Incidentally detected sellar spine in a patient with Cushing’s syndrome: a case report

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Abstract

Sellar spine, a bony spur extending anteriorly from the dorsum sellae, is a very rare anatomical variant. Several hypotheses regarding its etiology have been proposed, including the strongly supported theory of a cephalic ossified notochordal remnant. Sellar spine is usually detected incidentally in patients who have no definite symptoms, but several cases have reportedly accompanied endocrinopathies such as precocious puberty, hypopituitarism, or galactorrhea/oligomenorrhea. However, no published reports have described sellar spine in a patient with Cushing’s syndrome. We herein report a case of sellar spine detected during the evaluation of Cushing’s disease in a 29-year-old woman who underwent inferior petrosal sinus sampling, computed tomography, magnetic resonance imaging, and exploratory surgery. There was no evidence of a pituitary microadenoma, but a sellar spine was present in the operative field. Thus, the sellar spine might have caused Cushing’s syndrome in this case, although the exact mechanism is unknown.

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Bilateral inferior petrosal sinus sampling

Benedetta Zampetti, Erika Grossrubatscher, Paolo Dalino Ciaramella … (2016)

Simultaneous bilateral inferior petrosal sinus sampling (BIPSS) plays a crucial role in the diagnostic work-up of Cushing’s syndrome. It is the most accurate procedure in the differential diagnosis of hypercortisolism of pituitary or ectopic origin, as compared with clinical, biochemical and imaging analyses, with a sensitivity and specificity of 88–100% and 67–100%, respectively. In the setting of hypercortisolemia, ACTH levels obtained from venous drainage of the pituitary are expected to be higher than the levels of peripheral blood, thus suggesting pituitary ACTH excess as the cause of hypercortisolism. Direct stimulation of the pituitary corticotroph with corticotrophin-releasing hormone enhances the sensitivity of the procedure. The procedure must be undertaken in the presence of hypercortisolemia, which suppresses both the basal and stimulated secretory activity of normal corticotrophic cells: ACTH measured in the sinus is, therefore, the result of the secretory activity of the tumor tissue. The poor accuracy in lateralization of BIPSS (positive predictive value of 50–70%) makes interpetrosal ACTH gradient alone not sufficient for the localization of the tumor. An accurate exploration of the gland is recommended if a tumor is not found in the predicted area. Despite the fact that BIPSS is an invasive procedure, the occurrence of adverse events is extremely rare, particularly if it is performed by experienced operators in referral centres.

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Desmopressin test during petrosal sinus sampling: a valuable tool to discriminate pituitary or ectopic ACTH-dependent Cushing's syndrome.

F Castinetti, I Morange, Thierry Brue … (2007)

Corticotropin-releasing hormone (CRH)-stimulated petrosal sinus sampling is currently the gold standard method for the differential diagnosis between pituitary and ectopic ACTH-dependent Cushing's syndrome. Our objective was to determine sensitivity and specificity of desmopressin test during petrosal sinus sampling. Forty-three patients had petrosal sinus sampling because of the lack of visible adenoma on magnetic resonance imaging (MRI) and/or because of discordant cortisol response to high-dose dexamethasone suppression test. ACTH sampling was performed in an antecubital vein, right and left petrosal sinuses, then at each location 5 and 10 min after injection of desmopressin. Diagnosis was based on the ACTH ratio between petrosal sinus and humeral vein ACTH after desmopressin test. Diagnosis was confirmed after surgery. A receiver operating characteristics curve was used to determine optimal sensitivity and specificity. Thirty-six patients had Cushing's disease (CD) and seven had ectopic ACTH secretion. A ratio > 2 after desmopressin was found in 35 of the 36 cases of CD (sensitivity: 95%). A ratio < or = 2 was found in the seven patients with ectopic ACTH secretion (specificity: 100%). Sinus sampling was ineffective in determining the left or right localization of the adenoma (sensitivity = 50%). No major adverse effects were observed during or after the procedure. Desmopressin test during petrosal sinus sampling is a safe and effective diagnostic procedure in ACTH-dependent Cushing's syndrome. It thus represents a valuable alternative to CRH.

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Assay-Specific Spurious ACTH Results Lead to Misdiagnosis, Unnecessary Testing, and Surgical Misadventure—A Case Series

Loren Greene, Eliza B. Geer, Gabrielle Page-Wilson … (2019)

Abstract The proper clinical evaluation of pituitary and adrenal disorders depends on the accurate measurement of plasma ACTH. The modern two-site sandwich ACTH immunoassay is a great improvement compared with older methods but still has the potential for interferences such as heterophile antibodies and pro-opiomelanocortin (POMC) and ACTH fragments. We report the cases of five patients in whom the diagnosis or differential diagnosis of Cushing syndrome was confounded by erroneously elevated results from the Siemens ACTH Immulite assay [ACTH(Immulite)] that were resolved using the Roche Cobas or Tosoh AIA [ACTH(Cobas) and ACTH(AIA), respectively]. In one case, falsely elevated ACTH(Immulite) results owing to interfering antibodies resulted in several invasive differential diagnostic procedures (including inferior petrosal sinus sampling), MRI, and unnecessary pituitary surgery. ACTH(Cobas) measurements were normal, and further studies excluded the diagnosis of Cushing syndrome. In three cases, either Cushing disease or occult ectopic ACTH were suspected owing to elevated ACTH(Immulite) results. However, adrenal (ACTH-independent) Cushing syndrome was established using ACTH(AIA) or ACTH(Cobas) and proved surgically. In one case, ectopic ACTH was suspected owing to elevated ACTH(Immulite) results; however, the ACTH(Cobas) findings led to the diagnosis of alcohol-induced hypercortisolism that resolved with abstinence. We have concluded that ACTH(Immulite) results can be falsely increased and alternate ACTH assays should be used in the diagnosis or differential diagnosis of clinical disorders of the hypothalamic–pituitary–adrenal axis.

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Journal

Journal ID (nlm-ta): J Int Med Res

Journal ID (iso-abbrev): J Int Med Res

Journal ID (publisher-id): IMR

Journal ID (hwp): spimr

Title: The Journal of International Medical Research

Publisher: SAGE Publications (Sage UK: London, England )

ISSN (Print): 0300-0605

ISSN (Electronic): 1473-2300

Publication date (Electronic): 16 July 2020

Publication date Collection: July 2020

Volume: 48

Issue: 7

Electronic Location Identifier: 0300060520940159

Affiliations

[1 ]Department of Radiology, Chonnam National University Hwasun Hospital, Korea

[2 ]Department of Radiology, Chonnam National University Hospital, Korea

[3 ]Department of Radiology, Chonnam National University Medical School, Korea

Author notes

[*]Seul Kee Kim, Chonnam National University Hwasun Hospital, 322 Seoyang-ro, Hwasun-eup, Hwasun-gun, Jeollanam-do 58128, Republic of Korea. Email: kimsk.rad@ 123456gmail.com

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Seul Kee Kim https://orcid.org/0000-0002-1508-5057

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Publisher ID: 10.1177_0300060520940159

DOI: 10.1177/0300060520940159

PMC ID: 7557794

PubMed ID: 32672486

SO-VID: b4671fb1-acd3-4422-9755-452325d337b9

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Creative Commons Non Commercial CC BY-NC: This article is distributed under the terms of the Creative Commons Attribution-NonCommercial 4.0 License ( https://creativecommons.org/licenses/by-nc/4.0/) which permits non-commercial use, reproduction and distribution of the work without further permission provided the original work is attributed as specified on the SAGE and Open Access pages ( https://us.sagepub.com/en-us/nam/open-access-at-sage).

History

Date received : 3 March 2020

Date accepted : 12 June 2020

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Keywords: sellar spine,cushing’s syndrome,inferior petrosal sinus sampling,pituitary adenoma,exploratory surgery,sellar magnetic resonance imaging

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Keywords: sellar spine, cushing’s syndrome, inferior petrosal sinus sampling, pituitary adenoma, exploratory surgery, sellar magnetic resonance imaging

Incidentally detected sellar spine in a patient with Cushing’s syndrome: a case report

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Most cited references 18

Bilateral inferior petrosal sinus sampling

Desmopressin test during petrosal sinus sampling: a valuable tool to discriminate pituitary or ectopic ACTH-dependent Cushing's syndrome.

Assay-Specific Spurious ACTH Results Lead to Misdiagnosis, Unnecessary Testing, and Surgical Misadventure—A Case Series

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