Parathyroid Hormone Resistance and Autoantibodies to the PTH1 Receptor
We describe two cases of acquired parathyroid hormone (PTH) resistance consequent to the development of serum PTH type 1 receptor (PTH1R) autoantibodies, which block PTH binding and signaling. Both cases were associated with other autoimmune manifestations, and one case was associated with atypical...
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| Veröffentlicht in: | The New England journal of medicine 2021-11, Vol.385 (21), p.1974-1980 |
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| container_end_page | 1980 |
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| container_issue | 21 |
| container_start_page | 1974 |
| container_title | The New England journal of medicine |
| container_volume | 385 |
| creator | Mandl, Adel Burbelo, Peter D Di Pasquale, Giovanni Tay, You Sher Welch, James Lionakis, Michail S Rosenzweig, Sergio D Waldman, Meryl A Warner, Blake M Walitt, Brian Collins, Michael T Balow, James E Chiorini, John A Simonds, William F Agarwal, Sunita K Blau, Jenny E Weinstein, Lee S |
| description | We describe two cases of acquired parathyroid hormone (PTH) resistance consequent to the development of serum PTH type 1 receptor (PTH1R) autoantibodies, which block PTH binding and signaling. Both cases were associated with other autoimmune manifestations, and one case was associated with atypical membranous glomerulonephritis. In vitro binding and signaling assays identified the presence of PTH1R-blocking IgG autoantibodies, which were not present in serum samples from patients with other renal or autoimmune disorders. (Funded by the Intramural Research Programs of the National Institute of Diabetes and Digestive and Kidney Diseases and others.)
In this article, the authors discuss two cases of acquired PTH resistance secondary to serum PTH1-receptor autoantibodies that block PTH binding and signaling. In both cases, there were also other manifestations of autoimmunity. The presence of PTH1-receptor autoantibodies should be considered in patients with acquired PTH resistance. |
| doi_str_mv | 10.1056/NEJMoa2109409 |
| format | Article |
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In this article, the authors discuss two cases of acquired PTH resistance secondary to serum PTH1-receptor autoantibodies that block PTH binding and signaling. In both cases, there were also other manifestations of autoimmunity. The presence of PTH1-receptor autoantibodies should be considered in patients with acquired PTH resistance.</description><identifier>ISSN: 0028-4793</identifier><identifier>EISSN: 1533-4406</identifier><identifier>DOI: 10.1056/NEJMoa2109409</identifier><identifier>PMID: 34788508</identifier><language>eng</language><publisher>United States: Massachusetts Medical Society</publisher><subject>Adult ; Age ; Aged ; Allergy ; Antibodies ; Autoantibodies ; Autoantibodies - blood ; Autoimmune Disease ; Autoimmune diseases ; Biopsy ; Creatinine ; Diabetes mellitus ; Disease ; DNA Mutational Analysis ; Endocrinology ; Endocrinology General ; Family medical history ; Female ; Glomerulonephritis ; Glycopeptides - blood ; Humans ; Hypocalcemia ; Hypocalcemia - etiology ; Hypocalcemia - genetics ; Immunoglobulin G ; Immunoglobulin G - blood ; Immunology ; Immunophenotyping ; Kidney diseases ; Kidney Glomerulus - pathology ; Lupus ; Membranous glomerulonephritis ; Microscopy, Electron ; Mouth ; Muscle pain ; Mutation ; Parathyroid ; Parathyroid hormone ; Parathyroid Hormone - metabolism ; Patients ; Pseudohypoparathyroidism - genetics ; Receptor, Parathyroid Hormone, Type 1 - immunology ; Research programs ; Rheumatology ; Rheumatology General ; Thrombocytopenia ; Thyroid gland</subject><ispartof>The New England journal of medicine, 2021-11, Vol.385 (21), p.1974-1980</ispartof><rights>Copyright © 2021 Massachusetts Medical Society. All rights reserved.</rights><rights>Copyright © 2021 Massachusetts Medical Society.</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c445t-37c9127c601d40941d0c85b3a7f9955747b26e58f7fabadf8786420836bb00873</citedby><cites>FETCH-LOGICAL-c445t-37c9127c601d40941d0c85b3a7f9955747b26e58f7fabadf8786420836bb00873</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://www.nejm.org/doi/pdf/10.1056/NEJMoa2109409$$EPDF$$P50$$Gmms$$H</linktopdf><linktohtml>$$Uhttps://www.nejm.org/doi/full/10.1056/NEJMoa2109409$$EHTML$$P50$$Gmms$$H</linktohtml><link.rule.ids>230,314,776,780,881,2746,2747,26080,27901,27902,52357,54039</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/34788508$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Mandl, Adel</creatorcontrib><creatorcontrib>Burbelo, Peter D</creatorcontrib><creatorcontrib>Di Pasquale, Giovanni</creatorcontrib><creatorcontrib>Tay, You Sher</creatorcontrib><creatorcontrib>Welch, James</creatorcontrib><creatorcontrib>Lionakis, Michail S</creatorcontrib><creatorcontrib>Rosenzweig, Sergio D</creatorcontrib><creatorcontrib>Waldman, Meryl A</creatorcontrib><creatorcontrib>Warner, Blake M</creatorcontrib><creatorcontrib>Walitt, Brian</creatorcontrib><creatorcontrib>Collins, Michael T</creatorcontrib><creatorcontrib>Balow, James E</creatorcontrib><creatorcontrib>Chiorini, John A</creatorcontrib><creatorcontrib>Simonds, William F</creatorcontrib><creatorcontrib>Agarwal, Sunita K</creatorcontrib><creatorcontrib>Blau, Jenny E</creatorcontrib><creatorcontrib>Weinstein, Lee S</creatorcontrib><title>Parathyroid Hormone Resistance and Autoantibodies to the PTH1 Receptor</title><title>The New England journal of medicine</title><addtitle>N Engl J Med</addtitle><description>We describe two cases of acquired parathyroid hormone (PTH) resistance consequent to the development of serum PTH type 1 receptor (PTH1R) autoantibodies, which block PTH binding and signaling. Both cases were associated with other autoimmune manifestations, and one case was associated with atypical membranous glomerulonephritis. In vitro binding and signaling assays identified the presence of PTH1R-blocking IgG autoantibodies, which were not present in serum samples from patients with other renal or autoimmune disorders. (Funded by the Intramural Research Programs of the National Institute of Diabetes and Digestive and Kidney Diseases and others.)
In this article, the authors discuss two cases of acquired PTH resistance secondary to serum PTH1-receptor autoantibodies that block PTH binding and signaling. In both cases, there were also other manifestations of autoimmunity. The presence of PTH1-receptor autoantibodies should be considered in patients with acquired PTH resistance.</description><subject>Adult</subject><subject>Age</subject><subject>Aged</subject><subject>Allergy</subject><subject>Antibodies</subject><subject>Autoantibodies</subject><subject>Autoantibodies - blood</subject><subject>Autoimmune Disease</subject><subject>Autoimmune diseases</subject><subject>Biopsy</subject><subject>Creatinine</subject><subject>Diabetes mellitus</subject><subject>Disease</subject><subject>DNA Mutational Analysis</subject><subject>Endocrinology</subject><subject>Endocrinology General</subject><subject>Family medical history</subject><subject>Female</subject><subject>Glomerulonephritis</subject><subject>Glycopeptides - blood</subject><subject>Humans</subject><subject>Hypocalcemia</subject><subject>Hypocalcemia - etiology</subject><subject>Hypocalcemia - genetics</subject><subject>Immunoglobulin G</subject><subject>Immunoglobulin G - blood</subject><subject>Immunology</subject><subject>Immunophenotyping</subject><subject>Kidney diseases</subject><subject>Kidney Glomerulus - pathology</subject><subject>Lupus</subject><subject>Membranous glomerulonephritis</subject><subject>Microscopy, Electron</subject><subject>Mouth</subject><subject>Muscle pain</subject><subject>Mutation</subject><subject>Parathyroid</subject><subject>Parathyroid hormone</subject><subject>Parathyroid Hormone - metabolism</subject><subject>Patients</subject><subject>Pseudohypoparathyroidism - genetics</subject><subject>Receptor, Parathyroid Hormone, Type 1 - immunology</subject><subject>Research programs</subject><subject>Rheumatology</subject><subject>Rheumatology General</subject><subject>Thrombocytopenia</subject><subject>Thyroid gland</subject><issn>0028-4793</issn><issn>1533-4406</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2021</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><sourceid>BEC</sourceid><sourceid>BENPR</sourceid><sourceid>GUQSH</sourceid><sourceid>M2O</sourceid><recordid>eNp10c9LHDEUB_BQKrpVj73KQCl4mfZl8vtSWES7FtuK6DlkMhl3lp1kTTIF_3sjq6IFc3mHfPjyHl-EPmP4hoHx739Of_0OpsGgKKgPaIYZITWlwD-iGUAjayoU2UOfUlpBeZiqXbRHqJCSgZyhs0sTTV7exzB01SLEMXhXXbk0pGy8dZXxXTWfcjA-D23oBpeqHKq8dNXl9QIXad0mh3iAdnqzTu7wae6jm7PT65NFffH35_nJ_KK2lLJcE2EVboTlgLuyLsUdWMlaYkSvFGOCirbhjsle9KY1XS-F5LQBSXjbAkhB9tGPbe5makfXWedzNGu9icNo4r0OZtBvf_yw1Lfhn1YgZUNUCTh-CojhbnIp63FI1q3XxrswJd0wpYArELjQL__RVZiiL-c9Ksk5Ybgpqt4qG0NK0fUvy2DQjw3pNw0Vf_T6ghf9XEkBX7dgHJP2bjW-E_QAGB2WVA</recordid><startdate>20211118</startdate><enddate>20211118</enddate><creator>Mandl, Adel</creator><creator>Burbelo, Peter D</creator><creator>Di Pasquale, Giovanni</creator><creator>Tay, You Sher</creator><creator>Welch, James</creator><creator>Lionakis, Michail S</creator><creator>Rosenzweig, Sergio D</creator><creator>Waldman, Meryl A</creator><creator>Warner, Blake M</creator><creator>Walitt, Brian</creator><creator>Collins, Michael T</creator><creator>Balow, James E</creator><creator>Chiorini, John A</creator><creator>Simonds, William F</creator><creator>Agarwal, Sunita K</creator><creator>Blau, Jenny E</creator><creator>Weinstein, Lee S</creator><general>Massachusetts Medical Society</general><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>0TZ</scope><scope>7RV</scope><scope>7X7</scope><scope>7XB</scope><scope>8AO</scope><scope>8C1</scope><scope>8FE</scope><scope>8FH</scope><scope>8FI</scope><scope>ABUWG</scope><scope>AFKRA</scope><scope>AN0</scope><scope>AZQEC</scope><scope>BBNVY</scope><scope>BEC</scope><scope>BENPR</scope><scope>BHPHI</scope><scope>CCPQU</scope><scope>DWQXO</scope><scope>FYUFA</scope><scope>GHDGH</scope><scope>GNUQQ</scope><scope>GUQSH</scope><scope>HCIFZ</scope><scope>K0Y</scope><scope>LK8</scope><scope>M0R</scope><scope>M0T</scope><scope>M1P</scope><scope>M2M</scope><scope>M2O</scope><scope>M2P</scope><scope>M7P</scope><scope>MBDVC</scope><scope>NAPCQ</scope><scope>PQEST</scope><scope>PQQKQ</scope><scope>PQUKI</scope><scope>PRINS</scope><scope>PSYQQ</scope><scope>Q9U</scope><scope>7X8</scope><scope>5PM</scope></search><sort><creationdate>20211118</creationdate><title>Parathyroid Hormone Resistance and Autoantibodies to the PTH1 Receptor</title><author>Mandl, Adel ; Burbelo, Peter D ; Di Pasquale, Giovanni ; Tay, You Sher ; Welch, James ; Lionakis, Michail S ; Rosenzweig, Sergio D ; Waldman, Meryl A ; Warner, Blake M ; Walitt, Brian ; Collins, Michael T ; Balow, James E ; Chiorini, John A ; Simonds, William F ; Agarwal, Sunita K ; Blau, Jenny E ; Weinstein, Lee S</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c445t-37c9127c601d40941d0c85b3a7f9955747b26e58f7fabadf8786420836bb00873</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2021</creationdate><topic>Adult</topic><topic>Age</topic><topic>Aged</topic><topic>Allergy</topic><topic>Antibodies</topic><topic>Autoantibodies</topic><topic>Autoantibodies - blood</topic><topic>Autoimmune Disease</topic><topic>Autoimmune diseases</topic><topic>Biopsy</topic><topic>Creatinine</topic><topic>Diabetes mellitus</topic><topic>Disease</topic><topic>DNA Mutational Analysis</topic><topic>Endocrinology</topic><topic>Endocrinology General</topic><topic>Family medical history</topic><topic>Female</topic><topic>Glomerulonephritis</topic><topic>Glycopeptides - blood</topic><topic>Humans</topic><topic>Hypocalcemia</topic><topic>Hypocalcemia - etiology</topic><topic>Hypocalcemia - genetics</topic><topic>Immunoglobulin G</topic><topic>Immunoglobulin G - blood</topic><topic>Immunology</topic><topic>Immunophenotyping</topic><topic>Kidney diseases</topic><topic>Kidney Glomerulus - pathology</topic><topic>Lupus</topic><topic>Membranous glomerulonephritis</topic><topic>Microscopy, Electron</topic><topic>Mouth</topic><topic>Muscle pain</topic><topic>Mutation</topic><topic>Parathyroid</topic><topic>Parathyroid hormone</topic><topic>Parathyroid Hormone - metabolism</topic><topic>Patients</topic><topic>Pseudohypoparathyroidism - 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Academic</collection><collection>PubMed Central (Full Participant titles)</collection><jtitle>The New England journal of medicine</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Mandl, Adel</au><au>Burbelo, Peter D</au><au>Di Pasquale, Giovanni</au><au>Tay, You Sher</au><au>Welch, James</au><au>Lionakis, Michail S</au><au>Rosenzweig, Sergio D</au><au>Waldman, Meryl A</au><au>Warner, Blake M</au><au>Walitt, Brian</au><au>Collins, Michael T</au><au>Balow, James E</au><au>Chiorini, John A</au><au>Simonds, William F</au><au>Agarwal, Sunita K</au><au>Blau, Jenny E</au><au>Weinstein, Lee S</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Parathyroid Hormone Resistance and Autoantibodies to the PTH1 Receptor</atitle><jtitle>The New England journal of medicine</jtitle><addtitle>N Engl J Med</addtitle><date>2021-11-18</date><risdate>2021</risdate><volume>385</volume><issue>21</issue><spage>1974</spage><epage>1980</epage><pages>1974-1980</pages><issn>0028-4793</issn><eissn>1533-4406</eissn><abstract>We describe two cases of acquired parathyroid hormone (PTH) resistance consequent to the development of serum PTH type 1 receptor (PTH1R) autoantibodies, which block PTH binding and signaling. Both cases were associated with other autoimmune manifestations, and one case was associated with atypical membranous glomerulonephritis. In vitro binding and signaling assays identified the presence of PTH1R-blocking IgG autoantibodies, which were not present in serum samples from patients with other renal or autoimmune disorders. (Funded by the Intramural Research Programs of the National Institute of Diabetes and Digestive and Kidney Diseases and others.)
In this article, the authors discuss two cases of acquired PTH resistance secondary to serum PTH1-receptor autoantibodies that block PTH binding and signaling. In both cases, there were also other manifestations of autoimmunity. The presence of PTH1-receptor autoantibodies should be considered in patients with acquired PTH resistance.</abstract><cop>United States</cop><pub>Massachusetts Medical Society</pub><pmid>34788508</pmid><doi>10.1056/NEJMoa2109409</doi><tpages>7</tpages><oa>free_for_read</oa></addata></record> |
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| source | MEDLINE; Elektronische Zeitschriftenbibliothek - Frei zugängliche E-Journals; New England Journal of Medicine |
| subjects | Adult Age Aged Allergy Antibodies Autoantibodies Autoantibodies - blood Autoimmune Disease Autoimmune diseases Biopsy Creatinine Diabetes mellitus Disease DNA Mutational Analysis Endocrinology Endocrinology General Family medical history Female Glomerulonephritis Glycopeptides - blood Humans Hypocalcemia Hypocalcemia - etiology Hypocalcemia - genetics Immunoglobulin G Immunoglobulin G - blood Immunology Immunophenotyping Kidney diseases Kidney Glomerulus - pathology Lupus Membranous glomerulonephritis Microscopy, Electron Mouth Muscle pain Mutation Parathyroid Parathyroid hormone Parathyroid Hormone - metabolism Patients Pseudohypoparathyroidism - genetics Receptor, Parathyroid Hormone, Type 1 - immunology Research programs Rheumatology Rheumatology General Thrombocytopenia Thyroid gland |
| title | Parathyroid Hormone Resistance and Autoantibodies to the PTH1 Receptor |
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