SPONTANEOUS RUPTURE OF ADRENAL CARCINOMA DURING THE PUERPERAL PERIOD
GIOVANNI LIGUORI,* CARLO TROMBETTA, ROBERTO KNEZ, STEFANO BUCCI, ROSSANA BUSSANI AND EMANUELE BELGRANO
From the Departments of Urology (GL, CT, RK, SB, EB) and Pathology (RB), University of Trieste, Trieste, Italy KEY WORDS: adrenal glands; carcinoma; rupture, spontaneous
Spontaneous massive retroperitoneal hemorrhage from an adrenal gland is an extremely rare event and is usually fatal if unrecognized. We report a case of this critical event occur- ring in a young woman affected by a silent adrenocortical carcinoma in the postpartum period.
CASE REPORT
A 39-year-old lactating woman in the eighth month of puerperium presented with sudden onset of left abdominal pain and signs of shock. Computerized tomography (CT) showed a large (12 × 8 cm) hematoma in the retroperitoneal space around the left kidney and extending from the dia- phragm throughout the pararenal space along the entire course of the psoas muscle (fig. 1). The left adrenal gland was not visible in all CT images, so the adrenal origin of the hemorrhage was suspected.
The patient was placed under general anesthesia and a chevron incision was made. At surgical abdominal explo- ration a large hematoma was found in the retroperitoneum arising from a left adrenal gland tumor. Left adrenalec- tomy with preservation of the left kidney and drainage of the retroperitoneal hematoma were performed. To stabi- lize blood pressure during the operation 6 units blood was transfused and volume supplementation was performed. Hematoma volume was approximately 1,500 gm and oper- ative time was 180 minutes. Pathological diagnosis was surprisingly a primary highly aggressive adrenocortical carcinoma (fig. 2). The postoperative period was unevent- ful and blood pressure normalized without medication. There was no sign of endocrine dysfunction. Control total body CT and bone scintigraphy were negative for meta- Accepted for publication June 13, 2003.
* Corresponding author: Via Franca n5, 34123 Trieste, Italy (telephone: +39.040.399.4575; FAX: +39.040.399.4895; e-mail: gioliguori@libero.it).
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static lesions. The patient subsequently underwent chemo- therapy evaluation and was started on mitotane.
DISCUSSION
Adrenocortical carcinoma is a rare tumor, ranging between 0.5 and 2 cases per million population, and generally carries a poor prognosis. It displays a bimodal age distribution, with the first peak occurring before age 5 years and the second in the fourth to fifth decade of life. According to Ng and Libertino, only 62% of these tumors are functional with clinical and biochemical manifestations, while 38% are nonfunctional and, therefore, asymptomatic.1
Spontaneous massive retroperitoneal hemorrhage from an adrenal gland tumor is also rare, especially during the post- partum period.2 We do not know what triggered the adrenal gland laceration in our patient, but she had no related his- tory and was in the eighth month of puerperium, and lactat- ing. However, hematoma of the adrenal gland should always be suspected in patients with upper abdominal pain and retroperitoneal hematoma, even in the absence of trauma, stress or infection. Our case is unusual because of its rarity and presentation. A review of the literature reveals that adrenocortical tumors are infrequently encountered clini- cally and rarely hemorrhage.3 To our knowledge this is the first case of massive retroperitoneal hemorrhage as a com- plication in the postpartum period to be reported in the urological literature.
REFERENCES
1. Ng, L. and Libertino, J. M .: Adrenocortical carcinoma: diagnosis, evaluation and treatment. J Urol, 169: 5, 2003
2. O’Kane, H. F., Duggan, B., Lennon, G. and Russell, C .: Sponta- neous rupture of adrenocortical carcinoma. J Urol, 168: 2530, 2003
3. Kardar, A. H .: Rupture of adrenal carcinoma after biopsy. J Urol, 166: 984, 2001