Use of In-111 pentetreotide scintigraphy in the diagnosis of a midgut carcinoid causing Cushing's syndrome

Segu, Venkatesh Babu G.; Mahvi, David M.; Wilson, Michael A.; Hale, Sabine J.; Warner, Thomas F.; Meredith, Melissa; Shenker, Yoram

doi:10.1530/eje.0.1370079

Cited by 11 publications

(8 citation statements)

References 27 publications

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“…However, careful analysis of the 20 cases of ACTH-secreting carcinoids reported with sufficient details in English medical literature reveals that: 1) 12 were also evident using CT or MRI scanning (11, 12, 16, 18, 21-25); 2) 3 were not visualized initially because of the use of standard-cut CT, and these tumors were visualized when high-resolution CT of the chest was performed thereafter (17,19,20); and 3) SRS was useful only in confirming a suspected lesion seen with conventional imaging in 3 cases (13,15,26). Finally, SRS was of undeniable help and more sensitive than conventional imaging in only 2 (10%) cases (14,24). Conversely, and despite the bias of publication rules that favors the report of positive isolated clinical cases, patients in whom SRS was negative together with negative (11,27,28) or positive (11,29) conventional imaging have also been described.…”

Section: Discussionmentioning

confidence: 99%

“…ϩ ϩ De Herder (11) Pancreatic carcinoid (?) ϩ ϩ Iser (12) Bronchial carcinoid (20) ϩ ϩ Phlipponneau (13) Bronchial carcinoid (7) Ϯ ϩ Weiss (14) Bronchial carcinoid (10) Ϫ ϩ Lefebvre (15) Bronchial carcinoid (9) Ϯ ϩ Olario (16) Bronchial carcinoid (20) ϩ ϩ Tsagarakis (17) Bronchial carcinoid (6) ϩ ϩ Christin-Maitre (18) Bronchial carcinoid (10) ϩ ϩ Kristofferson (19) Bronchial carcinoid (20) ϩ ϩ Briganti (20) Bronchial carcinoid (20) ϩ ϩ Carreta (21) Bronchial carcinoid (10) ϩ ϩ Fernandez (22) Bronchial carcinoid (20) ϩ ϩ Mansi (23) Bronchial carcinoid (18) ϩ ϩ Segu (24) Midgut carcinoid (?) Ϫ ϩ Schrager (25) Bronchial carcinoid (?)…”

Section: Subjects and Methods Patientsmentioning

confidence: 99%

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Usefulness of Somatostatin Receptor Scintigraphy in Patients with Occult Ectopic Adrenocorticotropin Syndrome

Tabarin¹

1999

Journal of Clinical Endocrinology & Metabolism

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SRIF receptor scintigraphy (SRS) has been proposed for the localization of ectopic ACTH-secreting tumors responsible for Cushing's syndrome. However, in most cases reported, the tumors were also visible using conventional imaging. Therefore, the usefulness of SRS in localizing truly occult ectopic ACTH-secreting tumors remains unknown. We report the results of SRS in 12 patients with ectopic ACTH syndrome (EAS) and in whom the source of ACTH was occult at presentation despite carefully performed conventional imaging. The diagnosis of EAS was made by identification of an ACTH-secreting tumor during follow-up in 5 patients or given a pituitary-toperipheral ACTH ratio of 1.9 or less during petrosal sinus sampling combined with CRH injection and a negative pituitary magnetic resonance imaging (MRI). Whole-body planar SRS, using 111 In-pentetreotide, was performed 19 times in the 12 patients during initial workup and/or follow-up. Axial tomography imaging (single-photon emission-computed tomography) was performed in 7 of these. Conventional imaging was performed within a month of SRS, allowing comparison of the two approaches for the localization of the ACTHsecreting tumors. In addition, the response of plasma cortisol, after a single injection of 200 g octreotide, was studied in 6 patients.Five patients had negative SRS and conventional imaging studies. The source of ACTH secretion remains occult despite 10 -55 months of follow-up in four of these, whereas a 2-cm ileal carcinoid tumor, with liver micrometastases, was found at laparotomy in one patient, 14 months after presentation.SRS was positive in 4 of 12 patients. It was false-positive in 1 patient with follicular thyroid adenoma. Nineteen months after presentation, SRS identified liver metastasis that was also visible using MRI in one patient, but the primary tumor remains occult. SRS identified a 10-mm pancreatic tumor that became detectable, using computed tomography (CT) scanning 9 months later, in 1 patient; and 2 mediastinal lymph nodes of 10 mm, previously ignored by MRI, in another patient, whereas no tumor was detectable within the parenchymal lung. SRS had little influence on therapeutic options in these 2 patients, in whom no final diagnosis could be made. Repetition of SRS during the follow-up of patients with previously negative scintiscans was useless.Conventional imaging was positive in 6 of 12 patients. In the 2 patients with pancreatic tumor and isolated mediastinal lymph nodes, conventional imaging studies were interpreted as positive only after the results of SRS. One patient had liver metastasis that was also visible using SRS. Thin-section CT scanning visualized ACTHsecreting bronchial tumors and metastatic mediastinal lymph nodes of 10 -15 mm in diameter in 3 patients after 14 -72 months of followup, whereas SRS was negative.There was no evident relationship between the endocrine status (hyper-or eucortisolism) and the results of SRS. The in vivo response of plasma cortisol to octreotide correlated to the results of SRS in 4 of 6 c...

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Section: Discussionmentioning

confidence: 99%

Section: Subjects and Methods Patientsmentioning

confidence: 99%

Usefulness of Somatostatin Receptor Scintigraphy in Patients with Occult Ectopic Adrenocorticotropin Syndrome

Tabarin¹

1999

Journal of Clinical Endocrinology & Metabolism

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“…Most commonly the ectopic ACTH syndrome is caused by occult carcinoid tumors of the bronchopulmonary system, thy- (Bankole et al, 1980;Anthoney et al, 1995;Dobnic et al, 1996;Hashi et al, 1996;Onishii et al, 1996;Orbetzova et al, 1997;Segu et al, 1997;Eggenberger et al, 1999;Valo et al, 2000;Uecker and Janzow, 2005). Cushing ' s syndrome caused by ileal neuroendocrine tumors has only been reported in three patients, one diagnosed at autopsy (Dombrovskaia and Romenskaia, 1977;Segu et al, 1997;Valo et al, 2000). Except for the autopsy report the time from detecting Cushing ' s syndrome until localization of the primary tumor was 2 months (reported by Segu and colleagues) and 18 months (reported by Valo and colleagues).…”

Section: Discussionmentioning

confidence: 99%

“…Neuroendocrine tumors within the ileum causing Cushing ' s syndrome by ectopically producing ACTH are extremely rare. Only three cases have been reported and one of those was diagnosed at autopsy (Dombrovskaia and Romenskaia, 1977;Segu et al, 1997;Valo et al, 2000).…”

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confidence: 99%

Ectopic Cushing's Syndrome Caused by a Well Differentiated ACTH-Secreting Neuroendocrine Carcinoma of the Ileum

Singer

Werner²,

Koch³

et al. 2010

Exp Clin Endocrinol Diabetes

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Cushing's syndrome can be caused by adrenocorticotropic hormone-secreting solid tumors. We report a rare case of an ileal endocrine carcinoma that produced ACTH and induced hypercortisolism. A now 47-year-old man presented at age 41 with weight gain, tremor, perspiration, and general fatigue. Laboratory testing showed hypercortisolism and diabetes mellitus. Further examinations revealed ectopic Cushing's syndrome. The search for the primary tumor was difficult. The patient underwent subtotal thyroidectomy and surgical removal of a pituitary lesion. After resection of an ACTH-producing metastasis of the mesentery, temporary remission of Cushing's syndrome ensued. At the age 45 the primary tumor was detected in the ileum by Ga-68 DOTATOC-PET scan and explorative laparotomy. After surgical removal of this well differentiated neuroendocrine carcinoma the patient significantly improved clinically. He experienced better blood pressure and remission of his diabetes mellitus in addition to increased muscular strength. Endocrine laboratory testing at follow-up examinations confirmed remission of hypercortisolism and diabetes mellitus. A Ga-68 DOTATOC PET scan and a 1 mg dexamethasone suppression test 5 months after surgery showed normal results. Ectopic ACTH secretion within the small bowel is very rare. This case underscores the difficulty in locating the source of ectopic ACTH secretion and suggests using small bowel barium study, tubus endoscopy or video endoscopy for preoperative localization if the small bowel is suspected as tumor source.

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“…Finally, in 12-19% of cases the source of ACTH secretion could not be identified despite repeated studies and long-term follow-up [2,4]. The tumors most often implicated in occult ectopic ACTH secretion are the bronchial carcinoid, thymic carcinoid, and pancreatic islet tumors [6].…”

Section: Introductionmentioning

confidence: 99%

Carcinoid tumor of the thymus associated with Cushing′s syndrome and dysgeusia: case report and review of the literature

Claret

Chillarón

Flores

et al. 2009

Endocr

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A 30-year-old man was hospitalized with edema, polyuria, and abnormalities in taste. ACTH and cortisol levels at admission were markedly elevated, even after attempted suppression with 8 mg dexamethasone. A thoracic-abdominal CT revealed an anterior mediastinal lesion and hyperplasia of both adrenal glands. After excision of the mediastinal mass, which confirmed the presence of a carcinoid thymic tumor, the patient became totally asymptomatic, with normal ACTH and cortisol levels. A carcinoid thymic tumor has a poor prognosis, especially when it is associated with Cushing's syndrome. Most patients will present recidivism or metastasis within 5 years after surgery. However, the low number of cases available for analysis makes it difficult to establish optimum therapeutic approaches.

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Use of In-111 pentetreotide scintigraphy in the diagnosis of a midgut carcinoid causing Cushing's syndrome

Cited by 11 publications

References 27 publications

Usefulness of Somatostatin Receptor Scintigraphy in Patients with Occult Ectopic Adrenocorticotropin Syndrome

Usefulness of Somatostatin Receptor Scintigraphy in Patients with Occult Ectopic Adrenocorticotropin Syndrome

Ectopic Cushing's Syndrome Caused by a Well Differentiated ACTH-Secreting Neuroendocrine Carcinoma of the Ileum

Carcinoid tumor of the thymus associated with Cushing′s syndrome and dysgeusia: case report and review of the literature

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