Adrenocorticotropic Hormone-Independent Bilateral Adrenocortical Macronodular Hyperplasia Treated with Mitotane.

Nagai, Masaaki; Narita, Ichiei; Omori, Kentarou; Komura, Satoru; Arakawa, Masaaki

doi:10.2169/internalmedicine.38.969

Cited by 18 publications

(8 citation statements)

References 9 publications

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“…Because it is metabolically activated by an adrenal-specific cytochrome P450 (Jonsson et al, 1994), mitotane has been found to be quite selective in its effects - for example, an earlier study found that mitotane had no effects on testis size or testosterone titers in male house sparrows (Breuner et al, 2000). However, there are documented instances of mitotane treatment causing liver damage (Nagai et al, 1999), which can lead to hepatocyte proliferation (Sell, 2003). Therefore, this change in liver size is potentially a side effect of mitotane treatment rather than a meaningful difference related to decreased stress-induced CORT titers in these animals.…”

Section: Discussionmentioning

confidence: 99%

Experimentally reducing corticosterone mitigates rapid captivity effects on behavior, but not body composition, in a wild bird

Lattin

Pechenenko

Carson

2017

Hormones and Behavior

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Wild animals and captives display physiological and behavioral differences, and it has been hypothesized, but rarely tested, that these differences are caused by sustained elevation of the hormone corticosterone. We used repeated computed tomography (CT) imaging to examine body composition changes in breeding male and female wild house sparrows (Passer domesticus; n=20) in response to two weeks of captivity, and assessed behavioral changes using video recordings. Half of the birds received the drug mitotane, which significantly decreased stress-induced corticosterone titers compared to controls. Based on the CT images, fat volumes increased, and pectoralis muscle density and heart and testes volumes decreased, over the two weeks of captivity in both groups of birds. However, beak-wiping, a behavior that can indicate anxiety and aggression, showed increased occurrence in controls compared to mitotane-treated birds. While our results do not support the hypothesis that these body composition changes were primarily driven by stress-induced corticosterone, our data suggest that experimentally reducing stress-induced corticosterone may mitigate some captivity-induced behavioral changes. Broadly, our results emphasize that researchers should take behavioral and physiological differences between free-living animals and captives into consideration when designing studies and interpreting results. Further, time in captivity should be minimized when birds will be reintroduced back to the wild.

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Section: Discussionmentioning

confidence: 99%

Experimentally reducing corticosterone mitigates rapid captivity effects on behavior, but not body composition, in a wild bird

Lattin

Pechenenko

Carson

2017

Hormones and Behavior

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“…The treatment of choice for AIMAH is total bilateral adrenalectomy (Swain et al, 1998;Doppman et al, 2000), which was successfully performed in three female members of the family reported here. Recent publications indicate that medical treatment of AIMAH may be possible in the future (Nagai et al, 1999;Mircescu et al, 2000).…”

Section: Discussionmentioning

confidence: 99%

Familial ACTH-independent Cushing's syndrome with bilateral macronodular adrenal hyperplasia clinically affecting only female family members

Nies¹,

Bartsch²,

Ehlenz³

et al. 2002

Exp Clin Endocrinol Diabetes

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Primary adrenal hyperplasia, which may occur as a familial disorder, is a rare cause of ACTH-independent Cushing's syndrome. In most of these cases the underlying pathology is primary adrenocortical micronodular dysplasia. Very few cases of familial Cushing's syndrome due to primary macronodular adrenal hyperplasia have been described. We report a family with seven affected family members. The pedigree indicates an autosomal dominantly inherited disorder. Interestingly only female family members developed the clinically apparent syndrome. The only available obligatory male gene carrier failed to adequately suppress his plasma cortisol level on overnight dexamethasone suppression test. His adrenal glands showed nodular enlargement on abdominal computed tomographic imaging. Screening of the MEN 1 gene and genetic analysis of the hot spot regions of the GNAS 1 (codons 201 and 227) and GNAI 2 (codons 179 and 205) genes did not show any mutations in the constitutional DNA or the adrenal tissue DNA of the index patient. In conclusion, this family is the largest kindred reported in the literature with ACTH-independent Cushing's syndrome due to autosomal dominant inherited macronodular adrenocortical hyperplasia. Four currently alive and affected family members in two generations and further careful observation of the yet unaffected members of the third available generation might offer the opportunity to identify the still unknown gene defect in the future.

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“…There has only been one report on the use of trilostane for AIMAH (16). Moreover, there have only been a few reports discussing the treatment of AIMAH with mitotane (17) or metyrapone (18), which are other inhibitors of cortisol synthesis. According to those reports, serum cortisol decreased with treatment, but the follow-up period was only around 10 months and they did not provide data about the size of the adrenal glands.…”

Section: Discussionmentioning

confidence: 99%

Long-term Efficacy of Trilostane for Cushing's Syndrome due to Adrenocorticotropin-Independent Bilateral Macronodular Adrenocortical Hyperplasia

et al. 2011

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A 66-year-old man with Cushing's syndrome due to adrenocorticotropin-independent bilateral macronodular adrenocortical hyperplasia (AIMAH) was treated for 7 years with trilostane, a 3β-hydroxysteroid dehydrogenase inhibitor. Administration of trilostane reduced the serum cortisol level to around the upper limit of normal for 7 years, and symptoms of excessive glucocorticoid production (such as moon face and obesity) were gradually improved. On the other hand, the size of both adrenal glands gradually increased despite treatment with trilostane. Though trilostane therapy could not prevent adrenal growth, it did suppress cortisol secretion over the long term, so it might be a reasonable option for AIMAH in addition to adrenalectomy.

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Adrenocorticotropic Hormone-Independent Bilateral Adrenocortical Macronodular Hyperplasia Treated with Mitotane.

Cited by 18 publications

References 9 publications

Experimentally reducing corticosterone mitigates rapid captivity effects on behavior, but not body composition, in a wild bird

Experimentally reducing corticosterone mitigates rapid captivity effects on behavior, but not body composition, in a wild bird

Familial ACTH-independent Cushing's syndrome with bilateral macronodular adrenal hyperplasia clinically affecting only female family members

Long-term Efficacy of Trilostane for Cushing's Syndrome due to Adrenocorticotropin-Independent Bilateral Macronodular Adrenocortical Hyperplasia

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