Pitfall of Petrosal Sinus Sampling in a Cushing's Syndrome Secondary to Ectopic Adrenocorticotropin-Corticotropin Releasing Hormone (ACTH-CRH) Secretion

Young, Jacques

doi:10.1210/jc.83.2.305

Cited by 38 publications

(25 citation statements)

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“…Some tumors co-secrete CRH which thereby could counteract a suppression of normal pituitary corticotrophs during hypercortisolism (36)(37)(38)(39)(40). In these reports, CRH levels in peripheral blood were found to be clearly elevated in two of the patients (38,40) and within the normal range in one case (38). In our patient, CRH was below the detection limit in plasma at the time of BIPSS.…”

Section: Discussionmentioning

confidence: 98%

Dual bronchial carcinoids and Cushing's syndrome with a paradoxical response to dexamethasone and a false positive outcome of inferior petrosal sinus sampling

Burman¹,

Lethagen

Ivancev³

et al. 2008

European Journal of Endocrinology

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Context: Establishing the cause of Cushing's syndrome (CS) can be a considerable challenge, in particular in ectopic adrenocorticotropic hormone (ACTH) syndrome, and often requires a combination of biochemical tests and imaging procedures. Subject: A 27-year-old man presented with signs of CS. P-ACTH levels were three times above the upper limit of normal (ULN) and free urinary cortisol around 2000 nmol/24 h. The work-up showed remarkable results. Results: A 2-day low-dose dexamethasone suppression test demonstrated paradoxical increases in cortisol. Sampling from the bilateral inferior petrosal sinus sampling (BIPSS) showed a central to peripheral ACTH ratio of 4.7 after corticotrophin-releasing hormone (CRH) stimulation, i.e. indicated pituitary disease, but magnetic resonance imaging of the pituitary was normal. Computed tomography (CT) scan of the lungs showed two oval-shaped masses, 1.3!1.8 and 1.3!2 cm, in the middle lobe. Both were positive at somatostatin receptor scintigraphy, compatible with tumors or inflammatory lesions. Subsequently, 11 C-5-hydroxytryptophan-PET showed distinct uptake in the tumors but not elsewhere. Two carcinoids situated 3 cm apart, both staining for ACTH, were removed at surgery. Conclusion: This unique case with dual bronchial carcinoids inducing hypercortisolism illustrates the problems with identifying the source of ACTH in CS. Possibly, an abnormal regulation of ACTH production in response to dexamethasone, or steroid-induced tumor necrosis, explains the paradoxical outcome at dexamethasone suppression, and the false positive result at BIPSS reflects an unusual sensitivity of the pituitary corticotrophs to CRH in this patient. The work-up illustrates the great value of 11 C-5-hydroxytryptophan-PET as a diagnostic procedure when other investigations have produced ambiguous results. European Journal of Endocrinology 159 483-488

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Section: Discussionmentioning

confidence: 98%

Dual bronchial carcinoids and Cushing's syndrome with a paradoxical response to dexamethasone and a false positive outcome of inferior petrosal sinus sampling

Burman¹,

Lethagen

Ivancev³

et al. 2008

European Journal of Endocrinology

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“…A scattering of cases have been reported in major case series (Table 1) as well as in individual case reports. 9,30,42,58,62,69 Overall, although the paucity of reported cases allows only an approximate calculation, the specificity of IPSS may be estimated at 90%-95%. In a number of these cases, patients were subjected to transsphenoidal surgery, but the postoperative course suggested an ectopic source, and the causative lesion-in some cases already evident prior to pituitary surgery but deemed incidental at the time-was subsequently removed.…”

Section: Evidence On the Use Of Petrosal Sinus Samplingmentioning

confidence: 99%

“…58,62,69 Lastly, IPSS has been used in patients with ACTHdependent Cushing's syndrome who did not present a clear pituitary lesion, even though testing was indicative of a pituitary lesion. This approach is burdened by the low prevalence of visible pituitary tumors in patients with Cushing's disease; indeed, even the most sensitive pituitary imaging techniques proved negative in 20%-50% of patients in whom an ACTH-secreting adenoma was confirmed by surgery.…”

Section: Rationale For the Use Of Petrosal Sinus Samplingmentioning

confidence: 99%

The role of inferior petrosal sinus sampling in ACTH-dependent Cushing's syndrome: review and joint opinion statement by members of the Italian Society for Endocrinology, Italian Society for Neurosurgery, and Italian Society for Neuroradiology

Giraldi

Cavallo

Tortora

et al. 2015

FOC

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In the management of adrenocorticotropic hormone (ACTH)–dependent Cushing's syndrome, inferior petrosal sinus sampling (IPSS) provides information for the endocrinologist, the neurosurgeon, and the neuroradiologist. To the endocrinologist who performs the etiological diagnosis, results of IPSS confirm or exclude the diagnosis of Cushing's disease with 80%–100% sensitivity and over 95% specificity. Baseline central-peripheral gradients have suboptimal accuracy, and stimulation with corticotropin-releasing hormone (CRH), possibly desmopressin, has to be performed. The rationale for the use of IPSS in this context depends on other diagnostic means, taking availability of CRH and reliability of dynamic testing and pituitary imaging into account. As regards the other specialists, the neuroradiologist may collate results of IPSS with findings at imaging, while IPSS may prove useful to the neurosurgeon to chart a surgical course. The present review illustrates the current standpoint of these 3 specialists on the role of IPSS.

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“…It has been suggested that no single endocrine test and/or imaging procedure are accurate enough to diagnose and localise ectopic ACTH/CRH-producing bronchial carcinoids, particularly as false positive IPSS results may occasionally be obtained, albeit very rarely (Young et al 1998, de Herder & Lamberts 1999, Baudin et al 2001, Loli et al 2003. In such cases, scintigraphy with 111 In-octreotide, particularly after correction of hypercortisolaemia, and PET using several novel tracers can be used to reveal confounding cases eluding localisation (de Herder et al 1994, Tsagarakis et al 2003, Kaltsas et al 2004b, Markou et al 2005.…”

Section: Humoral Pnss In Net Cushing's Syndromementioning

confidence: 99%

Paraneoplastic syndromes secondary to neuroendocrine tumours

Kaltsas

Androulakis²,

Herder³

et al. 2010

Endocrine-Related Cancer

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Neuroendocrine tumours may be either benign or malignant tumours, and have the ability to synthesise and secrete biologically active substances characteristic of the cell of origin that can cause distinct clinical syndromes. The term 'paraneoplastic syndromes' (PNSs) is used to denote syndromes secondary to substances secreted from tumours not related to their specific organ or tissue of origin and/or production of autoantibodies against tumour cells; such syndromes are mainly associated with hormonal and neurological symptoms. Appreciation of the presence of such syndromes is important as clinical presentation, if not identified, may delay the diagnosis of the underlying neoplasia. Conversely, early recognition can allow for more rapid diagnosis, particularly as the coexistence of a neoplasm with a clinical or biochemical marker offers an additional determinant of tumour status/progression. PNSs can complicate the patient's clinical course, response to treatment, impact prognosis and even be confused as metastatic spread. Their diagnosis involves a multidisciplinary approach, and detailed endocrinological, neurological, radiological and histological studies are required. Correct diagnosis is essential as the treatment of choice will be different for each disorder, particularly in the case of malignant tumours; it is therefore important to develop appropriate means to correctly identify and localise these tumours. Clinical awareness and the incorporation into clinical practise of 111 In-octreotide scintigraphy, chromogranin A and other evolving biochemical marker measurement techniques have substantially contributed to the identification of patients harbouring such syndromes. Diseasespecific medical therapies are mandatory in order to prevent recurrence and/or further tumour growth. Owing to their rarity, central registration of these syndromes is very helpful in order to be able to provide evidence-based diagnostic and therapeutic approaches.

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Pitfall of Petrosal Sinus Sampling in a Cushing's Syndrome Secondary to Ectopic Adrenocorticotropin-Corticotropin Releasing Hormone (ACTH-CRH) Secretion

Cited by 38 publications

References 0 publications

Dual bronchial carcinoids and Cushing's syndrome with a paradoxical response to dexamethasone and a false positive outcome of inferior petrosal sinus sampling

Dual bronchial carcinoids and Cushing's syndrome with a paradoxical response to dexamethasone and a false positive outcome of inferior petrosal sinus sampling

The role of inferior petrosal sinus sampling in ACTH-dependent Cushing's syndrome: review and joint opinion statement by members of the Italian Society for Endocrinology, Italian Society for Neurosurgery, and Italian Society for Neuroradiology

Paraneoplastic syndromes secondary to neuroendocrine tumours

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