitis and 
      that adalimumab is required for severe cases. Further experience is needed to 
      determine the pathology, severity, and prognosis of this type of arthritis.
FAU - Miyamae, Takako
AU  - Miyamae T
AUID- ORCID: 0000-0002-0568-1496
AD  - Institute of Rheumatology, Tokyo Women's Medical University, 10-22 Kawada-cho, 
      Shinjuku-ku, Tokyo 162-0054, Japan.
AD  - Department of Pediatrics, Tokyo Women's Medical University, 8-1 Kawada-cho, 
      Shinjuku-ku, Tokyo 162-8666, Japan.
FAU - Hara, Takuma
AU  - Hara T
AD  - Department of Pediatrics, Tokyo Women's Medical University, 8-1 Kawada-cho, 
      Shinjuku-ku, Tokyo 162-8666, Japan.
FAU - Hanaya, Aki
AU  - Hanaya A
AD  - Department of Pediatrics, Tokyo Women's Medical University, 8-1 Kawada-cho, 
      Shinjuku-ku, Tokyo 162-8666, Japan.
FAU - Tani, Yumi
AU  - Tani Y
AD  - Institute of Rheumatology, Tokyo Women's Medical University, 10-22 Kawada-cho, 
      Shinjuku-ku, Tokyo 162-0054, Japan.
AD  - Department of Pediatrics, Tokyo Women's Medical University, 8-1 Kawada-cho, 
      Shinjuku-ku, Tokyo 162-8666, Japan.
FAU - Kishi, Takayuki
AU  - Kishi T
AD  - Institute of Rheumatology, Tokyo Women's Medical University, 10-22 Kawada-cho, 
      Shinjuku-ku, Tokyo 162-0054, Japan.
AD  - Department of Pediatrics, Tokyo Women's Medical University, 8-1 Kawada-cho, 
      Shinjuku-ku, Tokyo 162-8666, Japan.
FAU - Yamanaka, Hisashi
AU  - Yamanaka H
AUID- ORCID: 0000-0001-8453-6731
AD  - Institute of Rheumatology, Tokyo Women's Medical University, 10-22 Kawada-cho, 
      Shinjuku-ku, Tokyo 162-0054, Japan.
LA  - eng
PT  - Case Reports
DEP - 20180206
PL  - United States
TA  - Case Rep Rheumatol
JT  - Case reports in rheumatology
JID - 101585353
PMC - PMC5818952
EDAT- 2018/03/20 06:00
MHDA- 2018/03/20 06:01
PMCR- 2018/02/06
CRDT- 2018/03/20 06:00
PHST- 2017/09/01 00:00 [received]
PHST- 2017/11/12 00:00 [revised]
PHST- 2017/11/28 00:00 [accepted]
PHST- 2018/03/20 06:00 [entrez]
PHST- 2018/03/20 06:00 [pubmed]
PHST- 2018/03/20 06:01 [medline]
PHST- 2018/02/06 00:00 [pmc-release]
AID - 10.1155/2018/2164312 [doi]
PST - epublish
SO  - Case Rep Rheumatol. 2018 Feb 6;2018:2164312. doi: 10.1155/2018/2164312. 
      eCollection 2018.