SPONTANEOUS RUPTURE OF ADRENOCORTICAL CARCINOMA
H. F. O’KANE, B. DUGGAN, G. LENNON AND C. RUSSELL
From the Urology Department, Altnagelvin Hospital, Londonderry and Department of Endocrine Surgery, Royal Victoria Hospital, Belfast, Northern Ireland
KEY WORDS: carcinoma, adrenal cortical; neoplasm metastasis; adrenal glands; rupture
We report a case of spontaneous rupture of a primary adrenocortical carcinoma. To our knowledge this is only the second such case described in the literature.
CASE REPORT
A 52-year-old male presented with acute severe abdominal pain and signs of shock. Computerize tomography (CT) of the abdomen showed a large retroperitoneal mass on the left side (fig. 1). Contrast enhanced CT suggested a mixed density lesion originating from the left adrenal gland with hemor- rhage within the mass (fig. 2). Preliminary diagnosis was ruptured adrenal carcinoma.
At exploration via a thoracoabdominal approach a large tumor mass and associated hematoma were identified in the retroperitoneum extending from the diaphragm to the left renal pedicle. The mass was resected together with the spleen, with preservation of the left kidney and pancreas. Small nodules found within the liver were confirmed on fro- zen section to represent metastatic adrenal carcinoma. Histological examination of the resected specimen confirmed primary adrenocortical carcinoma. Postoperative recovery was uneventful and the patient subsequently received mito- tane chemotherapy.
DISCUSSION
Primary adrenocortical carcinoma is an extremely rare tumor with an annual incidence of 1 to 2 cases per million population. The tumor displays a bimodal age incidence, Accepted for publication June 21, 2002.
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peaking in the first and fourth decades of life with an appar- ent worse prognosis associated with the latter. These tumors may be classified as functional when there are clinical and biochemical manifestations of hormone production present, or nonfunctional when no excess hormone release can be demonstrated.1 Therefore, the patient may present with symptoms suggestive of excess hormone release such as in Cushing’s syndrome, the virilization syndrome or the femi- nization syndrome, or with other nonspecific symptoms such as anorexia, abdominal pain and weight loss.
Presentation of a solid tumor following spontaneous rupture is extremely rare. Spontaneous retroperitoneal he- matoma has previously been reported as the mode of presen- tation for adrenal adenoma, myelolipoma and pheochromo- cytoma.2 A review of the literature revealed only 1 previously reported case of spontaneous rupture of primary adrenal adenocarcinoma, and in that case rupture occurred following fine needle biopsy.3
REFERENCES
1. Wajchenberg, B. L., Albergaria Pereira, M. A., Medonca, B. B., Latronico, A. C., Campos Carneiro, P., Alves, V. A. et al: Adrenocortical carcinoma: clinical and laboratory observa- tions. Cancer, 88: 711, 2000
2. Machuca Santa Cruz, J., Julve Villalta, E., Galacho Bech, A., Perez Rodriguez, D., Quinonero Diaz, A., Alonso Dorrego, J. M. et al: Spontaneous retroperitoneal haematoma: our experi- ence. Actas Urol Esp, 23: 43, 1999
3. Kardar, A. H .: Rupture of adrenal carcinoma after biopsy. J Urol, 166: 984, 2001