Cargando…
Remitting Seronegative Symmetrical Synovitis With Subsequent IgA Hypergammaglobulinemia. A Case of Chronic Inflammation Causing Macroprolactinemia and a Misdiagnosis
Prolactin is a pituitary hormone that functions in breast development and milk production in women and also plays an important role in immunoregulation and human immune responses, including autoimmune diseases. Macroprolactin, known as “big-big prolactin”, is due to the presence of marked hyperprola...
Autores principales: | , |
---|---|
Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Oxford University Press
2021
|
Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8265976/ http://dx.doi.org/10.1210/jendso/bvab048.1238 |
_version_ | 1783719844816879616 |
---|---|
author | Zarghamravanbakhsh, Paria Sanchez, Jose Gonzalo |
author_facet | Zarghamravanbakhsh, Paria Sanchez, Jose Gonzalo |
author_sort | Zarghamravanbakhsh, Paria |
collection | PubMed |
description | Prolactin is a pituitary hormone that functions in breast development and milk production in women and also plays an important role in immunoregulation and human immune responses, including autoimmune diseases. Macroprolactin, known as “big-big prolactin”, is due to the presence of marked hyperprolactinemia associated with evidence of prolactin-Ig (typically IgG4 or less frequently IgA) circulating complexes. We describes a case of 51 year-old female, with more than a 4 year history of reported hyperprolcatinemia who was treated with Cabergoline 0.5 mg weekly. Prior to treatment, she reported menses every 30-40 days, but denied galactorrhea or symptoms of sellar mass effect. Our patient had mildly elevated prolactin levels of 40-50 ng/dl. Her Thyroid function test were within normal limits. Patient had two pituitary MRIs in 2017 and 2019 which did not show sellar abnormalities. Prior to cabergolibe initiation, she was diagnosed with RS3PE syndrome (Remitting seronegative symmetrical synovitis with pitting edema) due to bilateral swelling in the dorsum of her hands. She was found to have hypergammaglobulinemia which was related to IgA elevation from chronic inflammation. Further investigation showed actual bio-active monomeric prolactin level was normal (4.8 ng/dl) and macroprolactin elevation from hypergammagolbulinemia. Before diagnosing her paraproteinemia and her macroprolactin predominance, she had received years of dopamine agonist therapy which was discontinued after diagnosis. We report a novel association of IgA predominant hypergammaglobulinemia from a chronic rheumatologic condition, leading to a misdiagnosed hyperprolcatinemia. Care should be taken to determine monomeric Prolactin levels prior to treatment, specially when symptoms are equivocal and/or imaging studies are negative. |
format | Online Article Text |
id | pubmed-8265976 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2021 |
publisher | Oxford University Press |
record_format | MEDLINE/PubMed |
spelling | pubmed-82659762021-07-09 Remitting Seronegative Symmetrical Synovitis With Subsequent IgA Hypergammaglobulinemia. A Case of Chronic Inflammation Causing Macroprolactinemia and a Misdiagnosis Zarghamravanbakhsh, Paria Sanchez, Jose Gonzalo J Endocr Soc Neuroendocrinology and Pituitary Prolactin is a pituitary hormone that functions in breast development and milk production in women and also plays an important role in immunoregulation and human immune responses, including autoimmune diseases. Macroprolactin, known as “big-big prolactin”, is due to the presence of marked hyperprolactinemia associated with evidence of prolactin-Ig (typically IgG4 or less frequently IgA) circulating complexes. We describes a case of 51 year-old female, with more than a 4 year history of reported hyperprolcatinemia who was treated with Cabergoline 0.5 mg weekly. Prior to treatment, she reported menses every 30-40 days, but denied galactorrhea or symptoms of sellar mass effect. Our patient had mildly elevated prolactin levels of 40-50 ng/dl. Her Thyroid function test were within normal limits. Patient had two pituitary MRIs in 2017 and 2019 which did not show sellar abnormalities. Prior to cabergolibe initiation, she was diagnosed with RS3PE syndrome (Remitting seronegative symmetrical synovitis with pitting edema) due to bilateral swelling in the dorsum of her hands. She was found to have hypergammaglobulinemia which was related to IgA elevation from chronic inflammation. Further investigation showed actual bio-active monomeric prolactin level was normal (4.8 ng/dl) and macroprolactin elevation from hypergammagolbulinemia. Before diagnosing her paraproteinemia and her macroprolactin predominance, she had received years of dopamine agonist therapy which was discontinued after diagnosis. We report a novel association of IgA predominant hypergammaglobulinemia from a chronic rheumatologic condition, leading to a misdiagnosed hyperprolcatinemia. Care should be taken to determine monomeric Prolactin levels prior to treatment, specially when symptoms are equivocal and/or imaging studies are negative. Oxford University Press 2021-05-03 /pmc/articles/PMC8265976/ http://dx.doi.org/10.1210/jendso/bvab048.1238 Text en © The Author(s) 2021. Published by Oxford University Press on behalf of the Endocrine Society. https://creativecommons.org/licenses/by-nc-nd/4.0/This is an Open Access article distributed under the terms of the Creative Commons Attribution-NonCommercial-NoDerivs licence (http://creativecommons.org/licenses/by-nc-nd/4.0/ (https://creativecommons.org/licenses/by-nc-nd/4.0/) ), which permits non-commercial reproduction and distribution of the work, in any medium, provided the original work is not altered or transformed in any way, and that the work is properly cited. For commercial re-use, please contact journals.permissions@oup.com |
spellingShingle | Neuroendocrinology and Pituitary Zarghamravanbakhsh, Paria Sanchez, Jose Gonzalo Remitting Seronegative Symmetrical Synovitis With Subsequent IgA Hypergammaglobulinemia. A Case of Chronic Inflammation Causing Macroprolactinemia and a Misdiagnosis |
title | Remitting Seronegative Symmetrical Synovitis With Subsequent IgA Hypergammaglobulinemia. A Case of Chronic Inflammation Causing Macroprolactinemia and a Misdiagnosis |
title_full | Remitting Seronegative Symmetrical Synovitis With Subsequent IgA Hypergammaglobulinemia. A Case of Chronic Inflammation Causing Macroprolactinemia and a Misdiagnosis |
title_fullStr | Remitting Seronegative Symmetrical Synovitis With Subsequent IgA Hypergammaglobulinemia. A Case of Chronic Inflammation Causing Macroprolactinemia and a Misdiagnosis |
title_full_unstemmed | Remitting Seronegative Symmetrical Synovitis With Subsequent IgA Hypergammaglobulinemia. A Case of Chronic Inflammation Causing Macroprolactinemia and a Misdiagnosis |
title_short | Remitting Seronegative Symmetrical Synovitis With Subsequent IgA Hypergammaglobulinemia. A Case of Chronic Inflammation Causing Macroprolactinemia and a Misdiagnosis |
title_sort | remitting seronegative symmetrical synovitis with subsequent iga hypergammaglobulinemia. a case of chronic inflammation causing macroprolactinemia and a misdiagnosis |
topic | Neuroendocrinology and Pituitary |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8265976/ http://dx.doi.org/10.1210/jendso/bvab048.1238 |
work_keys_str_mv | AT zarghamravanbakhshparia remittingseronegativesymmetricalsynovitiswithsubsequentigahypergammaglobulinemiaacaseofchronicinflammationcausingmacroprolactinemiaandamisdiagnosis AT sanchezjosegonzalo remittingseronegativesymmetricalsynovitiswithsubsequentigahypergammaglobulinemiaacaseofchronicinflammationcausingmacroprolactinemiaandamisdiagnosis |