ArticlePituitary2018
Isolated adrenocorticotropic hormone deficiency as a form of paraneoplastic syndrome.
Article in Pituitary, 2018. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 18 papers, 2 of them syntheses that pooled it.
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Who cites it
18 citing papers in PubMed, 2 syntheses or guidelines pooled it, 33 citations in OpenAlex.
- Idiopathic isolated adrenocorticotropic hormone deficiency: a systematic review of a heterogeneous and underreported disease.Pituitary · 2024Pooled it
- Pooled it
- Utility of gel filtration chromatography in evaluating successful resection of ectopic adrenocorticotropic hormone-producing tumor: a case report and literature review.Endocrine journal · 2025Review
- Hypopituitarism: genetic, developmental, and acquired etiologies with a focus on the emerging concept of autoimmune hypophysitis.Endocrine journal · 2025Review
- Retrospective analysis of clinical characteristics and treatment of patients with immune checkpoint inhibitors-induced adrenal insufficiency.Frontiers in oncology · 2025Article
- Article
- An Update on Advances in Hypopituitarism: Etiology, Diagnosis, and Current Management.Journal of clinical medicine · 2024Review
- Fluctuations in plasma adrenocorticotropic hormone concentration may predict the onset of immune checkpoint inhibitor-related hypophysitis.Journal for immunotherapy of cancer · 2024Article
- Immune checkpoint inhibitor-associated adrenal insufficiency in Chinese cancer patients: a retrospective analysis.Journal of cancer research and clinical oncology · 2023Article
- Clinical characteristics of adrenal insufficiency induced by pembrolizumab in non-small-cell lung cancer.Thoracic cancer · 2023Article
- Isolated anterior pituitary dysfunction in adulthood.Frontiers in endocrinology · 2023Review
- Paraneoplastic isolated adrenocorticotropic hormone deficiency revealed after immune checkpoint inhibitors therapy: new insights into anti-corticotroph antibody.Frontiers in immunology · 2023Article
- Ectopic ACTH Cushing's syndrome caused by a large-cell neuroendocrine lung carcinoma responding to desmopressin.Endocrine oncology (Bristol, England) · 2023Article
- Hypophysitis, the Growing Spectrum of a Rare Pituitary Disease.The Journal of clinical endocrinology and metabolism · 2022Review
- Mechanistic insights into immune checkpoint inhibitor-related hypophysitis: a form of paraneoplastic syndrome.Cancer immunology, immunotherapy : CII · 2021Article
- Two Cases of anti-PIT-1 Hypophysitis Exhibited as a Form of Paraneoplastic Syndrome not Associated With Thymoma.Journal of the Endocrine Society · 2021Article
- Clinical Heterogeneity of Acquired Idiopathic Isolated Adrenocorticotropic Hormone Deficiency.Frontiers in endocrinology · 2021Article
- Anti-PD1 and Anti-PDL1-Induced Hypophysitis: A Cohort Study of 17 Patients with Longitudinal Follow-Up.Journal of clinical medicine · 2020Article
Corrections and comments
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Authors and funding
13 authors at 3 institutions in 1 country.
Funding
Abstract
purposeIn isolated adrenocorticoropic hormone (ACTH) deficiency (IAD), autoimmunity against corticotrophs has been suggested; however, the pathogenesis remains largely unknown. Large cell neuroendocrine carcinoma (LCNEC) of the lung is a pulmonary tumor of high-grade malignant neuroendocrine tumor and it reportedly caused paraneoplastic syndrome by autoimmunity in several cases.
methodsA 42-year-old woman with isolated adrenocorticotropic (ACTH) hormone deficiency (IAD) was diagnosed with large cell neuroendocrine carcinoma (LCNEC) 3 years after being diagnosed with IAD. We hypothesized that the LCNEC played a causal role in the development of IAD as a paraneoplastic syndrome and analyzed the autoimmunity. We also analyzed another case of ectopic ACTH syndrome to prove this hypothesis.
resultsThe LCNEC tissue revealed an ectopic ACTH expression and lymphocyte infiltration. Interestingly, autoantibody against the proopiomelanocortin (POMC) protein was detected in the peripheral blood. Although, patient's serum did not show any effects on cell viability, proliferation, nor pomc expression in a corticotroph cell line, AtT20 cells, patient's lymphocytes in the peripheral blood specifically reacted toward POMC protein, indicating a presence of cytotoxic T lymphocytes (CTLs). In addition, the analysis of another case of ectopic ACTH syndrome showed lymphocyte infiltration not only in the metastatic liver tumors but also in the pituitary. Moreover, most CD8-positive cells resided adjacent to corticotrophs.
conclusionsThese data indicate that the ectopic ACTH expression in the tumor evoked the autoimmunity to corticotrophs and caused IAD as a form of paraneoplastic syndrome.
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