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MON-346 A Rare Case of Recurring Adrenal Hemorrhage with Consecutive Pregnancies

Introduction - Adrenal hemorrhage (AH) during pregnancy is a rare condition with an incidence ranging from 0.14% to 1.1%. The objective of this report is to describe a case of recurring AH in successive pregnancies. Case Report - A 25-year-old female, G3P1011, 33 weeks of gestation, presented to the...

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Autores principales: Kumar, Monisha Priyadarshini, Dhamija, Yashu, Ciltea, Daniela
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Endocrine Society 2019
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6551066/
http://dx.doi.org/10.1210/js.2019-MON-346
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author Kumar, Monisha Priyadarshini
Dhamija, Yashu
Ciltea, Daniela
author_facet Kumar, Monisha Priyadarshini
Dhamija, Yashu
Ciltea, Daniela
author_sort Kumar, Monisha Priyadarshini
collection PubMed
description Introduction - Adrenal hemorrhage (AH) during pregnancy is a rare condition with an incidence ranging from 0.14% to 1.1%. The objective of this report is to describe a case of recurring AH in successive pregnancies. Case Report - A 25-year-old female, G3P1011, 33 weeks of gestation, presented to the ER with the chief complaint of left flank pain. Her past medical history was pertinent for spontaneous abortion of first pregnancy, spontaneous right AH and adrenal insufficiency during the second pregnancy, with an eventual spontaneous vaginal delivery of viable child following medical management. History was negative for personal or family history of thromboembolic disease, anticoagulation use, trauma, signs or symptoms of infection. On admission, her vitals were stable. Pertinent physical exam findings were tenderness to deep palpation of the abdomen on the left upper quadrant. Laboratory results, including hemoglobin, were within normal limits. The US of abdomen revealed fat stranding surrounding the left adrenal gland. Given the concern for AH, after discussing risks and benefits, a CT abdomen without IV contrast was done and revealed abnormally enlarged left adrenal gland with high density, suggestive of hemorrhage. Her random serum cortisol level was low (0.7μg/dL; 7-28 μg/dL). Basal, 30-minute, and 60-minute serum cortisol with low dose ACTH stimulation testing were 0.6μg/dL (minimum 5.5μg/dL), 2.0μg/dL (minimum 7μg/dL), 3.1μg/dL (minimum 18μg/dL), confirming primary adrenal insufficiency and she was started on IV hydrocortisone. Work up for hypercoagulability and bleeding disorder comprising ANA, anticardiolipin antibody, protein C, protein S, antithrombin 3, von Willebrand assay, ristocetin cofactor, plasminogen activity, paroxysmal nocturnal hemoglobinuria panel, platelet function screen, and genetic studies were all normal. Obstetrical US showed single live intrauterine pregnancy, and given hemodynamic stability, she was managed medically. Discussion - This report highlights a case of recurrent AH in consecutive pregnancies, involving opposite adrenal glands and no identifiable hypercoagulable state, which has not been previously reported. During pregnancy, placental CRH stimulates the maternal pituitary gland, increasing ACTH, which subsequently leads to adrenal hypertrophy. The hypertrophied adrenal glands are prone to vascular congestion and can lead to AH. Pregnancy, being a hypercoagulable state, adds to the risk factor as well. AH should be included in the differential when evaluating abdominal pain in prenatal individuals. Emergent surgery and preterm delivery are warranted in patients with hemodynamic instability.
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spelling pubmed-65510662019-06-13 MON-346 A Rare Case of Recurring Adrenal Hemorrhage with Consecutive Pregnancies Kumar, Monisha Priyadarshini Dhamija, Yashu Ciltea, Daniela J Endocr Soc Adrenal Introduction - Adrenal hemorrhage (AH) during pregnancy is a rare condition with an incidence ranging from 0.14% to 1.1%. The objective of this report is to describe a case of recurring AH in successive pregnancies. Case Report - A 25-year-old female, G3P1011, 33 weeks of gestation, presented to the ER with the chief complaint of left flank pain. Her past medical history was pertinent for spontaneous abortion of first pregnancy, spontaneous right AH and adrenal insufficiency during the second pregnancy, with an eventual spontaneous vaginal delivery of viable child following medical management. History was negative for personal or family history of thromboembolic disease, anticoagulation use, trauma, signs or symptoms of infection. On admission, her vitals were stable. Pertinent physical exam findings were tenderness to deep palpation of the abdomen on the left upper quadrant. Laboratory results, including hemoglobin, were within normal limits. The US of abdomen revealed fat stranding surrounding the left adrenal gland. Given the concern for AH, after discussing risks and benefits, a CT abdomen without IV contrast was done and revealed abnormally enlarged left adrenal gland with high density, suggestive of hemorrhage. Her random serum cortisol level was low (0.7μg/dL; 7-28 μg/dL). Basal, 30-minute, and 60-minute serum cortisol with low dose ACTH stimulation testing were 0.6μg/dL (minimum 5.5μg/dL), 2.0μg/dL (minimum 7μg/dL), 3.1μg/dL (minimum 18μg/dL), confirming primary adrenal insufficiency and she was started on IV hydrocortisone. Work up for hypercoagulability and bleeding disorder comprising ANA, anticardiolipin antibody, protein C, protein S, antithrombin 3, von Willebrand assay, ristocetin cofactor, plasminogen activity, paroxysmal nocturnal hemoglobinuria panel, platelet function screen, and genetic studies were all normal. Obstetrical US showed single live intrauterine pregnancy, and given hemodynamic stability, she was managed medically. Discussion - This report highlights a case of recurrent AH in consecutive pregnancies, involving opposite adrenal glands and no identifiable hypercoagulable state, which has not been previously reported. During pregnancy, placental CRH stimulates the maternal pituitary gland, increasing ACTH, which subsequently leads to adrenal hypertrophy. The hypertrophied adrenal glands are prone to vascular congestion and can lead to AH. Pregnancy, being a hypercoagulable state, adds to the risk factor as well. AH should be included in the differential when evaluating abdominal pain in prenatal individuals. Emergent surgery and preterm delivery are warranted in patients with hemodynamic instability. Endocrine Society 2019-04-30 /pmc/articles/PMC6551066/ http://dx.doi.org/10.1210/js.2019-MON-346 Text en Copyright © 2019 Endocrine Society https://creativecommons.org/licenses/by-nc-nd/4.0/ This article has been published under the terms of the Creative Commons Attribution Non-Commercial, No-Derivatives License (CC BY-NC-ND; https://creativecommons.org/licenses/by-nc-nd/4.0/).
spellingShingle Adrenal
Kumar, Monisha Priyadarshini
Dhamija, Yashu
Ciltea, Daniela
MON-346 A Rare Case of Recurring Adrenal Hemorrhage with Consecutive Pregnancies
title MON-346 A Rare Case of Recurring Adrenal Hemorrhage with Consecutive Pregnancies
title_full MON-346 A Rare Case of Recurring Adrenal Hemorrhage with Consecutive Pregnancies
title_fullStr MON-346 A Rare Case of Recurring Adrenal Hemorrhage with Consecutive Pregnancies
title_full_unstemmed MON-346 A Rare Case of Recurring Adrenal Hemorrhage with Consecutive Pregnancies
title_short MON-346 A Rare Case of Recurring Adrenal Hemorrhage with Consecutive Pregnancies
title_sort mon-346 a rare case of recurring adrenal hemorrhage with consecutive pregnancies
topic Adrenal
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6551066/
http://dx.doi.org/10.1210/js.2019-MON-346
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