| Unmasking of acquired autoimmune C1-inhibitor deficiency by an angiotensin-converting enzyme inhibitor. | |
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MedLine Citation:
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PMID: 11345293 Owner: NLM Status: MEDLINE |
Abstract/OtherAbstract:
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BACKGROUND: A majority of angioedema arise from unknown etiologies. Angioedema may also arise from medications or deficiency of C1-esterase inhibitor (C1-INH); either of these may lead to recurrent, sometimes life-threatening attacks of subcutaneous or submucosal edema if the angioedema involves the tongue, throat, or larynx. We describe a patient with unknown acquired C1-INH deficiency, who experienced only mild attacks of angioedema before treatment with an angiotensin-converting enzyme (ACE) inhibitor. This therapy led to life-threatening respiratory distress. OBJECTIVE: To investigate this patient's life-threatening angioedema. METHODS: Serum protein electrophoresis and immunofixation were performed. The titer of anti-C1-inhibitor autoantibody was determined by ELISA, and the specificity of the autoantibody demonstrated by using purified C1-INH to block binding in the ELISA. Finally, fractions from the immunoelectrophoresis gel were tested for C1-INH autoantibody by ELISA. RESULTS: Complement activation was documented by reduced C1-INH, C1q, and C4, and the patient was found to have an autoantibody of IgG2 isotype specific for C1-INH. After discontinuation of the ACE inhibitor, he continued to have decreased C1-INH and positive C1-INH autoantibodies. CONCLUSIONS: This case describes a patient who had a history of mild facial and extremity swelling with abdominal symptoms before ACE inhibitor treatment; this medication resulted in life-threatening respiratory distress. The use of the ACE inhibitor may have unmasked this patient's acquired autoimmune C1-INH deficiency. |
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Authors:
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G I Kleiner; P Giclas; G Stadtmauer; C Cunningham-Rundles |
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Publication Detail:
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Type: Case Reports; Journal Article; Research Support, Non-U.S. Gov't; Research Support, U.S. Gov't, P.H.S. |
Journal Detail:
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Title: Annals of allergy, asthma & immunology : official publication of the American College of Allergy, Asthma, & Immunology Volume: 86 ISSN: 1081-1206 ISO Abbreviation: Ann. Allergy Asthma Immunol. Publication Date: 2001 Apr |
Date Detail:
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Created Date: 2001-05-09 Completed Date: 2001-05-21 Revised Date: 2007-11-15 |
Medline Journal Info:
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Nlm Unique ID: 9503580 Medline TA: Ann Allergy Asthma Immunol Country: United States |
Other Details:
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Languages: eng Pagination: 461-4 Citation Subset: IM |
Affiliation:
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Division of Clinical Immunology, Mount Sinai-New York University Medical Center, New York, USA. klein014@mc.duke.edu |
Export Citation:
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| MeSH Terms | |
Descriptor/Qualifier:
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Aged Angioedema / immunology* Angiotensin-Converting Enzyme Inhibitors / adverse effects* Antibodies, Monoclonal / immunology Autoantibodies / immunology Autoimmune Diseases / immunology* Blood Protein Electrophoresis Complement C1 Inactivator Proteins / deficiency*, immunology* Humans Immunoglobulin G / analysis, immunology Male |
| Grant Support | |
ID/Acronym/Agency:
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AI-46732/AI/NIAID NIH HHS; FDA-001679/FD/FDA HHS |
| Chemical | |
Reg. No./Substance:
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0/Angiotensin-Converting Enzyme Inhibitors; 0/Antibodies, Monoclonal; 0/Autoantibodies; 0/Complement C1 Inactivator Proteins; 0/Immunoglobulin G |
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