Adrenal pseudocysts are rare entities and occurred in the 5th and the 6th decades of life. They are discovered accidentally, while appearing with nonspecific clinical and imaging findings. We report a case of a 28-year-old woman presented in our Emergency Department complaining about upper abdomen pain. Computed tomography revealed a hypodense cystic lesion containing hyperdense material. The size of a mass was 11. 7 × 9.3 × 6.6 cm in diameter close to the pancreas, but the origin was from the left adrenal gland. The mass was excised with surgical laparotomy. Giant adrenal pseudocysts are rare entities. Final diagnosis usually confirmed with the pathology examination. Management of such adrenal lesions depends on the unique characteristics, the surgeon’s experience, and local resources.
The Viennese anatomist Greiselius was the first who described in 1670 a ruptured adrenal cyst, filled with 12 pounds of fluid and 2 pounds of clot, as the cause of death in a 45-year-old nobleman [
According to the Levison classification [
A 28-year-old woman presented in the Emergency Department complaining about upper abdominal pain during the previous three days, without fever or anorexia, not related to food intake, and without bowel function abnormalities. Her surgical and clinical history was clear, and she had not noticed occurrence of the same symptoms before. During clinical examination, a palpable mass in the lower epigastric area was found, with focal tenderness.
Laboratory results, including cancer biological markers, liver function and renal tests, and serum hormone profile (cortisol, aldosterone, calcium, urinary catecholamines, 5-HIAA, and metanephrine
(a–d) Ultrasound imaging examination.
Surgical approach through left Kocher incision was decided, after thorough information and the patient’s consent. A unilocular mass sized 11.7 × 9.3 × 6.6 cm with 1.0430 cm3 total volume was revealed in close relevance to the pancreas but with the origin from the left adrenal gland and was excised within healthy borders and sent for further pathologic examination (Figures
(a) Intraoperative findings. (b) Final specimen after surgical resection.
Histological examination (Hematoxilina eosina X60).
Prisma chart.
Based on statistic analysis, it becomes clear that most adrenal pseudocysts are asymptomatic because of their small size and their location in the retroperitoneum; however, when they grow up significantly, they may cause compression of neighboring structures or/and increase intra-abdominal pressure [
Author name | Year | Gender | Calcification | Age | Hemorrhage | Size | Volume | Procedure | Side |
---|---|---|---|---|---|---|---|---|---|
Paramythiotis | 2017 | Female | No | 25 | Yes | 11.7 × 9.3 × 6.6 | 1.043 | Pseudocyst removal | Left |
Alvaro Martinez Manzano | 2017 | Female | Yes | 73 | Yes | 5 × 5 cm | 0.125 | Open adrenalectomy | Right |
Geleit | 2016 | Male | No | 33 | Yes | Large | 1.848 | Open adrenalectomy | Right |
Pathaik | 2015 | Male | No | 50 | Yes | 21 × 18 × 15 | 5.67 | Open adrenalectomy | Right |
Schrank | 2014 | Female | Yes | 62 | Yes | 9.1 × 7.4 × 6.9 | 1.145 | Laparoscopic adrenalectomy | Left |
Soroush | 2013 | Female | No | 32 | Yes | 15.1 × 11.5 × 17 | 2.95 | Open adrenalectomy | Left |
Angelico | 2013 | Female | No | 30 | Yes | 10 × 7 × 10 | 0.7 | Laparoscopic cyst removal | Left |
Cantisani | 2013 | Male | No | 75 | Yes | 19 × 14 × 12 | 3.192 | Open adrenalectomy | Right |
Passoni | 2013 | Male | Yes | 69 | Yes | 12 × 9 × 7 | 0.756 | Open adrenalectomy + splenectomy + pancreatic tail resection | Left |
Mahmodlou | 2011 | Female | No | 21 | Yes | 15 × 9.7 × 15 | 2.182 | Open adrenalectomy | Right |
Zanghi | 2012 | Female | No | 39 | Yes | 8 × 8 | 0.448 | Laparoscopic adrenalectomy | Left |
Karim | 2016 | Male | No | 70 | Yes | 22 × 20 × 19 | 8.36 | Pseudocyst open removal | Right |
Gupt | 2011 | Male | No | 58 | Yes | 23 × 15 × 12 | 4.14 | Open adrenalectomy | Left |
Ujam | 2011 | Female | No | 39 | Yes | 29 × 20 × 17 | 9.86 | Laparoscopic adrenalectomy | Right |
Marwah | 2011 | Male | No | 60 | Yes | 6 × 6 | 0.252 | Cyst removal | Left |
Yusuharu | 2011 | Female | No | 43 | Yes | 22 × 13 × 3 | 0.858 | Cyst removal | Left |
Momiyama | 2011 | Male | No | 52 | Yes | 18 × 18 | 5.508 | Open adrenalectomy | Right |
Salemis | 2011 | Male | No | 45 | Yes | 15.5 × 13.5 | 2.511 | Laparoscopic adrenalectomy | Right |
Wilkinson | 2011 | Female | No | 64 | Yes | 12 × 6 | 0.504 | Cyst unroofing | Left |
Karaman | 2011 | Female | No | 40 | Yes | 20 × 15 | 4.2 | Open adrenalectomy | Left |
Kim | 2009 | Female | No | 38 | Yes | 9 × 9 | 0.729 | Laparoscopic adrenalectomy | Left |
(a) Box plot diagram showing the median age of patients appearing with adrenal pseudocysts. (b) Box plot diagram of the mean volume of the mass in patients with adrenal pseudocysts.
Although pseudocysts represent the most frequently identified adrenal masses, endothelial cysts are found for up to 45% in autopsy series [
Origins of adrenal pseudocysts still are a matter of debate. Some authors suggest that these lesions result from an intra-adrenal hemorrhage caused by trauma, a sepsis event, or any such systemic insult [
Adrenal pseudocysts may also be functional, causing syndromes like adrenal hypofunction (Cushing’s syndrome) [
The most worrisome feature of adrenal cystic neoplasms, including the pseudocysts, is a reported incidence of malignancy estimated at 7% of the cases, especially if the pseudocyst’s size exceeds 6 cm [
US is usually the first imaging exam that may be employed for the evaluation of patients with a suspected adrenal mass, since it is a modality that has low cost and no exposure to radiation and allows for no confinement of claustrophobic or agitated patients. Nevertheless, the sensitivity of US for the detection of adrenal lesions is highly variable, with reported values from 66.7% to more than 90%, regardless of the lesion’s size. Recently, contrast-enhanced US (CEUS), although not widely utilized, has also been added to the imaging armamentarium, as it has been reported to be able to detect or disprove the enhancement of the lesion’s walls, thus accordingly pointing to a malignant or benign lesion [
CT characteristics of the adrenal pseudocyst include a well-determinated cyst that does not enhance with intravenous contrast, while the density of the cyst’s content measures at water density or much higher, depending on the amount of the encased hemorrhagic debris [
MRI features of adrenal pseudocysts include a solid component with low intensity on both T1- and T2-weighted images and a second, smaller, fluid component. Complicated or hemorrhagic adrenal cysts may demonstrate variable signal intensity and may be difficult to differentiate from solid neoplasms. A limitation of MRI is, however, the poor sensitivity for detection of calcifications [
Nuclear medicine imaging, such as 131I-MIBG scintigraphy, may be used for the differentiation of an adrenal pseudocyst from a cystic pheochromocytoma, apart from the pertinent biochemical investigations, which should consist of a comprehensive preoperative hormonal and functional evaluation (serum vanyl-mandelic acid, adrenocorticotropic hormone, and cortisol levels) [
The differential diagnosis of an adrenal pseudocyst essentially includes all the upper abdominal space-occupying lesions, such as splenic, hepatic, and renal cysts, as well as mesenteric or retroperitoneal cysts, urachal cysts, and solid adrenal tumours [
In excised specimens, the exact nature will be determined also with the aid of immunochemistry, as the adrenal pseudocyst’s wall will contain adrenal cortical tissues, confirmed by A103 (Melan-A). Furthermore, a strong expression of factor VIII-related antigen, laminin, and CD34 will be apparent, in the absence of epithelial membrane antigen or keratin expression, suggesting additionally a vascular origin of these lesions [
The treatment of choice for adrenal pseudocysts depends on several factors. According to the current treatment management, it is suggested that surgical excision should be done in all adrenal lesions > 5 cm, suspicious of malignancy, and functioning adrenal pseudocysts. However, it is recommended the excision of lesions even smaller, for example, 4 cm in size, if suspicious of malignancy. In lesions < 4 cm, a repeat CT scan 3 months later is usually advised, with a follow-up period at least for 18 months [
Adrenal pseudocysts are rare lesions that may cause infrequent morbidity. Symptoms are usually atypical and depend on the pseudocyst size. Since the reported symptoms are vague due to the location of these lesions, a high suspicion index of the surgeon is required for the prompt diagnosis as well as suitable laboratory studies and imaging techniques. However, it has to be mentioned that the final diagnosis will be provided by pathology examination of the final specimen after the procedure. There are various ways that these cysts may be managed, which depend on the characteristics of the lesions, the surgeon’s experience, and local resources, while the main treatment strategy remains the same in the last decade.
The authors declare that there are no conflicts of interest.