Adult combined GH, prolactin, and TSH deficiency associated with circulating PIT-1 antibody in humans.

نویسندگان

  • Masaaki Yamamoto
  • Genzo Iguchi
  • Ryoko Takeno
  • Yasuhiko Okimura
  • Toshiaki Sano
  • Michiko Takahashi
  • Hitoshi Nishizawa
  • Anastasia Evi Handayaningshi
  • Hidenori Fukuoka
  • Maya Tobita
  • Takatoshi Saitoh
  • Katsuyoshi Tojo
  • Atsuko Mokubo
  • Akio Morinobu
  • Keiji Iida
  • Hidesuke Kaji
  • Susumu Seino
  • Kazuo Chihara
  • Yutaka Takahashi
چکیده

The pituitary-specific transcriptional factor-1 (PIT-1, also known as POU1F1), is an essential factor for multiple hormone-secreting cell types. A genetic defect in the PIT-1 gene results in congenital growth hormone (GH), prolactin (PRL), and thyroid-stimulating hormone (TSH) deficiency. Here, we investigated 3 cases of adult-onset combined GH, PRL, and TSH deficiencies and found that the endocrinological phenotype in each was linked to autoimmunity directed against the PIT-1 protein. We detected anti-PIT-1 antibody along with various autoantibodies in the patients' sera. An ELISA-based screening revealed that this antibody was highly specific to the disease and absent in control subjects. Immunohistochemical analysis revealed that PIT-1-, GH-, PRL-, and TSH-positive cells were absent in the pituitary of patient 2, who also had a range of autoimmune endocrinopathies. These clinical manifestations were compatible with the definition of autoimmune polyendocrine syndrome (APS). However, the main manifestations of APS-I--hypoparathyroidism and Candida infection--were not observed and the pituitary abnormalities were obviously different from the hypophysitis associated with APS. These data suggest that these patients define a unique "anti-PIT-1 antibody syndrome," related to APS.

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عنوان ژورنال:
  • The Journal of clinical investigation

دوره 121 1  شماره 

صفحات  -

تاریخ انتشار 2011