{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,1,23]],"date-time":"2026-01-23T00:09:28Z","timestamp":1769126968025,"version":"3.49.0"},"reference-count":61,"publisher":"Oxford University Press (OUP)","issue":"Supplement_1","license":[{"start":{"date-parts":[[2009,10,30]],"date-time":"2009-10-30T00:00:00Z","timestamp":1256860800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/academic.oup.com\/journals\/pages\/open_access\/funder_policies\/chorus\/standard_publication_model"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2009,10,30]]},"abstract":"<jats:title>Summary<\/jats:title><jats:p>Primary immunodeficiencies (PIDs) are uncommon, chronic and severe disorders of the immune system in which patients cannot mount a sufficiently protective immune response, leading to an increased susceptibility to infections. The treatment of choice for PID patients with predominant antibody deficiency is intravenous immunoglobulin (Ig) replacement therapy. Despite major advances over the last 20 years in the molecular characterization of PIDs, many patients remain undiagnosed or are diagnosed too late, with severe consequences. Various strategies to ensure timely diagnosis of PIDs are in place, and novel approaches are being developed. In recent years, several patient registries have been established. Such registries shed light on the pathology and natural history of these varied disorders. Analyses of the registry data may also reveal which patients are likely to respond well to higher Ig infusion rates and may help to determine the optimal dosing of Ig products. Faster infusion rates may lead to improved convenience for patients and thus increase patient compliance, and may reduce nursing time and the need for hospital resources. Data from two recent studies suggest that Gamunex\u00ae and Privigen\u00ae are well tolerated at high infusion rates. Nevertheless, careful selection of patients for high infusion rates, based on co-morbid conditions and tolerance of the current infusion rate, is advisable. Based on the available data, intravenous Ig offers broad protection against encapsulated organisms. As vaccine trends change, careful monitoring of specific antibody levels in the general population, such as those against pneumococcal and meningococcal bacteria, should be implemented.<\/jats:p>","DOI":"10.1111\/j.1365-2249.2009.04023.x","type":"journal-article","created":{"date-parts":[[2009,10,30]],"date-time":"2009-10-30T18:33:24Z","timestamp":1256927604000},"page":"14-22","source":"Crossref","is-referenced-by-count":57,"title":["Immunodeficiencies"],"prefix":"10.1093","volume":"158","author":[{"given":"M","family":"Ballow","sequence":"first","affiliation":[{"name":"Women and Children's Hospital of Buffalo, State University of New York at Buffalo, Buffalo, NY"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"L","family":"Notarangelo","sequence":"additional","affiliation":[{"name":"Division of Immunology and The Manton Center for Orphan Disease Research, Children's Hospital, Harvard Medical School, Boston, MA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"B","family":"Grimbacher","sequence":"additional","affiliation":[{"name":"Royal Free Hospital, University College London, London"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"C","family":"Cunningham-Rundles","sequence":"additional","affiliation":[{"name":"Mount Sinai School of Medicine, New York, NY"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"M","family":"Stein","sequence":"additional","affiliation":[{"name":"Allergy Associates of the Palm Beaches, North Palm Beach, FL"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"M","family":"Helbert","sequence":"additional","affiliation":[{"name":"Central Manchester and Manchester Children's University Hospital, Manchester, UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"B","family":"Gathmann","sequence":"additional","affiliation":[{"name":"University Medical Center, Center of Chronic Immunodeficiency, Freiburg, Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"G","family":"Kindle","sequence":"additional","affiliation":[{"name":"University Medical Center, Center of Chronic Immunodeficiency, Freiburg, Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"A K","family":"Knight","sequence":"additional","affiliation":[{"name":"Mount Sinai School of Medicine, New York, NY"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"H D","family":"Ochs","sequence":"additional","affiliation":[{"name":"University of Washington and Seattle Children's, Seattle, WA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"K","family":"Sullivan","sequence":"additional","affiliation":[{"name":"Children's Hospital of Philadelphia, Philadelphia, PA, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"J L","family":"Franco","sequence":"additional","affiliation":[{"name":"University of Antioquia, Medell\u00edn, Colombia"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"286","published-online":{"date-parts":[[2009,10,30]]},"reference":[{"key":"2021122708525258700_b1","doi-asserted-by":"crossref","first-page":"722","DOI":"10.1542\/peds.9.6.722","article-title":"Agammaglobulinemia","volume":"9","author":"Bruton","year":"1952","journal-title":"Pediatrics"},{"key":"2021122708525258700_b2","doi-asserted-by":"crossref","first-page":"221","DOI":"10.1006\/clim.2002.5241","article-title":"Clinical, immunological, and molecular analysis in a large cohort of patients with x-linked agammaglobulinemia: an Italian multicenter study","volume":"104","author":"Plebani","year":"2002","journal-title":"Clin Immunol"},{"key":"2021122708525258700_b3","author":"European Society for Immunodeficiencies Registry"},{"key":"2021122708525258700_b4","doi-asserted-by":"crossref","first-page":"776","DOI":"10.1016\/j.jaci.2007.08.053","article-title":"Primary immunodeficiency diseases: an update from the International Union of Immunological Societies Primary Immunodeficiency Diseases Classification Committee","volume":"120","author":"Geha","year":"2007","journal-title":"J Allergy Clin Immunol"},{"key":"2021122708525258700_b5","doi-asserted-by":"crossref","first-page":"190","DOI":"10.1006\/clim.1999.4799","article-title":"Diagnostic criteria for primary immunodeficiencies. 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