{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,20]],"date-time":"2026-07-20T14:18:12Z","timestamp":1784557092081,"version":"3.55.0"},"reference-count":31,"publisher":"Bioscientifica","issue":"1","license":[{"start":{"date-parts":[[2024,1,1]],"date-time":"2024-01-01T00:00:00Z","timestamp":1704067200000},"content-version":"unspecified","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["eo.bioscientifica.com"],"crossmark-restriction":true},"short-container-title":[],"published-print":{"date-parts":[[2024,10,14]]},"abstract":"<jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Metastatic gastroenteropancreatic neuroendocrine neoplasms (GEPNEN) can cause ectopic Cushing\u2019s syndrome (ECS). ECS is highly morbid and medical therapy is complex and can be ineffective. Patients unsuitable for bilateral adrenalectomy (BA) have dismal outcomes. Peptide receptor radionuclide therapy (PRRT) is a rational option for hormone and disease control in ECS caused by NEN with high somatostatin receptor (SSTR) expression.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Aim<\/jats:title>\n                    <jats:p>To describe the characteristics and outcomes of patients with ECS treated with PRRT.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>Single-centre, retrospective analysis of imaging, biochemistry and outcomes of seven consecutive patients with ECS caused by metastatic GEPNEN treated with PRRT from 2006 to 2023.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      Patients were aged 17\u201375 (female\n                      <jats:italic>n<\/jats:italic>\n                      \u2009=\u20096). The primary site was the pancreas (5\/7) and rectum (2\/7). Six patients were on medical therapy for ECS at baseline (one had a previous BA). A median of 34.4 GBq of [\n                      <jats:sup>177<\/jats:sup>\n                      Lu]Lu-DOTA-octreotate was given. [\n                      <jats:sup>90<\/jats:sup>\n                      Y]Y-DOTA-octreotate (one patient) and [\n                      <jats:sup>111<\/jats:sup>\n                      In]In-octreotide (one patient) were also used. Five patients had radiosensitising chemotherapy. Five patients had a flare of ECS within 1 week of PRRT cycle 1 (PRRT-C1). Following PRRT-C1, 5\/7 patients had complete biochemical resolution of ECS at 1.5\u20136 months (four ongoing; one recurred after 12 months and had elective BA at 18 months). Best metabolic response on [\n                      <jats:sup>18<\/jats:sup>\n                      F]F-FDG PET\/CT: Four patients had a complete metabolic response (CMR), and one had a partial metabolic response. PFS was 3\u2013208 months. Two patients progressed at the first follow-up. The longest ECS remission and CMR continues at &gt;17 years.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>PRRT can be effective for ECS caused by metastatic SSTR-positive GEPNEN and should be considered in its treatment algorithm.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1530\/eo-24-0013","type":"journal-article","created":{"date-parts":[[2024,10,14]],"date-time":"2024-10-14T19:26:52Z","timestamp":1728934012000},"update-policy":"https:\/\/doi.org\/10.1530\/crossmarkpolicy-15","source":"Crossref","is-referenced-by-count":3,"title":["Peptide receptor radionuclide therapy for ectopic Cushing\u2019s syndrome caused by metastatic neuroendocrine neoplasms"],"prefix":"10.1530","volume":"4","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-7631-9147","authenticated-orcid":true,"given":"Emma","family":"Boehm","sequence":"first","affiliation":[{"name":"Department of Molecular Imaging and Therapeutic Nuclear Medicine, Peter MacCallum Cancer Centre, Melbourne, Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Terry","family":"Hung","sequence":"additional","affiliation":[{"name":"Department of Molecular Imaging and Therapeutic Nuclear Medicine, Peter MacCallum Cancer Centre, Melbourne, Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tim","family":"Akhurst","sequence":"additional","affiliation":[{"name":"Department of Molecular Imaging and Therapeutic Nuclear Medicine, Peter MacCallum Cancer Centre, Melbourne, Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ramin","family":"Alipour","sequence":"additional","affiliation":[{"name":"Department of Molecular Imaging and Therapeutic Nuclear Medicine, Peter MacCallum Cancer Centre, Melbourne, Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Cherie","family":"Chiang","sequence":"additional","affiliation":[{"name":"Department of Internal Medicine, Endocrinology, Peter MacCallum Cancer Centre, Melbourne, Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rodney J","family":"Hicks","sequence":"additional","affiliation":[{"name":"Department of Medicine, St Vincent\u2019s Medical School, The University of Melbourne, Melbourne, Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michael S","family":"Hofman","sequence":"additional","affiliation":[{"name":"Department of Molecular Imaging and Therapeutic Nuclear Medicine, Peter MacCallum Cancer Centre, Melbourne, Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Aravind S","family":"Ravi Kumar","sequence":"additional","affiliation":[{"name":"Department of Molecular Imaging and Therapeutic Nuclear Medicine, Peter MacCallum Cancer Centre, Melbourne, Australia"},{"name":"Sir Peter MacCallum Department of Oncology, University of Melbourne, Melbourne, Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nirupa","family":"Sachithanandan","sequence":"additional","affiliation":[{"name":"Department of Internal Medicine, Endocrinology, Peter MacCallum Cancer Centre, Melbourne, Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Javad","family":"Saghebi","sequence":"additional","affiliation":[{"name":"Department of Molecular Imaging and Therapeutic Nuclear Medicine, Peter MacCallum Cancer Centre, Melbourne, Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michael","family":"Michael","sequence":"additional","affiliation":[{"name":"Sir Peter MacCallum Department of Oncology, University of Melbourne, Melbourne, Australia"},{"name":"Department of Medical Oncology, Peter MacCallum Cancer Centre, Melbourne"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Grace","family":"Kong","sequence":"additional","affiliation":[{"name":"Department of Molecular Imaging and Therapeutic Nuclear Medicine, Peter MacCallum Cancer Centre, Melbourne, Australia"},{"name":"Sir Peter MacCallum Department of Oncology, University of Melbourne, Melbourne, Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"416","reference":[{"key":"bib1","series-title":"Virchows Archiv","first-page":"507","article-title":"Gene fusions are frequent in ACTH-secreting neuroendocrine neoplasms of the pancreas, but not in their non-pancreatic counterparts","volume":"482","author":"Agaimy","year":"2023","unstructured":"Agaimy AKasajima AStoehr RHaller FSchubart CT\u00f6gel LPfarr Nvon Werder APavel MESessa F, et al. 2023 Gene fusions are frequent in ACTH-secreting neuroendocrine neoplasms of the pancreas, but not in their non-pancreatic counterparts. 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