A successful case of Cushing's disease pregnancy treated with ketoconazole

Costenaro, Fabíola; Rodrigues, Ticiana C.; Lima, Paula Borges de; Ruszczyk, Juliana; Rollin, G; Czepielewski, Mauro Antônio

doi:10.3109/09513590.2014.995615

Cited by 15 publications

(7 citation statements)

References 12 publications

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“…Metyrapone, 31,35,70,[74][75][76][77][78] ketoconazole, 70,74,[79][80][81] cabergoline, 80,[82][83][84] mitotane, 85,86 cyproheptadine, [87][88][89] and aminogluthemide 5,6 have all been used in the management of patients with CS during pregnancy. Each is discussed in turn, along with their benefits and limitations, in Table 3.…”

Section: Medical Therapymentioning

confidence: 99%

The diagnosis and management of Cushing's syndrome in pregnancy

Hamblin

Coulden

Fountas

et al. 2022

J Neuroendocrinology

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Endogenous Cushing's syndrome (CS) is rarely encountered during pregnancy. Clinical and biochemical changes in healthy pregnancy overlap with those seen in pregnancy complicated by CS; the diagnosis is therefore challenging and can be delayed. During normal gestation, adrenocorticotrophic hormone, corticotrophin‐releasing hormone, cortisol, and urinary free cortisol levels rise. Dexamethasone administration fails to fully suppress cortisol in pregnant women without CS. Localisation may be hindered by non‐suppressed adrenocorticotrophic hormone levels in a large proportion of those with adrenal CS; smaller corticotroph adenomas may go undetected as a result of a lack of contrast administration or the presence of pituitary hyperplasia; and inferior petrosal sinus sampling is not recommended given the risk of radiation and thrombosis. Yet, diagnosis is essential; active disease is associated with multiple insults to both maternal and foetal health, and those cured may normalise the risk of maternal–foetal complications. The published literature consists mostly of case reports or small case series affected by publication bias, heterogeneous definitions of maternal or foetal outcomes or lack of detail on severity of hypercortisolism. Consequently, conclusive recommendations, or a standardised management approach for all, cannot be made. Management is highly individualised: the decision for surgery, medical control of hypercortisolism or adoption of a conservative approach is dependent on the timing of diagnosis (respective to stage of gestation), the ability to localise the tumour, severity of CS, pre‐existing maternal comorbidity, and, ultimately, patient choice. Close communication is a necessity with the patient placed at the centre of all decisions, with risks, benefits, and uncertainties around any investigation and management carefully discussed. Care should be delivered by an experienced, multidisciplinary team, with the resources and expertise available to manage such a rare and challenging condition during pregnancy.

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Section: Medical Therapymentioning

confidence: 99%

The diagnosis and management of Cushing's syndrome in pregnancy

Hamblin

Coulden

Fountas

et al. 2022

J Neuroendocrinology

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“…If the diagnosis of CD is confirmed during the gestational period and the decision to perform an intervention is reached, TSS could be the treatment of choice in the second trimester (29). For those who are not eligible for surgery or has failure of surgery, medical treatment should be cautiously used to achieve better outcomes of CD and pregnancy (6,30). Cabergoline seems to confer good control of hypercortisolism during pregnancy (7,8).…”

Section: Difficulties In Management Of Pregnancy-associated CDmentioning

confidence: 99%

The Incidence of Pregnancy-Associated Cushing's Disease and Its Relation to Pregnancy: A Retrospective Study

et al. 2020

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Conclusions: In this study, a total of 27.1% of women of reproductive age with CD had pregnancy-associated CD, which might be induced by the hormonal milieu Tang et al. Pregnancy-Associated Cushing's Disease of pregnancy. An increased risk of having a LBW newborn was observed among mothers with pregnancy-associated CD. A high degree of clinical suspicion for CD may be warranted in the peripartum period. Patients with symptoms suspicious for CD throughout pregnancy and after childbirth, such as early-onset hypertension, severe hyperglycemia, and persistent weight gain, should be carefully diagnosed and closely monitored by clinicians.

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“…26,27 Despite this, ketoconazole has successfully been used in a small number of cases of Cushing's syndrome without any adverse consequences. 27,28 Cushings disease: Owing to the rarity of this condition, there appear to be no guidelines with regards to diagnosis and management of Cushing's syndrome in pregnancy. A high index of clinical suspicion can aid swift diagnosis and early treatment, reducing maternal and fetal morbidity and improving pregnancy outcomes.…”

Section: Weeks Of Gestationmentioning

confidence: 99%

Surgery for Cushing’s disease in pregnancy: our experience and a literature review

Jolly

Darr

Arlt

et al. 2019

annals

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Cushing’s syndrome in pregnancy is a rare phenomenon and there is limited literature on its management. Cushing’s disease in pregnancy is even less common and there is little guidance to help in the treatment for this patient group. Diagnosis of Cushing’s syndrome in pregnancy is often delayed due to overlap of symptoms. In addition, there are no validated diagnostic tests or parameters documented. We present a case of a 30-year-old woman presenting to the antenatal clinic at 13 weeks of pregnancy with high suspicion of Cushing’s disease. Her 21-week fetal scan showed a congenital diaphragmatic hernia and she underwent pituitary magnetic resonance imaging, which confirm Cushing’s disease. She successfully underwent transsphenoidal adenomectomy with histology confirming a corticotroph adenoma. Tests following transsphenoidal surgery confirmed remission of Cushing’s disease and she underwent an emergency caesarean section at 38 weeks. Unfortunately, her baby died from complications associated with the congenital abnormality 36 hours after birth. The patient remains in remission following delivery. To date, there have been no reported cases of congenital diaphragmatic hernia associated with Cushing’s disease in pregnancy. In addition, we believe that this is only the eighth reported patient to have undergone successful transsphenoidal surgery for Cushing’s disease in pregnancy.

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A successful case of Cushing's disease pregnancy treated with ketoconazole

Cited by 15 publications

References 12 publications

The diagnosis and management of Cushing's syndrome in pregnancy

The diagnosis and management of Cushing's syndrome in pregnancy

The Incidence of Pregnancy-Associated Cushing's Disease and Its Relation to Pregnancy: A Retrospective Study

Surgery for Cushing’s disease in pregnancy: our experience and a literature review

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