Title :

Identification of Clinical Factors Related to Antibody-Mediated Immune Response to the Subfornical Organ.

Author(s) :

Nakamura-Utsunomiya, Akari [Auteur]
Goda, Satoshi [Auteur]
Hayakawa, Seiichi [Auteur]
Sonoko, Sakata [Auteur]
Hoorn, Ewout J. [Auteur]
Blanchard, Anne [Auteur]
Saito-Hakoda, Akiko [Auteur]
Kakimoto, Haruna [Auteur]
Hachiya, Rumi [Auteur]
Kamimura, Miki [Auteur]
Kawakita, Rie [Auteur]
Higuchi, Shinji [Auteur]
Fujimaru, Rika [Auteur]
Shirai, Yoko [Auteur]
Miyaoka, Daichi [Auteur]
Nagata, Yuki [Auteur]
Kishi, Yutaro [Auteur]
Wada, Aya [Auteur]
Mitsuboshi, Akari [Auteur]
Ozaki, Kayo [Auteur]
Komatsu, Nagisa [Auteur]
Niizuma, Hidetaka [Auteur]
Kanno, Junko [Auteur]
Fujiwara, Ikuma [Auteur]
Hasegawa, Yukihiro [Auteur]
Yorifuji, Tohru [Auteur]
Brickman, Wendy [Auteur]
Vantyghem, Marie-Christine [Auteur]
Recherche translationnelle sur le diabète (RTD) - U1190
Yamaguchi, Kei [Auteur]
Goshima, Naoki [Auteur]
Hiyama, Takeshi Y. [Auteur]

Journal title :

Clinical Endocrinology

Abbreviated title :

Clin Endocrinol (Oxf)

Volume number :

97

Pages :

72-80

Publication date :

2022-07

ISSN :

1365-2265

English keyword(s) :

adipsic hypernatremia
dysnatremia
hypothalamus dysfunction
Na-x channel
ROHHAD syndrome
sensory circumventricular organ
subfornical organ

HAL domain(s) :

Sciences du Vivant [q-bio]

English abstract : [en]

Objective We recently reported cases of adipsic hypernatremia caused by autoantibodies against the subfornical organ in patients with hypothalamic-pituitary lesions. This study aimed to clarify the clinical features of ...
Show more >Objective We recently reported cases of adipsic hypernatremia caused by autoantibodies against the subfornical organ in patients with hypothalamic-pituitary lesions. This study aimed to clarify the clinical features of newly identified patients with adipsic hypernatremia whose sera displayed immunoreactivity to the mouse subfornical organ. Design Observational cohort study of patients diagnosed with adipsic hypernatremia in Japan, United States, and Europe. Methods The study included 22 patients with adipsic hypernatremia but without overt structural changes in the hypothalamic-pituitary region and congenital disease. Antibody response to the mouse subfornical organ was determined using immunohistochemistry. The clinical characteristics were compared between the patients with positive and negative antibody responses. Results Antibody response to the mouse subfornical organ was detected in the sera of 16 patients (72.7%, female/male ratio, 1:1, 12 pediatric and 4 adult patients). The prolactin levels at the time of diagnosis were significantly higher in patients with positive subfornical organ (SFO) immunoreactivity than in those with negative SFO immunoreactivity (58.9 ± 33.5 vs. 22.9 ± 13.9 ng/ml, p < .05). Hypothalamic disorders were found in 37.5% of the patients with positive SFO immunoreactivity. Moreover, six patients were diagnosed with rapid-onset obesity with hypothalamic dysfunction, hypoventilation, and autonomic dysregulation/neural tumor syndrome after the diagnosis of adipsic hypernatremia. Plasma renin activity levels were significantly higher in patients with serum immunoreactivity to the Nax channel. Conclusions The patients with serum immunoreactivity to the SFO had higher prolactin levels and hypothalamic disorders compared to those without the immunoreactivity. The clinical characteristics of patients with serum immunoreactivity to the subfornical organ included higher prolactin levels and hypothalamic disorders, which were frequently associated with central hypothyroidism and the presence of retroperitoneal tumors.Show less >

Language :

Anglais

Audience :

Internationale

Popular science :

Non

Administrative institution(s) :

Université de Lille
Inserm
CHU Lille

Collections :

• Recherche translationnelle sur le diabète (RTD) - U1190

Submission date :

2024-01-19T22:39:53Z
2024-10-29T09:09:21Z
2024-10-29T09:29:18Z