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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...

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Autores principales: Xu, Shenghan, Liu, Miao, Xu, Jiamu, Che, Bangwei, Zhang, Wenjun, Li, Wei, Huang, Tao, Yu, Ying, Zha, Cheng, Peng, Zheng, Huang, Kunyuan, Tang, Kaifa
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2023
Materias:
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.
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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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