We report an unusual case of idiopathic unilateral adrenal haemorrhage (AH) in a 55-year-old patient. This rare case had two characteristics that made it worth of report. First, idiopathic adrenal haemorrhage is very uncommon, and second it was presented as a huge, 23 cm diameter and 2,123 gr weight, “silent” adrenal mass. It is important to distinguish a benign lesion like this from a neoplasm, although we were not able to identify it preoperatively and the diagnosis was only made after the excised specimen was examined by a group of experienced histopathologists. Only a few similar published cases, to our knowledge, are described in the worldwide literature and even fewer of this size.
Adrenal haemorrhage (AH) is an uncommon condition more frequently presented in adolescents than in adults (7 : 1). Symptoms and physical findings are not specific and vary among patients. Idiopathic, unilateral AH is a rare entity that either may have an acute presentation (e.g., idiopathic adrenal rupture) or may present as an asymptomatic adrenal mass, as has been reported by other series [
The patient was admitted to our department for the evaluation of a palpable mass in the right side of his abdomen. His past medical history and any medication he ever had were irrelevant with the current problem. Physical examination revealed a palpable mass, approximately 20 × 20 cm in size, occupying an area from the upper to the lower right abdominal quadrantus. No pain or tenderness around the mass was reported and the patient mentioned only some discomfort when he was lying at night.
His vital signs were within the normal range and the laboratory data showed mild anaemia (haemoglobin: 10.9 g/dL), WBC 8190 × 109/L, without any hepatic, renal, or adrenal dysfunction from the blood or urine examinations he had.
Moreover, his serum tumor markers and coagulation function were all within normal values.
Contrast-enhanced computed tomography (CT) showed a huge, well-circumscribed, 23 × 20 cm, cystic lesion between liver and right kidney with the enhancement of the capsule around the tumor.
No liver metastasis or lymph node swelling was noted. The gadolinium-enhanced magnetic resonance imaging (MRI) that followed showed a huge multicystic lesion, in the region of the right adrenal gland, with a thick capsule containing several septations especially in the periphery of the lesion (Figures
Coronal plane of patients preoperative MRI scan, demonstrating the right quadrant huge mass.
Patient’s MRI T1-weighted image with characteristic high-intensity signal from the mass.
Patient’s MRI T2-weighted image with heterogeneously low signals.
The abnormality deviated the liver upwards, the right kidney downwards and posteriorly, the pancreas to the left, and the abdominal aorta and IVC were deviated to the left. The vascularity of the lesion was not clearly demonstrated, but there were large venous structures peripheral to the lesion draining into the IVC.
The previously described findings were discussed in the MDT meeting and the decision for surgery was made.
During the 3-hour exploratory laparotomy that followed the lesion was carefully dissected from the surrounding structures (i.e., liver, pancreas, and right kidney) and excised along with the surrounding fatty tissues. The lesion was firmly attached to the IVC and right renal vein, so a full isolation of both was necessary and successfully performed (Figures
Intraoperative image showing the close relation between IVC and the mass.
Intraoperative image during lesion dissection.
The histopathology report described a yellow-whitish lesion with traces of normal adrenal gland tissue and appearances of adrenal haemorrhage. They reported that the lesion was consisted entirely of blood clots, fibrinoid debris, and ghost outlines of proexisting vessels. Finally, they noted that it was covered by a fibrous pseudocapsule and no evidence of malignancy was noted. The final diagnosis for this patient was idiopathic adrenal haemorrhage.
Adrenal haemorrhage is a rare yet potentially life-threatening event that occurs both in traumatic conditions and in a variety of nontraumatic conditions. To our knowledge, it is an autopsy finding in most of the reported cases [
Clinical manifestations of adrenal haemorrhage can vary widely depending on the degree and rate of haemorrhage, as well as the amount of adrenal cortex compromised by haemorrhage, as similar cases have reported [
According to the underlying cause, clinical presentation varies and symptoms may include abdominal or flank pain, nausea, vomiting, weakness, weight loss, or mental confusion (adrenal insufficiency). Those are often accompanied with physical findings of fever, hypotension, abdominal tenderness or distension, and a palpable abdominal mass, as discussed elsewhere [
The laboratory findings also vary and may include a falling haematocrit level, leukocytosis, or electrolytes abnormalities [
Imaging assists and complements the clinical and endocrine evaluation of adrenal masses. Various studies showed that, although ultrasonography is a fast, low-cost, and widely available method, it is usually limited to infants and children [
Some have proposed a descent approach to incidentally discovered adrenal mass, which includes assessing the mass size, determination of whether is solid or cystic, and excluding biochemically active tumors by all the appropriate laboratory studies [
Adrenal haemorrhage is a rare cause of adrenal mass and is generally associated with trauma, infection, or bleeding diathesis. Only a few isolated cases of similar pathology (idiopathic adrenal haemorrhage) exist in the literature and nearly all of them were postoperatively diagnosed as such. Though rare, signs of adrenal haemorrhage may include an asymptomatic adrenal mass and should be considered in the differential diagnosis of this problem.