ArticleEndocrinology, diabetes & metabolism case reports2023
Ectopic Cushing's syndrome from a corticotropin-releasing hormone-secreting medullary thyroid carcinoma: a rare pitfall of inferior petrosal sinus sampling.
Article in Endocrinology, diabetes & metabolism case reports, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 5 papers, 1 of them a synthesis that pooled it.
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Who cites it
5 citing papers in PubMed, 1 synthesis or guideline pooled it, 6 citations in OpenAlex.
- Management of ectopic Cushing syndrome in medullary thyroid carcinoma: from case reports to systematic literature review.Frontiers in endocrinology · 2026Pooled it
- Ectopic ACTH Production in Medullary Thyroid Carcinoma-A Study of Two Cases.Case reports in endocrinology · 2026Article
- Clinical integration and application of the 2022 WHO pituitary tumor classification.Neuro-oncology advances · 2025Article
- Case Report: Ectopic CRH production by adrenal adenoma as a unique cause of the ACTH-dependent Cushing's syndrome.Frontiers in endocrinology · 2025Article
- Ectopic corticotropin-releasing hormone syndrome caused by rectal large cell neuroendocrine carcinoma: a rare case report.Therapeutic advances in endocrinology and metabolism · 2024Article
Corrections and comments
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Authors and funding
9 authors at 3 institutions in 2 countries.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Summary: This case report describes a rare presentation of ectopic Cushing's syndrome (CS) due to ectopic corticotropin-releasing hormone (CRH) production from a medullary thyroid carcinoma (MTC). The patient, a 69-year-old man, presented with symptoms of muscle weakness, facial plethora, and easy bruising. An inferior petrosal sinus sampling test (IPSS) demonstrated pituitary adrenocorticotrophic hormone (ACTH) secretion, but a whole-body somatostatin receptor scintigraphy (68Ga-DOTATOC PET/CT) revealed enhanced uptake in the right thyroid lobe which, in addition to a grossly elevated serum calcitonin level, was indicative of an MTC. A 18F-DOPA PET/CT scan supported the diagnosis, and histology confirmed the presence of MTC with perinodal growth and regional lymph node metastasis. On immunohistochemical analysis, the tumor cell stained positively for calcitonin and CRH but negatively for ACTH. Distinctly elevated plasma CRH levels were documented. The patient therefore underwent thyroidectomy and bilateral adrenalectomy. This case shows that CS caused by ectopic CRH secretion may masquerade as CS due to a false positive IPSS test. It also highlights the importance of considering rare causes of CS when diagnostic test results are ambiguous. Learning points: Medullary thyroid carcinoma may secrete CRH and cause ectopic CS. Ectopic CRH secretion entails a rare pitfall of inferior petrosal sinus sampling yielding a false positive test. Plasma CRH measurements can be useful in selected cases.
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Registered trials
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