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Pregnancy complicated with adrenal adenoma causing ACTH-independent Cushing’s syndrome, accompanied by obstetric antiphospholipid syndrome and severe pre-eclampsia: case report and literature review
This case report shares the management experience of a patient with pregnancy combined with adrenal adenoma causing ACTH-independent Cushing’s syndrome (CS), accompanied by obstetric antiphospholipid syndrome (OAPS) and severe pre-eclampsia. The case was a 26-year-old that presented with typical cli...
Autores principales: | , , , , , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
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Frontiers Media S.A.
2023
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Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10235702/ https://www.ncbi.nlm.nih.gov/pubmed/37274343 http://dx.doi.org/10.3389/fendo.2023.1147316 |
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author | Xu, Shenghan Liu, Miao Xu, Jiamu Che, Bangwei Zhang, Wenjun Li, Wei Huang, Tao Yu, Ying Zha, Cheng Peng, Zheng Huang, Kunyuan Tang, Kaifa |
author_facet | Xu, Shenghan Liu, Miao Xu, Jiamu Che, Bangwei Zhang, Wenjun Li, Wei Huang, Tao Yu, Ying Zha, Cheng Peng, Zheng Huang, Kunyuan Tang, Kaifa |
author_sort | Xu, Shenghan |
collection | PubMed |
description | This case report shares the management experience of a patient with pregnancy combined with adrenal adenoma causing ACTH-independent Cushing’s syndrome (CS), accompanied by obstetric antiphospholipid syndrome (OAPS) and severe pre-eclampsia. The case was a 26-year-old that presented with typical clinical symptoms and signs of CS. The patient had a history of 4 spontaneous abortions in the last 4 years. The 24-hour urinary free cortisol was significantly increased, an abnormal cortisol circadian rhythm was demonstrated by a high late-night salivary cortisol, blood ACTH was suppressed (< 1ng/dL), anticardiolipin antibody was positive, and imaging examination showed an adrenal tumor. The patient underwent laparoscopic adrenal tumor resection under general anesthesia at 23 weeks of gestation. The tumor was pathologically confirmed to be an adrenocortical adenoma. The patient underwent a cesarean section at 39 weeks of gestation to give birth to a healthy baby girl with an Apgar score of 10. Pregnancy complicated by CS is clinically rare, easily masked by normal physiological changes of pregnancy, and is difficult to diagnose. The determination of 24-hour urinary free cortisol, the circadian rhythm of serum cortisol, ultrasound, and MRI can be helpful in the diagnosis of CS during pregnancy. Surgery is the first choice for the treatment of CS during pregnancy. As a subtype of antiphospholipid syndrome, patients with OAPS are prone to thrombotic events and recurrent miscarriages if not treated accordingly. To our knowledge no cases of CS with OAPS and severe pre-eclampsia have been reported. We summarize the experience of the treatment of this patient and review the literature to improve clinicians’ awareness of this disease. |
format | Online Article Text |
id | pubmed-10235702 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2023 |
publisher | Frontiers Media S.A. |
record_format | MEDLINE/PubMed |
spelling | pubmed-102357022023-06-03 Pregnancy complicated with adrenal adenoma causing ACTH-independent Cushing’s syndrome, accompanied by obstetric antiphospholipid syndrome and severe pre-eclampsia: case report and literature review Xu, Shenghan Liu, Miao Xu, Jiamu Che, Bangwei Zhang, Wenjun Li, Wei Huang, Tao Yu, Ying Zha, Cheng Peng, Zheng Huang, Kunyuan Tang, Kaifa Front Endocrinol (Lausanne) Endocrinology This case report shares the management experience of a patient with pregnancy combined with adrenal adenoma causing ACTH-independent Cushing’s syndrome (CS), accompanied by obstetric antiphospholipid syndrome (OAPS) and severe pre-eclampsia. The case was a 26-year-old that presented with typical clinical symptoms and signs of CS. The patient had a history of 4 spontaneous abortions in the last 4 years. The 24-hour urinary free cortisol was significantly increased, an abnormal cortisol circadian rhythm was demonstrated by a high late-night salivary cortisol, blood ACTH was suppressed (< 1ng/dL), anticardiolipin antibody was positive, and imaging examination showed an adrenal tumor. The patient underwent laparoscopic adrenal tumor resection under general anesthesia at 23 weeks of gestation. The tumor was pathologically confirmed to be an adrenocortical adenoma. The patient underwent a cesarean section at 39 weeks of gestation to give birth to a healthy baby girl with an Apgar score of 10. Pregnancy complicated by CS is clinically rare, easily masked by normal physiological changes of pregnancy, and is difficult to diagnose. The determination of 24-hour urinary free cortisol, the circadian rhythm of serum cortisol, ultrasound, and MRI can be helpful in the diagnosis of CS during pregnancy. Surgery is the first choice for the treatment of CS during pregnancy. As a subtype of antiphospholipid syndrome, patients with OAPS are prone to thrombotic events and recurrent miscarriages if not treated accordingly. To our knowledge no cases of CS with OAPS and severe pre-eclampsia have been reported. We summarize the experience of the treatment of this patient and review the literature to improve clinicians’ awareness of this disease. Frontiers Media S.A. 2023-05-19 /pmc/articles/PMC10235702/ /pubmed/37274343 http://dx.doi.org/10.3389/fendo.2023.1147316 Text en Copyright © 2023 Xu, Liu, Xu, Che, Zhang, Li, Huang, Yu, Zha, Peng, Huang and Tang https://creativecommons.org/licenses/by/4.0/This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms. |
spellingShingle | Endocrinology Xu, Shenghan Liu, Miao Xu, Jiamu Che, Bangwei Zhang, Wenjun Li, Wei Huang, Tao Yu, Ying Zha, Cheng Peng, Zheng Huang, Kunyuan Tang, Kaifa Pregnancy complicated with adrenal adenoma causing ACTH-independent Cushing’s syndrome, accompanied by obstetric antiphospholipid syndrome and severe pre-eclampsia: case report and literature review |
title | Pregnancy complicated with adrenal adenoma causing ACTH-independent Cushing’s syndrome, accompanied by obstetric antiphospholipid syndrome and severe pre-eclampsia: case report and literature review |
title_full | Pregnancy complicated with adrenal adenoma causing ACTH-independent Cushing’s syndrome, accompanied by obstetric antiphospholipid syndrome and severe pre-eclampsia: case report and literature review |
title_fullStr | Pregnancy complicated with adrenal adenoma causing ACTH-independent Cushing’s syndrome, accompanied by obstetric antiphospholipid syndrome and severe pre-eclampsia: case report and literature review |
title_full_unstemmed | Pregnancy complicated with adrenal adenoma causing ACTH-independent Cushing’s syndrome, accompanied by obstetric antiphospholipid syndrome and severe pre-eclampsia: case report and literature review |
title_short | Pregnancy complicated with adrenal adenoma causing ACTH-independent Cushing’s syndrome, accompanied by obstetric antiphospholipid syndrome and severe pre-eclampsia: case report and literature review |
title_sort | pregnancy complicated with adrenal adenoma causing acth-independent cushing’s syndrome, accompanied by obstetric antiphospholipid syndrome and severe pre-eclampsia: case report and literature review |
topic | Endocrinology |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10235702/ https://www.ncbi.nlm.nih.gov/pubmed/37274343 http://dx.doi.org/10.3389/fendo.2023.1147316 |
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