Postoperative adrenal cortical insufficiency following adrenal adenoma removal: a case report

Introduction

The adrenal glands are one of the body’s important endocrine organs. With advances in medical technology, the diagnosis rates of adrenal diseases, such as adrenal tumors and adrenal cortical hyperplasia, have been gradually increasing (1). Surgery is the mainstream approach for addressing adrenal masses. However, unilateral adrenalectomy can affect the patient’s adrenal cortical function, leading to postoperative adrenal cortical insufficiency (2). Adrenal cortical insufficiency is caused by a deficiency of glucocorticoids, with or without a reduction in mineralocorticoids and sex hormones. It can be classified into primary and secondary adrenal cortical insufficiency based on the underlying causes (3). Adrenal venous sampling (AVS) is often one of the diagnostic methods used to determine the presence of functional adenomas in the adrenal glands. For functional adrenal adenomas, unilateral adrenalectomy may result in adrenal cortical insufficiency. The clinical manifestations of this condition are prolonged and lack specific characteristics, which can sometimes lead to misdiagnosis and mistreatment (4).

Here, we summarize the diagnostic and therapeutic process of a case where a patient developed cortical insufficiency following unilateral adrenalectomy for an adrenal adenoma. Additionally, we further explore the use of AVS in the diagnosis and treatment of adrenal tumors. This approach aims to improve the preoperative diagnostic accuracy of functional adrenal adenomas, prevent postoperative adrenal cortical insufficiency, and enhance the success rate of clinical diagnosis and treatment. We present this article in accordance with the CARE reporting checklist (available at https://acr.amegroups.com/article/view/10.21037/acr-24-257/rc).

Case presentation

The patient is a 28-year-old female who was admitted to the hospital due to “hypertension for 2 years and a right adrenal mass detected over 3 months ago”. In the past, elevated blood pressure was discovered during a physical examination, with the highest reading being 160/90 mmHg. She did not experience dizziness, headaches, palpitations, chest tightness, blurred vision, or excessive sweating. Initially, there was no regular treatment or diagnosis. Later, a computed tomography (CT) scan at a lower-tier hospital revealed a right adrenal mass, prompting her to seek further diagnosis and treatment at Baoshan People’s Hospital. A CT examination conducted during hospitalization indicated a mass approximately 2.3 cm × 2 cm in size located near the junction of the lateral branch of the right adrenal gland. The CT value was about 43 Hounsfield unit (HU), with uneven and persistent enhancement observed. The CT findings suggested the possibility of a right adrenal pheochromocytoma (Figure 1). Upon admission, blood tests showed no hypokalemia, and the electrocardiogram (ECG) did not reveal any significant abnormalities. Comprehensive AVS and hormone level testing indicated varying degrees of elevation in angiotensin, aldosterone, and cortisol levels in the blood from the right adrenal vein compared to the opposite side and the vena cava, with aldosterone being predominantly elevated (Table 1), Furthermore, blood and urinary catecholamine levels in this patient showed no significant abnormalities. All procedures performed in this study were in accordance with the ethical standards of the institutional and/or national research committee(s) and the Declaration of Helsinki and its subsequent amendments. Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the editorial office of this journal.

Figure 1 The CT scan indicated a mass (red arrow) approximately 2.3 cm × 2 cm in size near the junction of the lateral branch of the right adrenal gland. The CT value was about 43 HU, with uneven and persistent enhancement. CT, computed tomography; HU, Hounsfield unit.

Oral phenoxybenzamine and spironolactone were administered to control blood pressure and for appropriate volume expansion as part of the preoperative preparation. One week later, a laparoscopic right adrenalectomy was performed. Postoperative pathological examination indicated a right adrenal cortical adenoma (Figure 2A). The immunohistochemical results indicate: calretinin (−), Melan-A (+), Inhibin-a (+), NSE (−), CgA (−), Syn (+), EMA (−), Vimentin (−), Bcl-2 (−), Ki67 (<2%) (Figure 2B). Two weeks post-surgery, the patient repeatedly experienced symptoms of nausea and vomiting, along with poor mental status. Treatments such as antiemetics and acid suppression for gastric protection administered outside the hospital proved ineffective, and the symptoms continued to worsen. Upon returning to our hospital, the patient’s blood pressure was measured at 126/90 mmHg, with a pulse rate of 100 beats per minute. Peripheral blood hormone levels showed an abnormally low cortisol level and elevated adrenocorticotropic hormone (ACTH) (Table 2). Considering the patient’s symptoms and test results, the postoperative condition is likely adrenal cortical insufficiency. After administering hydrocortisone replacement therapy, the patient’s symptoms improved compared to before.

Figure 2 Adrenal tumor mass and histopathological sections. (A) Postoperative pathological examination indicated an adrenal cortical adenoma. (B) H&E staining and the immunohistochemical results indicate: calretinin(−), Melan-A(+), Inhibin-a(+), NSE(−), CgA(−), Syn(+), EMA(−), Vimentin(−), Bcl-2(−), Ki67(<2%). H&E, hematoxylin and eosin.

Discussion

Adrenal tumors are common in urology. With the advancement of imaging technologies and the widespread practice of health check-ups, the detection rate of adrenal tumors has continuously increased (5). The adrenal gland is composed of the cortex and medulla. The cortex is divided into three distinct zones: the zona glomerulosa (which secretes mineralocorticoids like aldosterone), the zona fasciculata (which secretes glucocorticoids like cortisol), and the zona reticularis (which secretes sex hormones). The medulla produces stress hormones, including adrenaline (6). Functional adrenal tumors have different origins and abnormally secrete various types and levels of hormones, leading to differences in clinical manifestations, preoperative preparation, treatment, and postoperative management. In the past, the diagnosis of adrenal tumors typically relied on imaging studies for localization and characterization, but some patients may present with unclear clinical manifestations. For instance, in cases of primary aldosteronism (PA), a small number of patients can have completely normal blood pressure, and some may not exhibit hypokalemia. Additionally, imaging studies can sometimes lead to misdiagnosis (7). AVS is one of the techniques used to determine the functionality and classification of adrenal tumors, offering high sensitivity and specificity. It is often recommended for distinguishing between unilateral and bilateral PA, before making definitive decisions regarding surgical or medical treatment (8). In this case, preoperative imaging suggested a pheochromocytoma originating from the adrenal medulla. However, AVS indicated significantly elevated levels of aldosterone and cortisol on the affected side, which was consistent with the postoperative pathological findings.

Laparoscopic adrenalectomy is the “gold standard” surgical procedure for treating unilateral adrenal adenomas. However, there is still considerable debate over the choice between unilateral partial adrenalectomy and total adrenalectomy (9). When the contralateral adrenal gland is normal, unilateral total adrenalectomy can reduce the recurrence rate of adrenal adenomas. However, related literature reports indicate that it may increase the need for postoperative hormone replacement. The patient’s right adrenal tumor was located at the junction of the lateral branch and the confluence region, and imaging suggested no abnormalities in the contralateral adrenal gland. We opted for a laparoscopic right adrenalectomy. However, postoperatively, the patient developed symptoms such as nausea, vomiting, and poor mental status, which are clinical manifestations of cortisol insufficiency, requiring hormone replacement therapy (10). Reviewing the patient’s preoperative AVS results, PA was mostly considered, but it also indicated that the right adrenal gland was the dominant side for secreting cortisol. Therefore, for patients of this type, regardless of whether symptoms of cortical insufficiency appear postoperatively, prophylactic treatment with glucocorticoids can be considered. However, due to the lack of large-sample data studies, whether preoperative AVS results can provide significant evaluative value in choosing the surgical approach for adrenal adenomas and in determining the necessity for prophylactic glucocorticoid use postoperatively to minimize the risk of cortisol insufficiency still needs further exploration.

Following unilateral adrenalectomy, there can be a risk of adrenal insufficiency (AI). The clinical manifestations of AI are non-specific, and in most patients, the condition develops over a long period, with more subtle symptoms (11). This often leads to misdiagnosis. Common symptoms of AI generally include fatigue, weight loss, loss of appetite, nausea, vomiting, diarrhea, and dehydration; low blood pressure, orthostatic hypotension, hypovolemic shock; high fever; confusion, lethargy, and coma; muscle cramps in the back and legs; and widespread skin hyperpigmentation (12). There are also cases where patients exhibit atypical clinical presentations, and there is a report in the literature of AI patients presenting primarily with abdominal pain (1). If AI is not addressed promptly, the patient’s symptoms may further worsen, leading to an adrenal crisis (AC), which, in severe cases, can be life-threatening (13). Therefore, after unilateral adrenalectomy, it is crucial to closely monitor the patient’s vital signs. If the patient repeatedly experiences discomfort that cannot be explained by known conditions, it is essential to promptly conduct blood tests to assess adrenal hormone levels and initiate glucocorticoid replacement therapy as needed (14).

Conclusions

AVS for hormone level testing offers certain advantages in diagnosing the nature of functional adrenal adenomas before surgery and provides valuable reference information for selecting the surgical approach. Primary adrenal cortical insufficiency is relatively rare in clinical practice and is usually seen after unilateral adrenal surgery. Its clinical manifestations are non-specific and are often subject to misdiagnosis and mistreatment. Therefore, it is essential to enhance monitoring of patients who have undergone unilateral adrenalectomy and to administer hormone replacement therapy promptly when necessary.

Acknowledgments

None.

Footnote

Reporting Checklist: The authors have completed the CARE reporting checklist. Available at https://acr.amegroups.com/article/view/10.21037/acr-24-257/rc

Peer Review File: Available at https://acr.amegroups.com/article/view/10.21037/acr-24-257/prf

Funding: None.

Conflicts of Interest: All authors have completed the ICMJE uniform disclosure form (available at https://acr.amegroups.com/article/view/10.21037/acr-24-257/coif). The authors have no conflicts of interest to declare.

Ethical Statement: The authors are accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved. All procedures performed in this study were in accordance with the ethical standards of the institutional and/or national research committee(s) and the Declaration of Helsinki and its subsequent amendments. Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the editorial office of this journal.

Open Access Statement: This is an Open Access article distributed in accordance with the Creative Commons Attribution-NonCommercial-NoDerivs 4.0 International License (CC BY-NC-ND 4.0), which permits the non-commercial replication and distribution of the article with the strict proviso that no changes or edits are made and the original work is properly cited (including links to both the formal publication through the relevant DOI and the license). See: https://creativecommons.org/licenses/by-nc-nd/4.0/.

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