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Patient Demographics

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Rachel Wurth Section on Endocrinology and Genetics, Eunice Kennedy Shriver National Institute of Child Health and Human Development

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Crystal Kamilaris Section on Endocrinology and Genetics, Eunice Kennedy Shriver National Institute of Child Health and Human Development

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Naris Nilubol Surgical Oncology, National Cancer Institute, National Institutes of Health, Bethesda, Maryland, USA

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Samira M Sadowski Surgical Oncology, National Cancer Institute, National Institutes of Health, Bethesda, Maryland, USA

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Annabel Berthon Section on Endocrinology and Genetics, Eunice Kennedy Shriver National Institute of Child Health and Human Development

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Martha M Quezado Laboratory of Pathology Center for Cancer Research, National Cancer Institute, National Institutes of Health, Bethesda, Maryland, USA

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Fabio R Faucz Section on Endocrinology and Genetics, Eunice Kennedy Shriver National Institute of Child Health and Human Development

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Constantine A Stratakis Section on Endocrinology and Genetics, Eunice Kennedy Shriver National Institute of Child Health and Human Development

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Fady Hannah-Shmouni Section on Endocrinology and Genetics, Eunice Kennedy Shriver National Institute of Child Health and Human Development

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Summary

Primary bilateral macronodular adrenal hyperplasia (PBMAH) is a rare cause of ACTH-independent Cushing syndrome (CS). This condition is characterized by glucocorticoid and/or mineralocorticoid excess, and is commonly regulated by aberrant G-protein coupled receptor expression may be subclinical, allowing the disease to progress for years undetected. Inhibin A is a glycoprotein hormone and tumor marker produced by certain endocrine glands including the adrenal cortex, which has not been previously investigated as a potential tumor marker for PBMAH. In the present report, serum inhibin A levels were evaluated in three patients with PBMAH before and after adrenalectomy. In all cases, serum inhibin A was elevated preoperatively and subsequently fell within the normal range after adrenalectomy. Additionally, adrenal tissues stained positive for inhibin A. We conclude that serum inhibin A levels may be a potential tumor marker for PBMAH.

Learning points:

  • PBMAH is a rare cause of CS.

  • PBMAH may have an insidious presentation, allowing the disease to progress for years prior to diagnosis.

  • Inhibin A is a heterodimeric glycoprotein hormone expressed in the gonads and adrenal cortex.

  • Inhibin A serum concentrations are elevated in some patients with PBMAH, suggesting the potential use of this hormone as a tumor marker.

  • Further exploration of serum inhibin A concentration, as it relates to PBMAH disease progression, is warranted to determine if this hormone could serve as an early detection marker and/or predictor of successful surgical treatment.

Taxonomy:
Clinical Overview, Gland/Organ, Adrenal, Topic, Adrenal, Hormone, ACTH, Cortisol, Inhibin, Condition/ Syndrome, Cushing's syndrome, Macronodular Adrenal Hyperplasia, Macronodular hyperplasia, Patient Demographics, Age, Adult, Gender, Female, Male, Ethnicity, Asian - Korean, Black - African, White, Country of Treatment, United States, Diagnosis and Treatment, Signs and Symptoms, Amenorrhoea, Diabetes mellitus type 1, Diabetes mellitus type 2, Gynaecomastia, Hypercortisolaemia, Hyperglycaemia, Hypogonadism, Telangiectasias, Weight gain, Investigation, ACTH, CT scan, Histopathology, Immunohistochemistry, Molecular genetic analysis, Urinary free cortisol, Intervention, Adrenalectomy, Related Disciplines, Genetics, Publication Details, Case Report Type, Novel diagnostic procedure
DOI:
https://doi.org/10.1530/EDM-20-0006
Volume/Issue:
Volume 2020: Issue 1
Open access
Jiman Kim
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Eulsun Moon Seoul 365 Medical Clinic, Seoul, Korea

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Seungwon Kwon Department of Cardiovascular and Neurologic Diseases, College of Korean Medicine, KyungHee University, 1 Hoegi-dong, Dongdaemun-gu, Seoul 130-702, Korea

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Summary

Diabetic nephropathy, a microvascular complication of diabetes, is a progressive kidney disease caused by angiopathy of the capillaries in the kidney glomeruli. Herein, we report a case of a 62-year-old patient with a 30 year history of diabetes, who showed a substantial improvement in diabetic nephropathy on administration of 30 g of Astragalus membranaceus extract per day. After 1 month, estimated glomerular filtration rate increased from 47 to 72 ml/min per 1.73 m2 and was subsequently maintained at the 1-month follow-up. Urinary protein levels also decreased following treatment. Herein, we present and discuss the evidence and mechanism of A. membranaceus on diabetic nephropathy in this patient.

Learning points

  • Diabetic nephropathy is a progressive kidney disease.

  • Angiotensin-converting enzyme (ACE) inhibitors or angiotensin receptor blockers (ARBs) are currently used to prevent and delay the progression of diabetic nephropathy. However, their effects are not sufficient to prevent a decline in kidney function.

  • Furthermore, combination therapy with an ACE inhibitor and an ARB can produce adverse effects without additional benefits.

  • In the early phase of diabetic nephropathy, administration of Astragalus membranaceus can be a therapeutic option.

Taxonomy:
Clinical Overview, Gland/Organ, Pancreas, Topic, Diabetes, Hormone, Insulin, Condition/ Syndrome, Diabetes mellitus type 2, Diabetic nephropathy, Hypertension, Patient Demographics, Age, Geriatric, Gender, Male, Ethnicity, Asian - Korean, Country of Treatment, Korea, Republic of, Diagnosis and Treatment, Signs and Symptoms, Diabetic foot ulceration, Hypertension, Proteinuria, Retinopathy, Investigation, Creatinine (serum), Estimated glomerular filtration rate, Medication, Astragalus membranaceus, Atorvastatin, Calcium dobesilate, Clopidogrel, Diltiazem, DPP4 inhibitors, Ginkgo biloba, Insulin, Metformin, Perindopril, Sitagliptin, Related Disciplines, Nephrology, Publication Details, Case Report Type, Novel treatment
DOI:
https://doi.org/10.1530/EDM-14-0063
Volume/Issue:
Volume 2014: Issue 1
Open access