Clinical features of anti-pituitary-specific transcription factor-1 (PIT-1) hypophysitis: a new aspect of paraneoplastic autoimmune condition-Reference-Cited by-同舟云学术

Clinical features of anti-pituitary-specific transcription factor-1 (PIT-1) hypophysitis: a new aspect of paraneoplastic autoimmune condition

Published:2023-12-26 Issue:1 Volume:190 Page:K1-K7
ISSN:0804-4643
Container-title:European Journal of Endocrinology
language:en
Short-container-title:

Author:

Urai Shin¹^ORCID,Iguchi Genzo¹²³^ORCID,Kanie Keitaro¹^ORCID,Bando Hironori⁴^ORCID,Yamamoto Masaaki¹,Oi Yuka¹,Kashitani Yuya⁵,Iida Keiji⁵,Kanzawa Maki⁶^ORCID,Fukuoka Hidenori⁴^ORCID,Takahashi Michiko⁴⁷,Shintani Yasushi⁸^ORCID,Ogawa Wataru¹^ORCID,Takahashi Yutaka¹⁹

Affiliation:

1. Division of Diabetes and Endocrinology, Department of Internal Medicine, Kobe University Graduate School of Medicine , Kobe, Hyogo 650-0017 , Japan

2. Medical Center for Student Health, Kobe University , Kobe, Hyogo 657-8501 , Japan

3. Division of Biosignal Pathophysiology, Kobe University , Kobe, Hyogo 657-8501 , Japan

4. Division of Diabetes and Endocrinology, Department of Internal Medicine, Kobe University Hospital , Kobe, Hyogo 650-0017 , Japan

5. Division of Diabetes and Endocrinology, Hyogo Prefectural Kakogawa Medical Center , Kakogawa, Hyogo 675-8555 , Japan

6. Division of Diagnostic Pathology, Kobe University Graduate School of Medicine , Kobe, Hyogo 650-0017 , Japan

7. Department of Nutrition, Kobe University Hospital , Kobe, Hyogo 650-0017 , Japan

8. Department of General Thoracic Surgery, Osaka University Graduate School of Medicine , Suita, Osaka 565-0871 , Japan

9. Department of Diabetes and Endocrinology, Nara Medical University , Kashihara, Nara 634-8521 , Japan

Abstract

Abstract The pathogenesis of anti-pituitary-specific transcription factor-1 (PIT-1) hypophysitis was gradually revealed as cases emerged. Our comprehensive analysis, including all reported cases, identified a new instance of anti-PIT-1 hypophysitis postimmune checkpoint inhibitor therapy. All 9 patients exhibited extremely low growth hormone (GH), prolactin (PRL), and thyroid-stimulating hormone (TSH) levels; 2 had a slightly atrophic pituitary gland; 4 had thymoma, and 5 had malignant neoplasms of diffuse large B-cell lymphoma (DLBCL) and other origins. Patients with thymoma showed multiple autoimmune diseases. HLA-A*24:02 and/or A*02:06 were present in six and DR53 in 5 cases analyzed. High anti-PIT-1 antibody titers and ectopic PIT-1 expression in the cytosol and nucleus of the tumor tissues were observed in patients with thymoma or DLBCL, whereas it was exclusively observed in the nuclei of a bladder cancer patient. These findings provide new insights into the pathophysiology of paraneoplastic autoimmune hypophysitis.

Funder

Grant-in-Aid for Scientific Research from the Japan Society for the Promotion of Science

Harmonic Ito Foundation

Foundation for Promotion of Cancer Research

Ministry of Health, Labor and Welfare

Publisher

Oxford University Press (OUP)

Subject

Endocrinology,General Medicine,Endocrinology, Diabetes and Metabolism

Link

https://academic.oup.com/ejendo/advance-article-pdf/doi/10.1093/ejendo/lvad179/54869916/lvad179.pdf

Reference31 articles.

1. Dwarf locus mutants lacking three pituitary cell types result from mutations in the POU-domain gene pit-1;Li;Nature,1990

2. Hypophysitis, the growing spectrum of a rare pituitary disease;Langlois;J Clin Endocrinol Metab,2022

3. Executive summary of the consensus document on hypophysitis of the neuroendocrinology area of knowledge of the Spanish society of endocrinology and nutrition;Iglesias;Endocrinol Diabetes Nutr (Engl Ed),2023

4. Adult combined GH, prolactin, and TSH deficiency associated with circulating PIT-1 antibody in humans;Yamamoto;J Clin Invest,2011

5. A novel thymoma-associated autoimmune disease: anti-PIT-1 antibody syndrome;Bando;Sci Rep,2017