Bilateral adrenal hemorrhage: learning notes from clinical practice and literature review.

Adrenal hemorrhage is a rare, but important, diagnosis to recognize, in particular when there is involvement of both adrenal glands. Bilateral adrenal hemorrhage can in fact lead to adrenal insufficiency, with dramatic consequences if not promptly recognized and treated. It is normally caused by systemic conditions that lead to the vasoconstriction and thrombosis of the adrenal vein. Oftentimes, the clinical diagnosis of this condition can be very challenging, as its signs and symptoms are generalized and nonspecific (abdominal pain, nausea, and fatigue). Here, we present the cases of two patients admitted to the Emergency Department in 2016 and 2022 with acute abdominal pain, having recently undergone surgery and subsequently prescribed low-molecular-weight heparin. In both cases, laboratory results revealed neutrophilic leukocytosis and an unexplained anemia. Due to the persistence of abdominal pain despite medication, a CT scan was performed, showing an enlargement of both adrenal glands suggestive of bilateral adrenal hemorrhage. Adrenal function was tested that correlated with a diagnosis of adrenal insufficiency, and both patients were promptly treated with parenteral hydrocortisone as a result. On 5 years' follow-up from the acute event, the second patient's adrenal function had returned to normal, and he has not needed further adrenal replacement therapy; the first patient however demonstrated persistence of adrenal failure requiring replacement therapy. In this paper, through our experience and a literature analysis, we will aim to outline some clues to identify patients at potential risk of bilateral adrenal hemorrhage.

Primary adrenal insufficiency due to bilateral adrenal hemorrhage-adrenal infarction in a patient with systemic lupus erythematosus and antiphospholipid syndrome: case presentation and review of the literature.

Primary adrenal insufficiency (PAI) is a rare disease which represents the end stage of a destructive process involving the adrenal cortex. Occasionally it may be caused by bilateral adrenal hemorrhagic infarction in patients with antiphospholipid syndrome (APS). We herein report the challenging case of a 30-year-old female patient with systemic lupus erythematosus (SLE) and secondary APS who was admitted to the emergency department (ED) due to fever, lethargy, and syncopal episodes. Hyponatremia, hyperkalemia, hyperpigmentation, shock, altered mental status, and clinical response to glucocorticoid administration were features highly suggestive of an acute adrenal crisis. The patient's clinical status required admission to the intensive care unit (ICU), where steroid replacement, anticoagulation, and supportive therapy were provided, with a good outcome. Imaging demonstrated bilateral adrenal enlargement attributed to recent adrenal hemorrhage. This case highlights the fact that bilateral adrenal vein thrombosis and subsequent hemorrhage can be part of the thromboembolic complications seen in both primary and secondary APS and which, if misdiagnosed, may lead to a life-threatening adrenal crisis. High clinical suspicion is required for its prompt diagnosis and management. A literature search of past clinical cases with adrenal insufficiency (AI) in the setting of APS and SLE was conducted using major electronic databases. Our aim was to retrieve information about the pathophysiology, diagnosis, and management of similar conditions.

Acute adrenal insufficiency secondary to bilateral adrenal haemorrhage.

Not required for Clinical Vignette.

Adrenal Hemorrhage: A Review.

Importance: Adrenal hemorrhage in pregnancy is rare. The prevalence of pregnant patients whose pregnancies are complicated by preeclampsia or eclampsia is hypothesized to be slightly higher than the 0.4% to 1.1% occurrence rate in the nonpregnant population. However, the mortality rate of adrenal hemorrhage is reportedly as high as 15%. Therefore, it is critical for obstetric providers to have basic knowledge on the presentation, diagnosis, and management of adrenal hemorrhage in the pregnant population so that prompt diagnosis can be made. Objective: This review highlights incidence, pathophysiology, risk factors, diagnosis, management, and complications of adrenal hemorrhage in the peripartum period. Evidence Acquisition: A literature search was undertaken by our research university librarian using the search engines of PubMed, CINAHL, and EMBASE (Medline items removed). The search terms used included "adrenal hemorrhage" OR "adrenal gland hemorrhage" AND "pregnancy" OR "maternal." The search was limited to articles in English, and the years searched were from January 1, 2015 to December 31, 2021. Results: There were 130 abstracts identified, and 30 of the articles were ultimately used as the basis for this review. Presenting signs and symptoms of adrenal hemorrhage were typically abdominal, back, and flank pain. Diagnosis was typically made with ultrasound and computed tomography or magnetic resonance imaging without contrast for confirmation. Management options include conservative management versus surgical management with adrenalectomy or interventional radiology embolization in the unstable patient. For patients with evidence of adrenal insufficiency, steroid replacement was used. Most patients with adrenal hemorrhage in the literature had unilateral adrenal hemorrhage; however, several cases of bilateral adrenal hemorrhage have been reported. Patients with bilateral adrenal hemorrhage were more likely to require steroids for adrenal insufficiency. There are no known contraindications to vaginal delivery in this group of patients, and patients who were managed conservatively were often able to continue the pregnancy to term. Conclusions: Early recognition and management are integral in decreasing the morbidity and mortality associated with adrenal hemorrhage. Relevance Statement: This is an evidence-based review of adrenal hemorrhage in pregnancy and how to diagnose and manage a pregnancy complicated by adrenal hemorrhage.

Approach to the Patient With Adrenal Hemorrhage.

Adrenal hemorrhage is an uncommon, underrecognized condition that can be encountered in several clinical contexts. Diagnosing adrenal hemorrhage is challenging due to its nonspecific clinical features. Therefore, it remains a diagnosis that is made serendipitously on imaging of acutely unwell patients rather than with prospective clinical suspicion. Adrenal hemorrhage can follow abdominal trauma or appear on a background of predisposing conditions such as adrenal tumors, sepsis, or coagulopathy. Adrenal hemorrhage is also increasingly reported in patients with COVID-19 infection and in the context of vaccine-induced immune thrombocytopenia and thrombosis. Unexplained abdominal pain with hemodynamic instability in a patient with a predisposing condition should alert the physician to the possibility of adrenal hemorrhage. Bilateral adrenal hemorrhage can lead to adrenal insufficiency and potentially fatal adrenal crisis without timely recognition and treatment. In this article, we highlight the clinical circumstances that are associated with higher risk of adrenal hemorrhage, encouraging clinicians to prospectively consider the diagnosis, and we share a diagnostic and management strategy.

Neonatal bilateral adrenal hemorrhage and adrenal insufficiency accompanied by Subgaleal hematoma: a case report with brief review of literature.

BACKGROUND: Neonatal adrenal hemorrhage (NAH) is an almost infrequent phenomenon (0.2-0.55%). Mechanical compression and alterations of venous pressure during delivery are considered the most probable explanations. Approximately 10% of the cases might have bilateral involvement. Clinical symptoms include abdominal mass, poor feeding, vomiting, prolonged jaundice, and anemia. Subgaleal hemorrhage (SGH) is one of the most clinically remarkable and potentially hazardous postnatal cranial injuries. CASE PRESENTATION: An early-term Iranian male neonate who was born through spontaneous vaginal delivery and experienced shoulder dystocia was diagnosed with bilateral NAH leading to adrenal insufficiency requiring glucocorticoid and mineralocorticoid supplementation. The SGH and jaundice were other postnatal complications. Serial monthly abdominal and brain ultrasound revealed complete regression of lesions after 70 days. However, after 16 months, the neonate has been still treated with hydrocortisone and fludrocortisone for the adrenal insufficiency diagnosis. He has a lower limit weight for age; however, developmental milestones have been appropriate for age. DISCUSSION AND CONCLUSION: Adrenal hemorrhage and SGH should be examined and looked for, particularly with proven evidence of difficult delivery and asphyxia in at-risk newborns. Clinical and ultrasound follow-up is mandatory for the assessment of hemorrhage resolution and conservative management. The early detection and treatment of adrenal insufficiency by laboratory examination is strongly recommended in bilateral cases. Furthermore, the early recognition of postnatal SGH to prevent clinical and neurological outcomes seems essential.

Waterhouse-Friderichsen Syndrome with Bilateral Adrenal Hemorrhage Associated with Methicillin-Resistant Staphylococcus aureus (MRSA) Bacteremia in an Adult Patient with History of Intravenous Drug Use.

BACKGROUND Waterhouse-Friderichsen syndrome, also known as acute adrenal insufficiency due to adrenal gland hemorrhage, is an uncommon and frequently fatal condition classically presenting with fever, shock, rash, and coagulopathy. Although most often associated with Meningococcemia, many other etiologies have been implicated, including reports of Staphylococcus aureus infection on autopsy examinations. This report details an adult intravenous drug user with adrenal hemorrhage associated with methicillin-resistant Staphylococcus aureus (MRSA) bacteremia. CASE REPORT A 58-year-old man with a history of intravenous drug use presented to the hospital with weakness. Vitals were initially normal and exam findings were notable for decreased right-sided motor strength. Magnetic resonance imaging (MRI) revealed a cervical epidural abscess with spinal cord compression. Despite initiation of broad-spectrum antibiotics and intravenous fluids, the patient progressed to shock, requiring vasopressor administration, and his blood cultures later grew MRSA. Further imaging of the abdomen/pelvis was completed, revealing bilateral adrenal hemorrhage. Random cortisol at that time was 5.6 µg/dL, confirming a diagnosis of critical illness-related corticosteroid insufficiency in addition to likely septic and spinal shock. The patient was initiated on hydrocortisone with improvement in his hypotension. He was transitioned to prednisone and fludrocortisone in addition to 8 weeks of antibiotics after achieving clinical stability. CONCLUSIONS This report brings to attention the risk of adrenal hemorrhage and acute adrenal insufficiency as a sequela of the relatively common illness of Staphylococcus aureus bacteremia. As symptoms of adrenal insufficiency can overlap with septic shock related to the primary condition, this diagnosis requires a high index of suspicion in the critically ill patient.

[Bilateral macronodular adrenal hyperplasia: a rare cause of Cushing's syndrome (a case report)]. / Hyperplasie macronodulaire bilatérale des surrénales: cause rare du syndrome de Cushing (à propos d'un cas).

Bilateral macronodular adrenal hyperplasia (BMAH) is a rare cause of adrenal Cushing's syndrome, accounting for less than 1% of cases. We here report the case of a 48-year-old patient with diabetes and hypertension presenting with clinical signs of Cushing's syndrome. Etiological assessment enabled clinicians to retain the diagnosis of Adrenocorticotropic hormone (ACTH)-independent hypercortisolism associated with BMAH. Unilateral left adrenalectomy was performed based on noriodocholesterol scintigraphy results, with good outcome. However, given the risk of recurrence and cardiovascular complications, long-term monitoring was scheduled.

Adrenocorticotropic hormone (ACTH) independent Cushing's syndrome due to unilateral adrenocortical hyperplasia: two case reports.

Adrenal unilateral macronodular hyperplasia is a rare cause of Cushing's syndrome. We discuss the case of two patients who present Cushing syndrome due to unilateral adrenal hyperplasia. They presented the signs of clinical hypercorticism as well as metabolic, cardiovascular and osteoporotic complications. Both patients presented clinical and laboratory signs of adrenocorticotropic hormone (ACTH)-independent Cushing syndrome with elevated urinary free cortisol (UFC) levels, adrenal computed tomography (CT) scan revealed the appearance of unilateral adrenal adenoma and normal contralateral adrenal gland. Adrenalectomy was performed under laparoscopic surgery; the resected mass was pathologically diagnosed as unilateral nodular adrenal hyperplasia. Unilateral adrenal hyperplasia is a very rare etiology of ACTH-independent Cushing syndrome, often mistaken for adenoma on CT and only pathological examination can confirm the diagnosis.

Evaluation of metabolic parameters and aortic elasticity in normotensive children with premature adrenarche.

OBJECTIVES: Premature adrenarche may be associated with an intrauterine programmed metabolic syndrome which should be considered as a warning sign for coronary heart disease due to accelerated atherosclerosis, hypertension, type 2 diabetes mellitus (DM), and polycystic ovary syndrome. METHODS: Seventy-three patients with premature adrenarche were evaluated for metabolic parameters and aortic elasticity to evaluate the susceptibility to atherosclerosis and compared with a control group. The patients were examined in two groups as overweight and nonoverweight, and metabolic and cardiac parameters were also compared among these groups. Strain, distensibility, and stiffness index parameters were used to evaluate aortic elasticity. RESULTS: Biochemical parameters and cardiac measurements were not statistically different between patients and controls. They also did not differ between patients with normal weight and overweight groups. Atherogenic index and insulin resistance were closely related and a positive correlation between cholesterol and triglyceride, and ascending aortic stiffness was found. CONCLUSIONS: The results may suggest that cholesterol and triglyceride-related arterial involvement is more involved in the pathogenesis of arterial stiffness. It can be considered that 'being overweight' or 'having metabolic profile characterized by insulin resistance and dyslipidemia' are the major coexisting factors influencing the vascular structure, rather than increased androgens and premature adrenarche itself.

COVID-19 and Pneumocystis jirovecii Pulmonary Coinfection-The First Case Confirmed through Autopsy.

Background: Establishing the diagnosis of COVID-19 and Pneumocystisjirovecii pulmonary coinfection is difficult due to clinical and radiological similarities that exist between the two disorders. For the moment, fungal coinfections are underestimated in COVID-19 patients. Case presentation: We report the case of a 52-year-old male patient, who presented to the emergency department for severe dyspnea and died 17 h later. The RT-PCR test performed at his admission was negative for SARS-CoV-2. Retesting of lung fragments collected during autopsy revealed a positive result for SARS-CoV-2. Histopathological examination showed preexisting lesions, due to comorbidities, as well as recent lesions: massive lung thromboses, alveolar exudate rich in foam cells, suprapleural and intra-alveolar Pneumocystisjirovecii cystic forms, and bilateral adrenal hemorrhage. Conclusion: COVID-19 and P.jirovecii coinfection should be considered, particularly in critically ill patients, and we recommend the systematic search for P. jirovecii in respiratory samples.

Chronic kidney disease in adrenal disorders.

PURPOSE OF REVIEW: This review will focus on hypertension due to underlying adrenal disorders in chronic kidney disease (CKD). Diagnosis of adrenal hypertension and particularly primary aldosteronism (PA) in CKD is frequently not pursued. We outline limitations that advanced kidney disease poses on the diagnostic work up of these disorders and provide a framework for approaching CKD patients suspected of having an adrenal disorder. Recognition of these disorders is important as they are often underdiagnosed which leads to poorer outcomes. RECENT FINDINGS: Adrenal disease associated with hypertension in CKD is most commonly due to PA whereas pheochromocytoma and Cushing's disease are important but less common. Diagnosis of these diseases is important as their identification leads to better blood pressure control and can possibly mitigate the risk of progression of CKD. Work up and treatment of PA has been shown to be safe and is associated with less antihypertensive medication requirement for the associated hypertension and slower progression of CKD. SUMMARY: This chapter summarizes the importance of recognizing adrenal hypertension in CKD and reinforces the need for physicians to pursue these diagnoses in CKD patients as this is safe and improves both BP control and delays progression of CKD.

A huge haemorrhagic suprarenal pseudocyst: an unusual presentation of a rare condition.

Haemorrhagic suprarenal pseudocysts are very rare and are often incidental findings at surgery or autopsy, though they can sometimes present with predominantly gastrointestinal or endocrine symptoms, including intraperitoneal bleeding or sepsis. Our case report is of a 48-year-old man who presented at our primary healthcare centre with 2-month history of predominantly respiratory symptoms of cough and shortness of breath. CT scan revealed a suprarenal cyst measuring 14.2×13.5×13.1 cm for which he was operated and made a full recovery. A detailed literature review reveals that there has never been a case of a haemorrhagic suprarenal pseudocyst presenting with predominantly respiratory symptoms, which is why we decided to document this case report.

Robotic and Laparoscopic Approaches for Adrenal Surgery in Obese Patients.

OBJECTIVE: Minimally invasive adrenalectomy is a challenging procedure in obese patients. Few recent studies have advocated against robot-assisted adrenalectomy, particularly in obese patients. This study aims to compare operative outcomes between the robotic and laparoscopic adrenalectomy, particularly in obese patients. MATERIALS AND METHODS: A retrospective analysis was performed on all consecutive patients undergoing adrenalectomy for benign disease by a single surgeon using either a laparoscopic or robotic approach. Adrenal surgeries for adrenal cancer were excluded. Demographics, operative time, length of hospital stays, estimated blood loss (EBL), and intraoperative and postoperative complications were evaluated. Patients were divided into 2 groups; obese and nonobese. A sub-analysis was performed comparing robotic and laparoscopic approaches in obese and nonobese patients. RESULTS: Out of 120, 55 (45.83%) were obese (body mass index ≥ 30 kg/m2). 14 (25.45%) of the obese patients underwent a laparoscopic approach, and 41 (74.55%) underwent a robotic approach. Operative times were longer in the obese vs. nonobese groups (173.30 ± 72.90 minutes and 148.20 ± 61.68 minutes, P = .04) and were associated with less EBL (53.77 ± 82.48 vs. 101.30 ± 122, P = .01). The robotic approach required a longer operative time when compared to the laparoscopic approach (187 ± 72.42 minutes vs. 126.60 ± 54.55 minutes, P = .0102) in the obese but was associated with less blood loss (29.02 ± 51.05 mL vs. 138.30 ± 112.20 mL, P < .01) and shorter hospital stay (1.73 ± 1.23 days vs. 3.17 ± 1.27 days, P < .001). CONCLUSION: Robot-assisted adrenal surgery is safe in obese patients and appears to be longer; however, it provides improvements in postoperative outcomes, including EBL and shorter hospital stay.

Modified three-level techniques of retroperitoneal laparoscopic procedures to treat adrenal lesions for patients with BMI ≥ 25 Kg/m2.

Objective: To evaluate the modified Zhang's 'three-level' technique of retroperitoneal laparoscopic adrenalectomy (RLA) to treat adrenal lesions for patients with BMI of 25-30 Kg/m2. Methods: A retrospective analysis was performed in all patients with BMI of 25-30 Kg/m2 in our hospital from January 2014 to December 2019. Those who underwent laparoscopic adrenal surgery were divided into two groups on the basis of the technique used: the Zhang's technique (the ZT group) and the modified technique (the MT group). Results: Herein, 170 operations were included (ZT, 91 patients; MT, 79 patients). RLA was successfully performed in all of them. Compared with the ZT group patients, the MT group patients showed shorter operation time (p = 0.007), lesser intraoperative blood loss (p = 0.023), shorter operation time, earlier postoperative diet recovery (p < 0.001), shorter postoperative drainage time (p < 0.001) and shorter postoperative hospitalization period (p = 0.001). It was also worth noting that the unplanned total adrenalectomy rate was significantly less in the MT group than in the ZT group (0% vs. 10.8%, p = 0.020). There was no significant difference in the complications between the two groups (3.3% vs. 2.5%, p = 0.567). Conclusions: We found that MT was a beneficial retroperitoneal laparoscopic treatment for adrenal lesions in patients who had a BMI of 25-30 Kg/m2. It may provide a reference for the treatment of adrenal surgical diseases in such patients.

A giant adrenal hemorrhagic pseudocyst mimicking a parapelvic renal cyst: A case report and review of the literature.

Adrenal pseudocysts are rare, nonfunctional, asymptomatic cystic masses that originate from the adrenal gland and are usually located in the suprarenal area. They are usually incidentally discovered during imaging, but diagnosis can be challenging because they are similar to benign and malignant cystic lesions of the adrenal gland and adjacent organs. We describe a giant, adrenal hemorrhagic pseudocyst that was atypically located, extending from the middle to the lower poles of the kidney, admixed with a renal cortical cyst.

Cardiomyopathies and Adrenal Diseases.

Cardiomyopathies are myocardial disorders in which heart muscle is structurally and/or functionally abnormal. Previously, structural cardiomyocyte disorders due to adrenal diseases, such as hyperaldosteronism, hypercortisolism, and hypercatecholaminism, were misunderstood, and endomyocardial biopsy (EMB) was not performed because was considered dangerous and too invasive. Recent data confirm that, if performed in experienced centers, EMB is a safe technique and gives precious information about physiopathological processes implied in clinical abnormalities in patients with different systemic disturbances. In this review, we illustrate the most important features in patients affected by primary aldosteronism (PA), Cushing's syndrome (CS), and pheochromocytoma (PHEO). Then, we critically describe microscopic and ultrastructural aspects that have emerged from the newest EMB studies. In PA, the autonomous hypersecretion of aldosterone induces the alteration of ion and water homeostasis, intracellular vacuolization, and swelling; interstitial oedema could be a peculiar feature of myocardial toxicity. In CS, cardiomyocyte hypertrophy and myofibrillolysis could be related to higher expression of atrogin-1. Finally, in PHEO, the hypercontraction of myofilaments with the formation of contraction bands and occasional cellular necrosis has been observed. We expect to clear the role of EMB in patients with cardiomyopathies and adrenal disease, and we believe EMB is a valid tool to implement new management and therapies.