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10 September 2020: Artilces

Autoimmune Myelofibrosis in Sjögren’s Syndrome: Report of a Case

Rare disease

Seiji Kakiuchi A* , Ikumi Takagi A , Hiroaki Akiyama B , Hiroyuki Matsuba B , Junpei Rikitake B , Kazuyoshi Kajimoto B , Yoshitake Hayashi B , Nobuko Iwata B

DOI: 10.12659/AJCR.924983

Am J Case Rep 2020; 21:e924983

Table 1. Previous reports of autoimmune myelofibrosis accompanied by Sjögren’s syndrome.

Age/SexInitial therapyHematological responseBone marrow responseRecurrenceMaintenance therapy
1. Gruson et al. []7 30/FPSL 1 mg/kg/day for 1 month→ tapered off over next 3 months(+)ND(+) at 5 monthsNormalized within 2 weeks after azathioprine administration
2. Hattori et al.72/FPSL 1 mg/kg/day (50 mg/body)→ tapered to 10 mg in just over 2 months(+) 1 month(+)(–)PSL 4 mg/day at 4 months
3. Marie et al. []8 59/FmPSL 500 mg×3 days→ PSL 1 mg/kg/day (70 mg/body)(+) 1 monthND(–)PSL 15 mg/day at 12 months
4. Rizzi et al. []9 43/MPSL 1 mg/kg/day→ tapered off over next 6 months(+)(–)(+)Restarted lowest effective dose of PSL
5. Rizzi et al. []9 66/FPSL 4 mg/day+CsA 50 mg/day for 4 days every week+hydroxychloroquine 200 mg/day for 12 months(–)NDNDHydroxychloroquine+low dose PSL
CsA – cyclosporine A; F – Female; M – Male; mPSL – methylprednisolone; ND – not described; PSL – prednisolone; WBC – white blood count.

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American Journal of Case Reports eISSN: 1941-5923
American Journal of Case Reports eISSN: 1941-5923