Isolated Adrenocorticotropin Deficiency Associated with an Empty Sella*

Nakagawa, Hiroo; Nagasaka, Akio; Koie, Katsuo; Yuasa, Tomoyo; Sakabe, Yoshiyuki; Kato, Osamu; Suzuki, Takashi; Hattori, Kyoko; Katada, Kazuhiro

doi:10.1210/jcem-55-4-795

Cited by 15 publications

(5 citation statements)

References 9 publications

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“…the etiology of ACTH deficiency in our case is not clear. A brain CT failed to reveal any structural abnormality of the pituitary, including an empty sella, which may be coincidental but often observed in this syndrome [6,7]. Evidence of Hashimoto's thyroiditis, frequently coexis tent and suggestive of an autoimmune process [7], is lacking.…”

Section: Discussionmentioning

confidence: 87%

Normoreninemic Hypoaldosteronism in a Case of Isolated ACTH Deficiency

Miyabo¹,

Minami²,

Inazu³

et al. 1989

Horm Res

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This paper documents the rare and hitherto unreported association between isolated ACTH deficiency and normoreninemic hypoaldosteronism in a 63-year-old woman. Baseline plasma aldosterone and 18-hydroxycorticosterone were extremely low. Both steroids did not respond to exogenous angiotensin II infusion, whereas they were increased in parallel to ACTH stimulation. Thus, acquired dysfunction or congenital dysgenesis of the zona glomeruk > sa was suspected. The upright posture-furosemide test showed a subnormal but definite plasma aldosterone response coupled with a normal increase in plasma renin activity, indicating that there may be a yet unidentified mechanism(s) underlying the postural increase of aldosterone.

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Section: Discussionmentioning

confidence: 87%

Normoreninemic Hypoaldosteronism in a Case of Isolated ACTH Deficiency

Miyabo¹,

Minami²,

Inazu³

et al. 1989

Horm Res

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“…Numerous cases of hyponatremia associated with an empty sella have been described since Nakagawa et al [12] reported the first such patient in 1982. The primary cause of hyponatremia in those cases was a deficiency of ACTH, as in the study of Nakagawa et al, although a patient with diabetes mellitus complicated by SIAD and the empty sella syndrome has also been reported [13].…”

Section: Discussionmentioning

confidence: 99%

Syndrome of Inappropriate Antidiuresis Seen Twice in Eight Years.

et al. 1995

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Abstract.We present a rare case of a 66-year-old woman with the syndrome of inappropriate antidiuresis (SIAD) accompanied by an empty sella whose symptoms were seen twice in the eight years after the administration of non-steroidal anti-inflammatory drugs (NSAID) or prochlorperazine.No diuresis or suppression of the plasma level of vasopressin (AVP) was observed after water loading upon cessation of the causative agents. Suppression of the renin-aldosterone system and a low plasma level of atrial natriuretic peptide (ANP) were observed during natriuresis. The plasma levels of AVP were increased after water loadings.Restriction of water intake ameliorated the symptoms and reduced hyponatremia. These findings suggest that NSAID or prochlorperazine caused overt SIAD twice in eight years. The water loading test itself stimulated the release of AVP and a suppression of the renin-aldosterone system played a more important role in natriuresis than ANP in this case.

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“…However, reports from the recent decades have revealed that primary empty sella could be associated with hypopituitarism. [2][3][4][5][6][7][8] While the etiology of PES is not completely clear, it is currently viewed as being related to the mechanical transmission of the cerebrospinal fluid pressure through an incomplete sellar diaphragm. 1 Recent case reports, however, suggest several other etiologies, one of which involves autoimmunity.…”

Section: Introductionmentioning

confidence: 99%

Primary Empty Sella with Partial Hypopituitarism and Autoimmune Thyroiditis

Silverio-Fernando¹,

Lantion-Ang²,

Jimeno³

2010

Phil. J. Internal Medicine

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Clinical Presentation: A 2 1-y e a r-o l d f e m a l e presented with primary amenorrhea, delayed puberty, continuous linear growth, and torticollis. She noted impaired memory and frequent respiratory infection. P h y s i c a l F i n d i n g s : P a t i e n t p r e s e n t e d w i t h eunuchoidal habitus, tor ticollis, facial asymmetry, micrognathia, and hyperelastosis. Thyroid gland was normal in size. Breast and pubic hair were Tanner Stage 2. Neurologic exam was normal. L a b o r a t o r y Wo r k-u p : L H a n d F S H w e r e l o w w i t h e s t r a d i o l a t p rep u b e r t a l l e v e l , s u g g e s t i n g hypogonadotropic hypogonadism. Prolactin was slightly elevated. Twenty four-hour urinary free cortisol and 8AM serum cortisol were low with low normal plasma ACTH, indicating secondary adrenal insufficiency. TSH was elevated with depressed FT4 and elevated antithyroglobulin, suggesting autoimmune thyroiditis. The following tests were normal: growth hormone, FBS and 2-hour OGTT. Patient had delayed bone age by the Greulich-Pyle method. Bone densitometry revealed osteoporosis. Patient had 46XX karyotype. Pelvic ultrasound revealed infantile uterus. Sellar MRI showed empty sella. Diagnosis: The patient was diagnosed with Primary Empty Sella based on Cranial MRI findings of fluid-filled sella with no history of pituitary adenoma or surgery; with hypogonadotropic hypogonadism, secondar y adrenal insufficiency, and autoimmune thyroiditis. Treatment/Outcome: Sex hormone replacement was star ted, with improvement in breast size and commencement of regular menstrual cycles. Levothyroxine was started, with normalization of the TSH. Prednisone was given at 5mg/day. Alendronate was added for osteoporosis. Significance: PES is rare, with 5.5% occurrence rate. Previous studies have reported preserved pituitary function in most cases. However, par tial or total hypopituitarism has been described in 25% of patients, and hyperprolactinemia in 10%. The coexistence of autoimmune thyroiditis may support the possibility of an autoimmune mechanism behind the development of hypopituitarism in PES. Recommendations: Full endocrine panel should be done in patients presenting with primary empty sella to screen for hormone deficiencies. Presence of autoantibodies should be determined since this syndrome may be linked to autoimmunity.

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Isolated Adrenocorticotropin Deficiency Associated with an Empty Sella*

Cited by 15 publications

References 9 publications

Normoreninemic Hypoaldosteronism in a Case of Isolated ACTH Deficiency

Normoreninemic Hypoaldosteronism in a Case of Isolated ACTH Deficiency

Syndrome of Inappropriate Antidiuresis Seen Twice in Eight Years.

Primary Empty Sella with Partial Hypopituitarism and Autoimmune Thyroiditis

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