Document Detail

Treatment of thyroid-associated orbitopathy with rituximab--a novel therapy for an old disease: case report and literature review.
MedLine Citation:
PMID:  20350915     Owner:  NLM     Status:  MEDLINE    
OBJECTIVE: To report the use of rituximab to treat thyroid-associated orbitopathy (TAO) in a patient with a concomitant B-cell organ-specific autoimmune disorder-the stiff person syndrome (SPS).
METHODS: We present a case report and a review of the related literature.
RESULTS: A 62-year-old man with SPS, latent autoimmune diabetes of the adult, and Graves-Basedow disease was referred to our medical center because of bilateral TAO. An ophthalmologic examination documented asymmetric bilateral NOSPECS (N = no signs or symptoms; O = only signs, no symptoms; S = soft tissue involvement; P = proptosis; E = extraocular muscle involvement; C = corneal involvement; and S = sight loss) class IV TAO (left eye>right eye) with a clinical activity score of 5 on a scale of 7. Magnetic resonance imaging of the orbits documented bilateral exophthalmos (left eye>right eye) due to retrobulbar fibroadipose infiltration, bilateral increase of extrinsic ocular muscle thickness, and enhancement of the left inferior rectus muscle on T2-weighted sequences. Because of concomitant incapacitating SPS and diet-controlled latent autoimmune diabetes of the adult, we excluded long-term corticosteroid therapy as an option and considered the use of rituximab, a mouse-human chimeric monoclonal antibody targeting the CD20 protein on pre-B and mature B lymphocytes. Rituximab was administered in accordance with the protocol for rheumatoid arthritis. During the subsequent 4 months, clinical signs and symptoms of TAO dramatically resolved (clinical activity score = 0 of 7) with a sustained improvement of the spastic paraparesis. The glutamic acid decarboxylase antibody titer remained high, and glycemic control and first-phase insulin secretion did not change.
CONCLUSION: Treatment of active TAO with rituximab should be considered when standard intravenous pulse glucocorticoid treatment is contraindicated or ineffective and when SPS or other organ-specific autoimmune disorders with involvement of humoral autoimmunity are present, inasmuch as more than 1 disease may benefit from the use of this chimeric monoclonal antibody.
Sara Madaschi; Alessandro Rossini; Ilaria Formenti; Vito Lampasona; Stefania Bianchi Marzoli; Gabriella Cammarata; Letterio S Politi; Vittorio Martinelli; Elena Bazzigaluppi; Marina Scavini; Emanuele Bosi; Roberto Lanzi
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Publication Detail:
Type:  Case Reports; Journal Article    
Journal Detail:
Title:  Endocrine practice : official journal of the American College of Endocrinology and the American Association of Clinical Endocrinologists     Volume:  16     ISSN:  1934-2403     ISO Abbreviation:  Endocr Pract     Publication Date:    2010 Jul-Aug
Date Detail:
Created Date:  2010-08-13     Completed Date:  2010-12-07     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  9607439     Medline TA:  Endocr Pract     Country:  United States    
Other Details:
Languages:  eng     Pagination:  677-85     Citation Subset:  IM    
Department of Internal Medicine, Endocrinology Unit, San Raffaele Scientific Institute and Università Vita-Salute San Raffaele, Milan, Italy.
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MeSH Terms
Antibodies, Monoclonal, Murine-Derived / adverse effects,  therapeutic use*
Autoantibodies / blood
Diabetes Mellitus, Type 1 / complications,  diet therapy,  immunology
Glucocorticoids / contraindications
Graves Ophthalmopathy / complications,  drug therapy*,  immunology,  therapy
Immunologic Factors / adverse effects,  therapeutic use*
Middle Aged
Paraparesis, Spastic / complications,  drug therapy,  immunology
Stiff-Person Syndrome / complications,  drug therapy,  immunology
Treatment Outcome
Reg. No./Substance:
0/Antibodies, Monoclonal, Murine-Derived; 0/Autoantibodies; 0/Glucocorticoids; 0/Immunologic Factors; 0/rituximab

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