Abdominal Wall Metastasis After Open Resection of an Adrenocortical Carcinoma
W. M. U. van Grevenstein,1 R. R. de Krijger,2 W. W. de Herder,3 H. W. Tilanus1 and H. J. Bonjer1 From the Departments of 1 Surgery, 2 Pathology and 3 Internal Medicine, University Hospital Rotterdam, Rotterdam, The Netherlands
Eur J Surg 2001; 167: 871-873
INTRODUCTION
Metastasis or recurrence of a tumour in the abdominal wall after open surgery is rare, and has been reported mostly after resection of colorectal carcinoma and hepatocellular carcinoma. In recent years an increasing number of abdominal wall recurrences after laparo- scopic resection of malignant colorectal, liver, gall- bladder, ovarian, and pancreatic tumours has been documented. Adrenocortical carcinoma is a tumour with a poor prognosis, though surgery can be curative when extent of disease is limited. Whether the resection will be an open or a laparoscopic intervention depends on the size of the tumour and its invasion into surrounding structures. Histological distinction be- tween benign and malignant adrenocortical tumours can be done with the Van Slooten index (7). We present a case of metastatic recurrence of an adrenocortical carcinoma in the abdominal surgical scar 4.5 years after open resection.
CASE REPORT
A 65-year-old woman was admitted to our hospital in December 1994 for evaluation of a tumour in the right hypochondrium. Except for mild pain in the upper abdomen, there were no other symptoms. Physical examination was unremarkable. Ultrasonography of the abdomen showed a tumour 15 cm in diameter located in the right hepatic lobe, which was confirmed by computed tomography. Percutaneous incisional biopsy showed a malignant epithelial tumour, with a histological appearance that was compatible either with a hepatocellular carcinoma or with a metastasis of a poorly differentiated carcinoma. Bone-scintigraphy and computed tomography of the chest showed no metastases. During operation, through a right subcostal incision the mass was found to be a tumour of the right adrenal gland. During removal of the tumour the capsule ruptured and there was some spill. Histological analysis of the tumour showed an adrenocortical
carcinoma with pseudocapsular invasion. The histo- logical index of this tumour was 25.1, based on the seven variables described by Van Slooten et al. (7). No adjuvant chemotherapy was given. The patient was discharged from hospital nine days postoperatively.
In July 1999, the patient noticed a lump in the abdominal scar, which was painful when she bent down. A solid, uninflamed and non-ulcerated nodule 5 cm in diameter was found on physical examination. Fine needle aspiration biopsy showed atypical epithe- lial cells consistent with metastatic adrenocortical carcinoma. Computed tomography of the abdomen showed a subcutaneous nodule 4 cm in diameter in the subcutaneous fat, but no other metastases (Fig. 1). Computed tomography of the chest was unremarkable. The tumour was resected radically in continuity with the external and internal oblique muscles. Histological examination was consistent with adrenocortical carci- noma (Fig. 2). All resection margins were free of tumour, but there was vascular invasion. No adjuvant
therapy was given. During 12 month follow up, there were no signs of recurrence.
DISCUSSION
The prognosis of adrenocortical carcinomas, which make up 0.02% of all carcinomas, is dismal. About 20%-40% of the patients have metastases at the time of presentation, and the overall 5-year survival varies from 19% to 35%. When there is localised disease at the time of initial operation, the 5-year survival may be higher, up to 40%-50%.
Initially, this tumour was considered to be a hepatocellular carcinoma located in the right hepatic lobe. Microscopic examination of a percutaneous biopsy was compatible with this diagnosis. Revision of the slides showed normal liver cells that were not attached to the tumour cells, allowing the alternative diagnosis of a hepatic metastasis of a poorly differ- entiated carcinoma or a primary tumour in a neigh- bouring organ, such as an adrenocortical carcinoma. Histological differentiation of adrenocortical carcino- ma and hepatocellular carcinoma may be difficult, because in both cases the tumour cells are polymorphic with abundant eosinophilic cytoplasm and atypical nuclei. Hepatocellular carcinomas usually have cen- trally-placed nuclei and adrenocortical carcinomas have eccentric nuclei.
A useful tool to differentiate between malignant and benign adrenocortical tumours is the Van Slooten index, which is based on a summation of seven different histological variables, yielding a maximum of 28.4. Mitoses and regressive changes have the highest discriminating value. In addition to these
variables, the weight and size of the tumour are also important. Tumours weighing more than 50 g and more than 6 cm in diameter are more likely to be malignant. In the study by Van Slooten et al., more than 96% of all adrenocortical tumours combined a high index number with a heavy tumour weight (7).
The effect of adjuvant therapy with mitotane (o,p- DDD) in patients with adrenocortical carcinoma is controversial. A recent study of Wajchenberg et al. concluded that mitotane has been the only drug that has proved to be effective in treating patients with metastatic disease, but that its usefulness is limited by its severe toxicity and little evidence that it prolongs life (8). Wooten and King reviewed the English publications from 1952 to 1992 and found 64 reports (551 patients) on the use of mitotane. Only one-third (194 of 551) at least partially responded to mitotane. They concluded that surgery remains the treatment of choice for patients with limited disease, but 68% of these tumours are discovered too late for curative resection. Treatment of metastatic disease with mito- tane has limited success (10).
Abdominal wall metastases after open resection of colorectal cancer occur in 0.6%-2.5% of all patients (2, 5, 6). A number of variables affects the incidence of recurrences, including stage of the tumour and extent of manipulation during operation. To our knowledge, abdominal wall recurrence after open resection of an adrenocortical carcinoma has not been described before. There are two reported cases of recurrences after laparoscopic resection of an adrenocortical carcinoma. Hamoir et al. reported a case of massive peritoneal recurrence six months after laparoscopic resection of a ruptured adrenocortical carcinoma with a diameter of 12 cm (4). The other case was described by Foxius et al. and had a peritoneal recurrence six months after laparoscopic adrenalectomy for a supposed Conn’s adenoma 2 cm in diameter (3).
Because solitary recurrences of adrenocortical can- cer have not been described after laparoscopic surgery, one might suggest that CO2 insufflation of the abdomen causes spread of the tumour cells with subsequent peritoneal carcinomatosis. In experimental studies, gasless laparoscopy has been shown to be associated with fewer tumour deposits than laparoscopy with CO2 insufflation (1). Transportation of the tumour cells as aerosols has been suggested as a cause of this. However, in another experimental study aerosolisation of tumour cells occured only in animals with a large tumour burden (9). The impact of CO2 insufflation on the spread of tumour cells therefore remains unre- solved.
Our patient had a non-functioning adrenocortical carcinoma. During operation the tumour ruptured, which may have caused the abdominal wall recurrence
after 4.5 years. Unlike locoregional recurrences after resection of adrenal tumours, this recurrence was discovered after 4.5 years, which is a relatively long interval for this type of tumour.
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Submitted December 1, 2000; submitted after revision March 9, 2001; accepted March 12, 2001
Address for correspondence:
W. M. U. van Grevenstein Department of Surgery University Hospital Rotterdam Dr. Molewaterplein 40 NL-3015 GD Rotterdam The Netherlands Telephone: +31 10 4639222
Fax: +31 104635058
E-mail: vangrevenstein@hotmail.com