A Case of Mid‐Trimester Intrauterine Fetal Death with Cushing's Syndrome

Murakami, Soh; Saitoh, Masahiro; Kubo, Takeshi; Kawakami, Yasushi; Yamashita, Kamejiro

doi:10.1111/j.1447-0756.1998.tb00067.x

Cited by 18 publications

(11 citation statements)

References 17 publications

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“…There is also the worry that 21% of tumours are malignant (Pickard et al 1990). Fetal outcome is not good in Cushing's syndrome with fetal loss of 25% having been reported where pregnancy has not been interrupted (Anderson and Walters 1976;Murakami et al 1998). The reason for fetal loss is not always clear but because of a high stillbirth rate, maternal hyperglycaemia may play an important role.…”

Section: Discussionmentioning

confidence: 93%

Cushing's syndrome in pregnancy

Tejura

Weiner

Gibby

et al. 2005

Journal of Obstetrics and Gynaecology

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Section: Discussionmentioning

confidence: 93%

Cushing's syndrome in pregnancy

Tejura

Weiner

Gibby

et al. 2005

Journal of Obstetrics and Gynaecology

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“…As in all cases of CS-associated pregnancy, fetal prognosis was poor. Premature births, intrauterine growth retardation, stillbirths, and intrauterine deaths have been reported in all studies (13,(20)(21)(22). The prognosis may be improved by treating the cause of hypercortisolism during pregnancy (13).…”

Section: Discussionmentioning

confidence: 98%

Adrenocortical carcinoma and pregnancy: clinical and biological features and prognosis

Abiven-Lepage¹,

Coste²,

Tissier

et al. 2010

European Journal of Endocrinology

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Objective: Adrenocortical carcinoma (ACC) is a rare, severe disease. Pregnancy-associated ACC has rarely been reported. We wished to evaluate the characteristics and prognosis of ACC diagnosed in patients during pregnancy or in the postpartum period, comparing them with those for ACC diagnosed in nonpregnant women. Design: Clinical presentation, hormonal secretion, staging, survival, and obstetric data are reported. Patients were included between 1963 and 2007. Mean follow-up was 48 months. Patients and methods: This is a retrospective cohort study carried out at a referral center. All female patients aged 16-49 years diagnosed with ACC during the observation period were included (nZ110). Twelve of these women were pregnant or in the first 6 months after delivery. Hormonal secretion, staging, obstetric data, and survival were analyzed. For the survival analysis, pregnant patients were compared with a subgroup of nonpregnant women matched for age, stage, and year of diagnosis (1 pregnant patient/2 controls). Results: Adrenocortical tumors diagnosed during pregnancy or in the postpartum period tend to be more often cortisol-secreting tumors (PZ0.06) and to be discovered at a more advanced stage than those in nonpregnant women, although the differences were not significant. Fetal outcome was poor. Overall survival of the mother was worse than that of matched controls (hazard ratio of death: 3.98, confidence intervalZ1.34-11.85, PZ0.013). Conclusion: ACC diagnosed during pregnancy or in the postpartum period is associated with a poor fetal outcome and a poorer prognosis than ACC diagnosed in nonpregnant women.

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“…The most common cause of CS in pregnant women is an adrenal adenoma, in contrast to non-pregnant women in whom a pituitary adenoma is the most common cause of CS. This may be attributed to the fact that patients with an adrenal adenoma are most likely to be purely cortisol producing, thus their ovulatory function remains unaffected (15,18,19). Alternatively, pregnancy-induced hCG levels might have triggered a preexisting subclinical CS in these patients due to the aberrant expression of LH receptors in adrenocortical cells of an adrenal adenoma, whereas pre-pregnancy LH levels might be insufficient for the development of CS.…”

Section: Discussionmentioning

confidence: 99%

Adrenal Cushing’s syndrome during pregnancy

Andreescu

Alwani

Hofland

et al. 2017

European Journal of Endocrinology

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Cushing syndrome (CS) during pregnancy is a rare condition with only a few cases reported in the literature. Misdiagnosis of CS is common because of overlapping features like fatigue, weight gain, striae and emotional changes that can occur during normal pregnancy. Changes in maternal hormones and their binding proteins complicate assessment of glucocorticoid hormone levels during gestation. CS during pregnancy is most frequently due to an adrenal adenoma and to a lesser degree to adrenocorticotropic hormone (ACTH) hypersecretion by a pituitary adenoma. Furthermore, aberrant expression of luteinizing hormone (LH) receptors in the adrenal cortex has been suggested to be involved in the pathogenesis of adrenal CS during pregnancy. We report three pregnant women with ACTH-independent Cushing's syndrome and an adrenal tumor. After uncomplicated delivery, patient 1 underwent testing for aberrant hormone receptor expression by the adenoma. Cortisol responses were found after administration of luteinizing hormone-releasing hormone (LHRH), human chorionic gonadotropin (hCG), glucagon, vasopressin and a standard mixed meal. All patients were treated with laparoscopic adrenalectomy. Adrenal tumor tissue of two patients showed positive immunohistochemical staining of LH receptors. Considering the cortisol responses to LHRH and hCG, and the development of CS during pregnancy in these patients, it is likely that ACTH-independent hypercortisolism was induced by the pregnancy-associated rise in hCG levels that activated aberrantly expressed LH receptors in the adrenal adenoma. Remarkably, adrenal adenomas may simultaneously express multiple aberrant receptors and individual ligands may play a role in the regulation of cortisol production in CS during pregnancy.

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A Case of Mid‐Trimester Intrauterine Fetal Death with Cushing's Syndrome

Cited by 18 publications

References 17 publications

Cushing's syndrome in pregnancy

Cushing's syndrome in pregnancy

Adrenocortical carcinoma and pregnancy: clinical and biological features and prognosis

Adrenal Cushing’s syndrome during pregnancy

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