| Treatment of thyroid-associated orbitopathy with rituximab--a novel therapy for an old disease: case report and literature review. | |
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MedLine Citation:
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PMID: 20350915 Owner: NLM Status: MEDLINE |
Abstract/OtherAbstract:
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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. |
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Authors:
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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:
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Type: Case Reports; Journal Article |
Journal Detail:
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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:
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Created Date: 2010-08-13 Completed Date: 2010-12-07 Revised Date: - |
Medline Journal Info:
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Nlm Unique ID: 9607439 Medline TA: Endocr Pract Country: United States |
Other Details:
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Languages: eng Pagination: 677-85 Citation Subset: IM |
Affiliation:
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Department of Internal Medicine, Endocrinology Unit, San Raffaele Scientific Institute and Università Vita-Salute San Raffaele, Milan, Italy. |
Export Citation:
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APA/MLA Format Download EndNote Download BibTex |
| MeSH Terms | |
Descriptor/Qualifier:
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Antibodies, Monoclonal, Murine-Derived
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adverse effects,
therapeutic use* Autoantibodies / blood Diabetes Mellitus, Type 1 / complications, diet therapy, immunology Glucocorticoids / contraindications Graves Ophthalmopathy / complications, drug therapy*, immunology, therapy Humans Immunologic Factors / adverse effects, therapeutic use* Male Middle Aged Paraparesis, Spastic / complications, drug therapy, immunology Stiff-Person Syndrome / complications, drug therapy, immunology Treatment Outcome |
| Chemical | |
Reg. No./Substance:
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0/Antibodies, Monoclonal, Murine-Derived; 0/Autoantibodies; 0/Glucocorticoids; 0/Immunologic Factors; 0/rituximab |
From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine
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