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Eligibility
A Case Report abstract should provide valuable teaching points or learning lessons pertinent to the field of Endocrinology. Case Report abstracts are typically accepted as poster presentations (sometimes as oral presentations). Case Report abstracts that do not provide meaningful teaching points will not be accepted.
Title
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Case Report abstracts must be submitted in the following structured format:
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Abbreviations that are familiar to endocrinologists (eg. PCR, GHRH, TSH, etc.) may be used without explanation.
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Maarten O van Aken, Richard A Feelders, Wouter W de Herder, Alberto M Pereira, Neveen A Hamdy, Steven W Lamberts, Aart Jan van der Lely, Leo J Hofland, Johannes A Romijn
Intern Med, Sect Endocrinol, Erasmus Med Ctr, Rotterdam, Netherlands; Endocrinol and Metab, Leiden Univ Med Ctr, Leiden, Netherlands
Background: In several animal species, histamine (H) has been shown to stimulate adrenal steroid secretion. In humans, however, this has not been reported yet.
Clinical Case: A 51‐year women showed typical symptoms of Cushing syndrome (CS). Initial tests were consistent with ACTH‐independent CS: elevated 24 hr urinary cortisol (449 and 517 nmol/24 hr, n<220 nmol/24 hr), abnormal
1 mg dexamethason overnight test (cortisol after 1 mg dex 620 nmol/l, n<50 nmol/l), elevated midnight serum cortisol (670 nmol/l, n<220 nmol/l), ACTH‐concentrations below detection (< 4 ng/l). Abdominal CT‐scan showed bilateral macro- nodular adrenal hyperplasia (diameter adrenal glands right 3 cm, left 2 cm). A screening protocol, as proposed by Lacroix et al. (1), revealed no change in cortisol secretion in response to LHRH, TRH, food, posture, etoclopramide, or cisapride. Unexpectedly, an increased 24 hr urinary N‐Methyl‐Histamine (N‐M‐H) excretion was found (2623 and 2720 μmol/mol creat, n < 200 μmol/mol creat). Bone marrow biopsy did not show mastocytosis. Evaluating a possible link between H and cortisol, a single i.v. dose of H1 and H2‐antagonists (clemastine 4 mg and ranitidine 300 mg) did not affect serum cortisol concentrations. The patient was treated by laparoscopic biadrenalectomy. Histological examination showed macronodular hyperplasia of the adrenal cortex. Of note, 24 hr urinary N‐M‐H fell to near normal level (370 μmol/mol creat), suggesting previous H‐excess from adrenal origin. In vitro studies: Directly after aparoscopic removal, adrenal cortical tissue was processed to cell suspensions for further testing. H (10‐7 M) increased cortisol production (131% of contr), similar to the effect of ACTH (615 pg/ml) (146% of contr). Ranitidine or clemastine completely aborted the H‐stimulated cortisol production. In control experiments on hyperplastic adrenal tissue from patients with persistent Cushing disease, H (10‐6 M) increased cortisol secretion in 1 of 6 subjects (189% of contr), with unaltered cortisol secretion in the other 5 subjects. Finally, mRNA from H type 1 receptor and H type 2 receptor was detected both in adrenocortical tissue from the patient and in normal or hyperplastic adrenocortical tissue (n=8), using a semi‐quantitative PCR‐technique.
Conclusion: This is the first case demonstrating the possible role of histamine in ACTH‐independent Cushing syndrome with in vitro proof of histamine‐induced cortisol secretion.
Reference: (1) Lacroix, A.,Ndiaye, N.,Tremblay, J.,Hamet, P. Ectopic and abnormal hormone receptors in adrenal Cushing's syndrome. Endocr. Rev. 2001;22;75‐110.
