PMID: 20647666Jul 22, 2010Paper

A case of Mikulicz's disease, IgG4-related plasmacytic syndrome, successfully treated by corticosteroid and mizoribine, followed by mizoribine alone

Internal Medicine
Yuki NankeShigeru Kotake

Abstract

We report a case of Mikulicz's disease, IgG4-related plasmacytic syndrome, successfully treated with corticosteroid and mizoribine, followed by mizoribine alone. A 56-year-old woman experienced persistent bilateral swelling in the eyelids and submandibular region accompanied by lymphadenopathy without pain. The patient was diagnosed as having Mikulicz's disease since her bilateral lacrimal glands showed persistent swelling and biopsy specimens of the submandibular glands and lymph node showed diffuse infiltration of lymphocytes and plasma cells. Symptoms and laboratory data were improved with corticosteroids (30 mg). However, after tapering prednisolone to 5 mg, submandibular swelling developed. Adding mizoribine resulted in clinical improvement. Prednisolone was tapered and finally discontinued. Since the levels of IgG4 in peripheral blood were increased even after corticosteroid therapy and IgG4 was positive in a submandibular gland specimen obtained before the start of corticoid therapy. Thus, this case may be associated with IgG4. The present case is the first case in which mizoribine was effective for Mikulicz's disease and IgG4-related plasmacytic syndrome.

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Citations

Dec 1, 2010·International Journal of Hematology·Ikuo ShimizuHikaru Kobayashi
Jul 22, 2010·Internal Medicine·Satoshi Ito
Aug 5, 2014·The Annals of Thoracic Surgery·Takehiro MoritaUichi Ikeda
Jun 2, 2011·Journal of Clinical and Experimental Hematopathology : JCEH·Yasufumi MasakiHisanori Umehara
Nov 20, 2013·Acta Oto-laryngologica·Nobuo OhtaSeiji Kakehata
May 9, 2014·Otolaryngology--head and Neck Surgery : Official Journal of American Academy of Otolaryngology-Head and Neck Surgery·Rachel B CainDevyani Lal
Jul 30, 2014·The British Journal of Dermatology·Y TokuraH Hashizume
Oct 17, 2014·Journal of Clinical and Experimental Hematopathology : JCEH·Yasufumi MasakiToshihiro Fukushima
Jan 14, 2012·The Laryngoscope·Nobuo OhtaKenji Izuhara
Nov 21, 2013·Modern Rheumatology·Chihiro HagiyaTakayuki Sumida

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