Laparoscopic Bilateral Adrenalectomy for Occult Ectopic ACTH Syndrome

Jihad, R Salameh; Karen, R Borman; George, Mariamma

doi:10.1089/lap.2007.0016

Cited by 12 publications

(11 citation statements)

References 11 publications

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“…Bilateral (or even unilateral) adrenalectomy may be considered when medical treatment is ineffective, poorly tolerated or rejected by the patient, in young women desiring pregnancy or when the ectopic source is unresectable or occult. Bilateral adrenalectomy has been shown to substantially improve the quality of life by relieving the symptoms and signs of Cushing's syndrome (7). A laparoscopic approach is preferable because of associated lower morbidity and mortality compared to an open approach (7).…”

Section: Discussionmentioning

confidence: 99%

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A case of severe ectopic ACTH syndrome from an occult primary – diagnostic and management dilemmas

Venugopal¹,

Griffin²,

Amer³

2015

Endocrinology, Diabetes &Amp; Metabolism Case Reports

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SummaryResection of primary tumour is the management of choice in patients with ectopic ACTH syndrome. However, tumours may remain unidentified or occult in spite of extensive efforts at trying to locate them. This can, therefore, pose a major management issue as uncontrolled hypercortisolaemia can lead to life-threatening infections. We present the case of a 66-year-old gentleman with ectopic ACTH syndrome from an occult primary tumour with multiple significant complications from hypercortisolaemia. Ectopic nature of his ACTH-dependent Cushing's syndrome was confirmed by non-suppression with high-dose dexamethasone suppression test and bilateral inferior petrosal sinus sampling. The primary ectopic source remained unidentified in spite of extensive anatomical and functional imaging studies, including CT scans and Dotatate-PET scan. Medical adrenolytic treatment at maximum tolerated doses failed to control his hypercortisolaemia, which led to recurrent intra-abdominal and pelvic abscesses, requiring multiple surgical interventions. Laparoscopic bilateral adrenalectomy was considered but decided against given concerns of technical difficulties due to recurrent intra-abdominal infections and his moribund state. Eventually, alcohol ablation of adrenal glands by retrograde adrenal vein approach was attempted, which resulted in biochemical remission of Cushing's syndrome. Our case emphasizes the importance of aggressive management of hypercortisolaemia in order to reduce the associated morbidity and mortality and also demonstrates that techniques like percutaneous adrenal ablation using a retrograde venous approach may be extremely helpful in patients who are otherwise unable to undergo bilateral adrenalectomy.Learning points Evaluation and management of patients with ectopic ACTH syndrome from an unidentified primary tumour can be very challenging.Persisting hypercortisolaemia in this setting can lead to debilitating and even life-threatening complications and hence needs to be managed aggressively.Bilateral adrenalectomy should be considered when medical treatment is ineffective or poorly tolerated.Percutaneous adrenal ablation may be considered in patients who are otherwise unable to undergo bilateral adrenalectomy.

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Section: Discussionmentioning

confidence: 99%

“…Bilateral adrenalectomy has been shown to substantially improve the quality of life by relieving the symptoms and signs of Cushing's syndrome (7). A laparoscopic approach is preferable because of associated lower morbidity and mortality compared to an open approach (7).…”

Section: Discussionmentioning

confidence: 99%

A case of severe ectopic ACTH syndrome from an occult primary – diagnostic and management dilemmas

Venugopal¹,

Griffin²,

Amer³

2015

Endocrinology, Diabetes &Amp; Metabolism Case Reports

View full text Add to dashboard Cite

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“…In these cases, bilateral adrenalectomy, preferably performed laparoscopically, represents a therapeutic option. It is effective treatment with low morbidity and mortality in experienced hands [15]. However, these patients will require long-term glucocorticoid and mineralocorticoid replacement.…”

Section: Discussionmentioning

confidence: 99%

“…Prognosis depends on the primary tumour histology. Patients with small cell lung carcinoma (SCLC) had the worst prognosis, usually dying within 12 months of diagnosis (median 6–8 months) [9, 15, 16]. Patients with bronchial carcinoids have the best prognosis and are usually considered to have low to moderate grade malignancy.…”

Section: Discussionmentioning

confidence: 99%

Cushing’s Syndrome due to Ectopic ACTH from Bronchial Carcinoid: A Case Report and Review

Kenchaiah

Hyer

2012

Case Reports in Endocrinology

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Despite advances in analytic and imaging techniques, the syndrome of ectopic adrenocorticotrophic hormone (ACTH) secretion from a tumour resulting in Cushing's syndrome continues to pose difficult diagnostic and therapeutic challenges. Dynamic testing may be equivocal and radiology indeterminate. We report a patient presenting with Cushing's syndrome associated with ectopic ACTH secretion from a bronchial carcinoid whose management presented diagnostic and therapeutic challenges.

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“…Such cases with ''occult'' ACTH-secreting tumours remain a challenge and may need repeated investigations for many years. In such cases, bilateral adrenalectomy is the next best therapeutic option, but with the need for lifelong steroid replacement (14).…”

Section: Case Reportmentioning

confidence: 99%

Challenges in the diagnosis and management of Cushing’s syndrome due to ectopic ACTH from bronchial carcinoid

Cooray

Somasundaram

Fernando

et al. 2015

Sri Lanka J. Diabetes Endocrinol. Metab.

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Laparoscopic Bilateral Adrenalectomy for Occult Ectopic ACTH Syndrome

Abstract: Laparoscopic bilateral adrenalectomy for ectopic ACTH syndrome refractory to medical management can be performed with low morbidity. Symptoms and signs of hypercortisolism rapidly improve postoperatively.

Cited by 12 publications

References 11 publications

A case of severe ectopic ACTH syndrome from an occult primary – diagnostic and management dilemmas

A case of severe ectopic ACTH syndrome from an occult primary – diagnostic and management dilemmas

Cushing’s Syndrome due to Ectopic ACTH from Bronchial Carcinoid: A Case Report and Review

Challenges in the diagnosis and management of Cushing’s syndrome due to ectopic ACTH from bronchial carcinoid

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