Unilateral adrenalectomy for a drug-resistant bilateral primary aldosteronism with heart failure: pathophysiology and surgical indication
Abstract Background Patients with bilateral primary aldosteronism (PA) generally are treated with antihypertensive drugs, but optimal treatment for patients with complications due to refractory hypertension has not been established. In this report, we present a case with bilateral PA who presented w...
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| Language: | English |
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BMC
2023-11-01
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| Series: | BMC Endocrine Disorders |
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| Online Access: | https://doi.org/10.1186/s12902-023-01503-2 |
| _version_ | 1827764400082124800 |
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| author | Seiji Hoshi Akifumi Onagi Ryo Tanji Ruriko Honda-Takinami Kanako Matsuoka Junya Hata Yuichi Sato Hidenori Akaihata Masao Kataoka Soichiro Ogawa Yoshiyuki Kojima |
| author_facet | Seiji Hoshi Akifumi Onagi Ryo Tanji Ruriko Honda-Takinami Kanako Matsuoka Junya Hata Yuichi Sato Hidenori Akaihata Masao Kataoka Soichiro Ogawa Yoshiyuki Kojima |
| author_sort | Seiji Hoshi |
| collection | DOAJ |
| description | Abstract Background Patients with bilateral primary aldosteronism (PA) generally are treated with antihypertensive drugs, but optimal treatment for patients with complications due to refractory hypertension has not been established. In this report, we present a case with bilateral PA who presented with persistent hypertension, despite treatment with 6 drugs, and left-dominant heart failure, which was improved after unilateral adrenalectomy. Case presentation A 61-year-old man was admitted to our hospital because of severe left-dominant heart failure. His heart rhythm was atrial fibrillation and the left ventricle was diffusely hypertrophic and hypokinetic. Coronary arteries were normal on coronary arteriogram. Primary aldosteronism was suspected based on severe hypokalemia (2.5 mEq/L) and plasma aldosterone concentration (PAC; 1,410 pg/mL). Although computed tomography (CT) showed a single left cortical nodule, adrenal vein sampling (AVS) indicated bilateral PA. Early in the case, heart failure and hyperkalemia in this patient were improved by treatment with a combination of 6 antihypertensive drugs (spironolactone 25 mg/day, eplerenone 100 mg/day, azosemide 60 mg/day, tolvaptan 7.5 mg/day, enalapril 5 mg/day, and bisoprolol fumarate 10 mg/day); however, heart failure relapsed after four months of treatment. We hypothesized that hypertension caused by excess aldosterone was inducing the patient’s heart failure. In order to reduce aldosterone secretory tissue, a laparoscopic adrenalectomy was performed for the left adrenal gland, given the higher level of aldosterone from the left gland compared to the right. Following surgery, the patient’s heart failure was successfully controlled despite the persistence of high PAC. Treatment with anti-hypertensive medications was reduced to two drugs (eplerenone 100 mg/day and bisoprolol fumarate 10 mg/day). In order to elucidate the mechanism of drug resistance, immunohistochemistry (IHC) and real time-polymerase chain reaction (RT-PCR) assays were performed to assess the expression of steroidogenic factor 1 (SF-1), a regulator of steroid synthesis in adrenal tissue. IHC and RT-PCR demonstrated that the expression of SF-1 in this patient (at both the protein and mRNA levels) was higher than that observed in unilateral PA cases that showed good responsivity to drug treatment. Conclusions Unilateral adrenalectomy to reduce aldosterone secretory tissue may be useful for patients with drug-refractory, bilateral PA. Elevated expression of SF-1 may be involved in drug resistance in PA. |
| first_indexed | 2024-03-11T11:04:56Z |
| format | Article |
| id | doaj.art-fd0e6e87245b4130a0719d8be4ef3586 |
| institution | Directory Open Access Journal |
| issn | 1472-6823 |
| language | English |
| last_indexed | 2024-03-11T11:04:56Z |
| publishDate | 2023-11-01 |
| publisher | BMC |
| record_format | Article |
| series | BMC Endocrine Disorders |
| spelling | doaj.art-fd0e6e87245b4130a0719d8be4ef35862023-11-12T12:20:07ZengBMCBMC Endocrine Disorders1472-68232023-11-012311610.1186/s12902-023-01503-2Unilateral adrenalectomy for a drug-resistant bilateral primary aldosteronism with heart failure: pathophysiology and surgical indicationSeiji Hoshi0Akifumi Onagi1Ryo Tanji2Ruriko Honda-Takinami3Kanako Matsuoka4Junya Hata5Yuichi Sato6Hidenori Akaihata7Masao Kataoka8Soichiro Ogawa9Yoshiyuki Kojima10Departments of Urology, Fukushima Medical University School of MedicineDepartments of Urology, Fukushima Medical University School of MedicineDepartments of Urology, Fukushima Medical University School of MedicineDepartments of Urology, Fukushima Medical University School of MedicineDepartments of Urology, Fukushima Medical University School of MedicineDepartments of Urology, Fukushima Medical University School of MedicineDepartments of Urology, Fukushima Medical University School of MedicineDepartments of Urology, Fukushima Medical University School of MedicineDepartments of Urology, Fukushima Medical University School of MedicineDepartments of Urology, Fukushima Medical University School of MedicineDepartments of Urology, Fukushima Medical University School of MedicineAbstract Background Patients with bilateral primary aldosteronism (PA) generally are treated with antihypertensive drugs, but optimal treatment for patients with complications due to refractory hypertension has not been established. In this report, we present a case with bilateral PA who presented with persistent hypertension, despite treatment with 6 drugs, and left-dominant heart failure, which was improved after unilateral adrenalectomy. Case presentation A 61-year-old man was admitted to our hospital because of severe left-dominant heart failure. His heart rhythm was atrial fibrillation and the left ventricle was diffusely hypertrophic and hypokinetic. Coronary arteries were normal on coronary arteriogram. Primary aldosteronism was suspected based on severe hypokalemia (2.5 mEq/L) and plasma aldosterone concentration (PAC; 1,410 pg/mL). Although computed tomography (CT) showed a single left cortical nodule, adrenal vein sampling (AVS) indicated bilateral PA. Early in the case, heart failure and hyperkalemia in this patient were improved by treatment with a combination of 6 antihypertensive drugs (spironolactone 25 mg/day, eplerenone 100 mg/day, azosemide 60 mg/day, tolvaptan 7.5 mg/day, enalapril 5 mg/day, and bisoprolol fumarate 10 mg/day); however, heart failure relapsed after four months of treatment. We hypothesized that hypertension caused by excess aldosterone was inducing the patient’s heart failure. In order to reduce aldosterone secretory tissue, a laparoscopic adrenalectomy was performed for the left adrenal gland, given the higher level of aldosterone from the left gland compared to the right. Following surgery, the patient’s heart failure was successfully controlled despite the persistence of high PAC. Treatment with anti-hypertensive medications was reduced to two drugs (eplerenone 100 mg/day and bisoprolol fumarate 10 mg/day). In order to elucidate the mechanism of drug resistance, immunohistochemistry (IHC) and real time-polymerase chain reaction (RT-PCR) assays were performed to assess the expression of steroidogenic factor 1 (SF-1), a regulator of steroid synthesis in adrenal tissue. IHC and RT-PCR demonstrated that the expression of SF-1 in this patient (at both the protein and mRNA levels) was higher than that observed in unilateral PA cases that showed good responsivity to drug treatment. Conclusions Unilateral adrenalectomy to reduce aldosterone secretory tissue may be useful for patients with drug-refractory, bilateral PA. Elevated expression of SF-1 may be involved in drug resistance in PA.https://doi.org/10.1186/s12902-023-01503-2Primary aldosteronismLaparoscopic adrenalectomyHeart Failure |
| spellingShingle | Seiji Hoshi Akifumi Onagi Ryo Tanji Ruriko Honda-Takinami Kanako Matsuoka Junya Hata Yuichi Sato Hidenori Akaihata Masao Kataoka Soichiro Ogawa Yoshiyuki Kojima Unilateral adrenalectomy for a drug-resistant bilateral primary aldosteronism with heart failure: pathophysiology and surgical indication BMC Endocrine Disorders Primary aldosteronism Laparoscopic adrenalectomy Heart Failure |
| title | Unilateral adrenalectomy for a drug-resistant bilateral primary aldosteronism with heart failure: pathophysiology and surgical indication |
| title_full | Unilateral adrenalectomy for a drug-resistant bilateral primary aldosteronism with heart failure: pathophysiology and surgical indication |
| title_fullStr | Unilateral adrenalectomy for a drug-resistant bilateral primary aldosteronism with heart failure: pathophysiology and surgical indication |
| title_full_unstemmed | Unilateral adrenalectomy for a drug-resistant bilateral primary aldosteronism with heart failure: pathophysiology and surgical indication |
| title_short | Unilateral adrenalectomy for a drug-resistant bilateral primary aldosteronism with heart failure: pathophysiology and surgical indication |
| title_sort | unilateral adrenalectomy for a drug resistant bilateral primary aldosteronism with heart failure pathophysiology and surgical indication |
| topic | Primary aldosteronism Laparoscopic adrenalectomy Heart Failure |
| url | https://doi.org/10.1186/s12902-023-01503-2 |
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