Retrobulbar metastasis from gallbladder carcinoma after laparoscopic cholecystectomy. A case report

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Retrobulbar metastasis from gallbladder carcinoma after laparoscopic cholecystectomy. A case report Article in Tumori · November 2004 Source: PubMed

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Tumori, 91: 428-431, 2005

RETROBULBAR METASTASIS FROM GALLBLADDER CARCINOMA AFTER LAPAROSCOPIC CHOLECYSTECTOMY. A CASE REPORT Francesco Puglisi1, Palma Capuano1, Antonia Gentile2, Pierluigi Lobascio1, Silvana Russo2, Gennaro Martines1, Giuseppe Lograno1, and Vincenzo Memeo1 1Unit

of General Surgery and Liver Transplantation, 2Department of Pathology and Genetics, University of Bari, Italy

Extra-abdominal metastases from gallbladder cancer are very rare; the sites outside the abdomen most frequently affected are the skin, bone and central nervous system. In the literature, only one case of orbital metastasis from gallbladder cancer has been reported, in a patient previously treated by open cholecystectomy. We report the case of a 53-year-old woman who underwent a laparoscopic cholecystectomy for symptomatic gallbladder stones. Postoperative histological examination revealed an unsuspected gallbladder adenocarcinoma. One

month later she came to our observation after having developed diplopia and ophthalmic pain due to an orbital metastasis. We decided not to perform a surgical second look because of the already rapid dissemination of the malignant tumor. The few cases of uncommon gallbladder cancer metastases after laparoscopic cholecystectomy described in the literature are discussed, as well as the possible role of laparoscopy in the dissemination and localized seeding of malignant cells.

Key words: diplopia, extra-abdominal metastasis, gallbladder carcinoma, laparoscopic cholecystectomy, orbital metastasis.

Introduction

Gallbladder cancer accounts for 5% of malignant tumors1. Before Misra et al. described their experience, no other cases of orbital metastasis from gallbladder carcinoma had been reported2. In fact, the most frequent sites of metastasis from this kind of neoplasm are intraabdominal and include the liver and para-aortic lymph nodes. Extra-abdominal metastases are rare: the few cases reported affected the skin, bone and central nervous system3-5. Laparoscopic cholecystectomy has been widely accepted as the procedure of choice for benign gallbladder diseases such as gallbladder stones and leiomyoma. Some surgeons have claimed that laparoscopic cholecystectomy is contraindicated in gallbladder cancer because of the risk of neoplastic dissemination by the laparoscopic tools along the endoscopic tract, or of intraabdominal dissemination; the laparoscopic approach could also compromise the prognosis in cases of unsuspected gallbladder cancer6-11. Case report

We observed a 53-year-old woman who had undergone a laparoscopic cholecystectomy in another surgical unit for symptomatic gallbladder stones confirmed by abdominal ultrasonography (US). The gallbladder was retrieved intact, without gross bile spillage. Histological examination of the gallbladder revealed an unsuspected undifferentiated gallbladder carcinoma consisting predominantly of cells with vesicular nuclei and

prominent nucleoli and some anaplastic cells. In the gallbladder the neoplastic cells were arranged in trabecular structures and in other areas the tumor exhibited squamous differentiation. An inflammatory infiltrate with a rich content of polymorphonuclear leukocytes and lymphocytes was present in the stroma, admixed with neoplastic cells (Figures 1 and 2). After about 30 days, the patient came to our observation for a surgical second look to evaluate the indication for en bloc resection of the gallbladder bed and bile duct together with lymphadenectomy. Total-body CT scan showed only the presence of para-aortic lymph nodes and some small retropancreatic nodes. The patient told us she had developed diplopia some days before and had discovered a small mass in the right supraclavicular region. This nodule was surgically removed and histologically examined, resulting in a diagnosis of lymph node metastases from gallbladder carcinoma. The cancer cells in the lymph node were similar to those of a primary gallbladder tumor (Figure 3). In the lymph nodes certain morphological features can be confused with those of malignant lymphomas, but in this case the immunohistochemical identification of cytokeratin and CEA excluded a lymphoma. Serum biochemistry revealed increased levels of the tumor markers alpha-fetoprotein, tissue polypeptide antigen, and carcinoembryonic antigen. The patient underwent ophthalmic and US orbital examinations which demonstrated an exophthalmus in the right eye due to an intra- and extraconal irregular retrobulbar mass involving the lateral rectus muscle and

Correspondence to: Dr Francesco Puglisi, DETO Sezione di Chirurgia Generale e Trapianto di Fegato, Università – Policlinico di Bari, Piazza G Cesare 11, 70100 Bari, Italy. Tel +39-080-5592975, +39-080-5592239; fax +39-080-5478735; e-mail [email protected] Received December 13, 2004; accepted April 22, 2005.

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Figure 1 - Gallbladder carcinoma (hematoxylin-eosin, x200). In the center, trabeculae of well-differentiated neoplastic squamous cell elements. At the margins, medium-sized poorly differentiated neoplastic cells.

Figure 2 - Immunohistochemistry (CK pool, x200). The neoplastic squamous cells in the center and less well-differentiated cells at the margins are positive for the anticytokeratin pool antibodies. There is also intense positivity of the cell elements lining a normal glandular introflexion (top right).

displacing the optic nerve. MRI and CT scans of the brain and orbit confirmed the presence of an enhancedcontrast orbital mass in the right eye compatible with metastases from the tumor (Figure 4). Orbital fine-needle aspiration biopsy (FNAB) confirmed the metastatic nature of the endo-ocular mass. Unfortunately, images of this biopsy are not available. In view of the disseminated neoplastic pathology, we decided not to operate the patient and left her under oncological care. At present, seven months after the laparoscopic cholecystectomy, the patient is alive and undergoing chemotherapy with gemcitabine and cisplatin; in addition, she has undergone radiotherapy to the ocular lesion. Nevertheless, there is ongoing progression of the tumor. Discussion

Figure 3 - Histological pattern of metastatic supraclavicular node.

Gallbladder carcinoma is a relatively rare tumor with an incidence of only 0.3-1.5% in patients undergoing cholecystectomy for benign disease12-13. It is difficult to make an early diagnosis of gallbladder carcinoma because the symptoms are indistinguishable from those of gallbladder stones; indeed, the association of the two diseases is highly frequent, ranging from 60% to 92%14. Most cases of gallbladder carcinoma are therefore iden-

tified during cholecystectomy procedures performed for gallbladder stones, or at histological examination of the removed organ. Apart from tumors in stages Tis and T1, for which cholecystectomy is considered quite sufficient, it is recommended to perform resection of the gallbladder bed

430

Figure 4 - CT scan shows the right retrobulbar metastatic mass.

and locoregional lymphadenectomy for locally advanced disease (T2 e T3). Patients with a T2 gallbladder carcinoma detected incidentally at cholecystectomy have a better survival if a second, radical operation is performed15 -17. The advent of the laparoscopic technique has undoubtedly increased the number of cholecystectomy procedures performed and hence the number of unsuspected carcinomas detected in the removed organ. The numerous reports describing the development of metastases in the abdominal wall, at the level of the incision made to extract the gallbladder or at the site of the trocars, have raised the suspicion that laparoscopic cholecystectomy may be potentially risky in cases of unsuspected gallbladder carcinoma18,19. Several hypotheses have been proposed to explain the worse prognosis of gallbladder carcinoma treated by laparoscopy. For instance, direct tumor spread to the anterior abdominal wall may occur during removal of the gallbladder. The laparoscopic instruments used to mobi-

F PUGLISI, P CAPUANO, A GENTILE ET AL

lize the gallbladder during surgery may be inoculated with tumor cells and may seed the anterior abdominal wall on removal20,21. In addition, tumor cells may undergo aerosolization at insufflation of the peritoneum, thus favoring peritoneal dissemination. The case described by us is remarkable because of the rare site of the metastasis: the ocular orbit. Orbital metastases are a rare occurrence in any case, manifesting with diplopia, proptosis, decreased vision, red eye, photophobia, and frequently a combination of these symptoms22. The first, historical description of altered vision due to orbital metastases was made by Horner in 186423. The solid tumors that most frequently develop metastases in the orbit are breast and lung carcinoma (30%), followed by urogenital and gastrointestinal tumors. Patients with orbital metastases have a mean survival of 1.3 years, regardless of the site of the primary tumor. There are few therapeutic options, considering that it is a question of distant spread of a solid tumor. FNAB is an efficacious, relatively uninvasive method to confirm the diagnosis24,25. Prior to our report, only one other case of orbital metastases from gallbladder carcinoma had been reported, which appeared one month after open cholecystectomy and was associated, as in our case, with supraclavicular lymph node metastases. As the laparoscopic cholecystectomy procedure was not performed by us, we have no detailed information about it (was there any perforation of the gallbladder, what protection was used during extraction, etc.). In any case, the brief interval between the laparocholecystectomy and the appearance of the orbital and supraclavicular metastases makes it likely that they were an expression of a biologically highly aggressive tumor rather than the consequence of the laparoscopic resection. The rapid progression of the disease in the two reported cases, despite the administration of chemotherapy, is a demonstration of the aggressive nature of advanced gallbladder carcinoma and its poor prognosis.

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