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      Successful Therapy Using Pasireotide Long-acting Release for Cushing's Disease Merged with Biochemical Acromegaly

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          Abstract

          It is quite rare that Cushing's disease shows acromegaly, and no pharmacotherapy has yet been discussed. A 21-year-old woman was diagnosed with Cushing's disease and underwent trans-sphenoidal surgery. Five years later, she was diagnosed with recurrent Cushing's disease and biochemical acromegaly because of elevated levels of serum growth hormone (GH), plasma insulin-like growth factor-1, plasma adrenocorticotropic hormone (ACTH), and the 24-hour urinary excretion of free cortisol. After treatment initiation with pasireotide-long-acting release (LAR), both the ACTH and GH declined. Our case is the first to show the efficacy of pasireotide-LAR in controlling both Cushing's disease and acromegaly.

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          Pasireotide versus continued treatment with octreotide or lanreotide in patients with inadequately controlled acromegaly (PAOLA): a randomised, phase 3 trial.

          Mônica Gadelha, Marcello D. Bronstein, Thierry Brue … (2014)
          Many patients with acromegaly do not achieve biochemical control despite receiving high doses of the first-generation somatostatin analogues octreotide or lanreotide. In the PAOLA trial, we aimed to assess the efficacy and safety of two different doses of the somatostatin analogue pasireotide long-acting release compared with active control (octreotide or lanreotide) in patients with inadequately controlled acromegaly.
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            Somatostatin receptor type 2A immunohistochemistry in neuroendocrine tumors: a proposal of scoring system correlated with somatostatin receptor scintigraphy.

            Ida Rapa, M P Brizzi, Carlo Capella … (2007)
            Typing somatostatin receptor expression in neuroendocrine tumors is of relevance to target somatostatin analogue-based diagnostic approach and treatment. The expanding use of immunohistochemistry to detect somatostatin receptors is to date not paralleled by an accurate methodological setting and standardized interpretation of the results. A multicentric study was designed to compare somatostatin receptor immunohistochemical expression with in vivo scintigraphic data and verify its usefulness in the clinical management of neuroendocrine tumors. After methodological setting by testing different somatostatin receptor antibodies, 107 cases of neuroendocrine tumors with available somatostatin receptor scintigraphy data and pathological material were retrospectively analyzed for somatostatin receptor types 2A, 3 and 5 immunohistochemical expression, and compared with scintigraphic images and, whenever available, with the clinical response to somatostatin analogue treatment. Restricting 'positive cases' to the presence of a membrane pattern of staining, an overall somatostatin receptor type 2A immunohistochemistry/somatostatin receptor scintigraphy agreement of 77% (chi2 test P<0.0001) was reached. Lower concordance ratios were detected in preoperative and metastatic tumor samples, possibly as a consequence of somatostatin receptor expression heterogeneity. Pure somatostatin receptor type 2A cytoplasmic staining showed poor correlation with somatostatin receptor scintigraphy (54% concordance rate). The immunohistochemical detection of somatostatin receptor types 3 and 5, which showed almost exclusively a cytoplasmic pattern, did not improve the concordance with scintigraphic data. In a pilot series, somatostatin receptor type 2A immunohistochemistry correlated with clinical response in 75% of cases. In conclusion, we propose a scoring system for somatostatin receptor type 2A immunohistochemistry in neuroendocrine tumors correlated with in vivo data, based on the evidence that only membrane (rather that cytoplasmic) staining should be considered for a reliable, standardized and clinically relevant report.
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              Efficacy and safety of once-monthly pasireotide in Cushing's disease: a 12 month clinical trial.

              Andre Lacroix, Feng Gu, Wilson Gallardo … (2018)
              Cushing's disease is a rare debilitating endocrine disorder for which few prospective interventional studies have been done. We report results of the first phase 3 trial assessing long-acting intramuscular pasireotide in patients with Cushing's disease.
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                Author and article information

                Journal
                Journal ID (nlm-ta): Intern Med
                Journal ID (iso-abbrev): Intern Med
                Title: Internal Medicine
                Publisher: The Japanese Society of Internal Medicine
                ISSN (Print): 0918-2918
                ISSN (Electronic): 1349-7235
                Publication date (Electronic): 9 November 2020
                Publication date (Print): 1 April 2021
                Volume: 60
                Issue: 7
                Pages: 1047-1053
                Affiliations
                [1 ]Department of Endocrinology and Metabolism, Faculty of Medicine, Kagawa University, Japan
                [2 ]Department of Diagnostic Pathology, Faculty of Medicine, Kagawa University, Japan
                [3 ]Department of Pathology, Tokyo Metropolitan Geriatric Hospital and Institute of Gerontology, Japan
                Author notes

                Correspondence to Dr.　Kensaku Fukunaga, fukunaga@ 123456med.kagawa-u.ac.jp

                Article
                DOI: 10.2169/internalmedicine.4663-20
                PMC ID: 8079926
                PubMed ID: 33162471
                SO-VID: 2b5a82c1-8c06-4033-9846-81f6bdb1f18d
                Copyright © Copyright © 2021 by The Japanese Society of Internal Medicine
                License:

                The Internal Medicine is an Open Access journal distributed under the Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International License. To view the details of this license, please visit ( https://creativecommons.org/licenses/by-nc-nd/4.0/).

                History
                Date received : 13 February 2020
                Date accepted : 18 September 2020
                Categories
                Subject: Case Report

                Keywords: acromegaly,cushing's disease,pasireotide long-acting release (lar),somatostatin analog,plurihormonal pituitary adenoma
                Data availability:
                Keywords: acromegaly, cushing's disease, pasireotide long-acting release (lar), somatostatin analog, plurihormonal pituitary adenoma

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