Lung carcinoid related Cushing's syndrome: report of three cases and review of the literature
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Abstract Three patients with lung carcinoid related Cushing's syndrome (LCRCS) treated at Frenchay Hospital, Bristol between 1984 and 1994 are described. The first patient presented with hyperpigmentation 13 years after bilateral adrenalectomy. The second patient had no recurrence or metastases 14 years after removal of a typical carcinoid tumour. The last patient survived nine years after diagnosis of liver metastasis. The possibility of LCRCS should be considered in every patient proved to have Cushing's disease and bilateral adrenal enlargement on abdominal computed tomography. Biochemical sets of investigation (for example, adrenocorticotrophic hormone (ACTH) stimulation, dexamethasone suppression, and metyrapone response) could be misleading and should not be relied upon solely. Search for an ectopic ACTH source should be called off only when ACTH has been demonstrated in the surgically removed specimen, and most importantly, when the serum ACTH concentration returns to normal after surgery. Lung carcinoid tumours are compatible with long survival, and liver metastasis could prove indolent and slowly growing.Keywords:
Metyrapone
Bilateral adrenalectomy
Bilateral adrenalectomy
Hypophysectomy
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Reported here is a case of a 31- year-old male with Cushing's syndrome accompanied by mild cutaneous melanin pigmentation and benign junctional nevi. The clinical and biochemical abnormalities were completely reversed by bilateral adrenalectomy. Twelve months following operation malignant melanoma developed, involving the mediastinal and supraclavicular lymph nodes, lungs and bones. During the ensuing 14 months until his death from melanoma, the patient demonstrated generalized progressive melanin pigmentation of the skin which eventually assumed a dark chocolate color. A pituitary tumor was not demonstrable. Cutaneous melanin pigmentation associated with high levels of ACTH following adrenalectomy for Cushing's syndrome has been well documented previously. The occurrence of malignant melanoma under similar circumstances has not been reported. The possibility exists that the high levels of ACTH and melanophore-stimulating hormone observed in the post-adrenalectomy state can contribute to the production of malignant melanoma. It is suggested that patients subjected to adrenalectomy for Cushing's syndrome be carefully followed with this possibility in mind.
Bilateral adrenalectomy
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Abstract Purpose In selected cases of severe Cushing’s syndrome due to uncontrolled ACTH secretion, bilateral adrenalectomy appears unavoidable. Compared with unilateral adrenalectomy (for adrenal Cushing’s syndrome), bilateral adrenalectomy has a perceived higher perioperative morbidity. The aim of the current study was to compare both interventions in endogenous Cushing’s syndrome regarding postoperative outcomes. Methods We report a single-center, retrospective cohort study comparing patients with hypercortisolism undergoing bilateral vs. unilateral adrenalectomy during 2008–2021. Patients with adrenal Cushing’s syndrome due to adenoma were compared with patients with ACTH-dependent Cushing’s syndrome (Cushing’s disease and ectopic ACTH production) focusing on postoperative morbidity and mortality as well as long-term survival. Results Of 83 patients with adrenalectomy for hypercortisolism (65.1% female, median age 53 years), the indication for adrenalectomy was due to adrenal Cushing’s syndrome in 60 patients (72.2%; 59 unilateral and one bilateral), and due to hypercortisolism caused by Cushing’s disease ( n = 16) or non-pituitary uncontrolled ACTH secretion of unknown origin ( n = 7) (27.7% of all adrenalectomies). Compared with unilateral adrenalectomy ( n = 59), patients with bilateral adrenalectomy ( n = 24) had a higher rate of severe complications (0% vs. 33%; p < 0.001) and delayed recovery (median: 10.2% vs. 79.2%; p < 0.001). Using the MTL30 marker, patients with bilateral adrenalectomy fared worse than patients after unilateral surgery (MTL30 positive: 7.2% vs. 25.0% p < 0.001). Postoperative mortality was increased in patients with bilateral adrenalectomy (0% vs. 8.3%; p = 0.081). Conclusion While unilateral adrenalectomy for adrenal Cushing’s syndrome represents a safe and definitive therapeutic option, bilateral adrenalectomy to control ACTH-dependent extra-adrenal Cushing’s syndrome or Cushing’s disease is a more complicated intervention with a mortality of nearly 10%.
Cushing's disease
Bilateral adrenalectomy
Adrenal adenoma
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Two cases of recurrent Cushing's syndrome after assumed total bilateral adrenalectomy are presented. Both cases were due to adrenal tissue left at the adrenalectomy. In the first case the lack of preoperative localization of the adrenal remnant caused severe postoperative complications. In the other patient localization of the adrenal tissue was achieved by scanning after administration of 131I-19-iodocholesterol. The reoperation was easy and the postoperative course smooth.
Bilateral adrenalectomy
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Bilateral adrenalectomy
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Bilateral adrenalectomy
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Bilateral adrenalectomy
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Aminoglutethimide
Bilateral adrenalectomy
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Forty-three patients (two males and 41 females) were submitted to bilateral adrenalectomy for disseminated mammary carcinoma. Several factors were considered for their prognostic value in predicting the response to endocrine ablation. The age of the patients, the treatment of the primary lesion, the free interval, the location of metastases and the involvement of regional lymph nodes did not affect the response to adrenalectomy. Response to therapeutic castration was directly correlated to the response to adrenalectomy (P less than 0.01); this is a valuable predicting factor that should be taken into consideration before proceeding to bilateral adrenalectomy. A statistically significant variation in survival between the responders and nonresponders was found (P less than 0.05). Finally, the high response rate to the operation (53.4%) can be attributed to the complete removal of the adrenal tissue during the operation.
Bilateral adrenalectomy
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Bilateral adrenalectomy
Estrogen therapy
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