journal homepage: Keywords : gallbladder cancer, metastasis, muscle 1,2 *

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1 台灣癌症醫誌 (J. Cancer Res. Pract.) 27(6), , 2011 Case Report journal homepage: Skeletal Muscle Metastasis of Gallbladder Cancer Chia-Lin Chang 1, Su-Peng Yeh 1,2, Chang-Fang Chiu 1,2, Li-Yuan Bai 1,2 * 1 Division of Hematology and Oncology, China Medical University Hospital, Taichung, Taiwan 2 College of Medicine, China Medical University, Taichung, Taiwan Abstract. Gallbladder cancer (GBC) is generally diagnosed at an advanced stage and carries a poor prognosis. The liver and regional lymph nodes are the most commonly invaded organs. Here we present a 69-year-old woman who was diagnosed to have a gallbladder adenocarcinoma and distant muscle metastasis. The patient responded poorly to chemotherapy and unfortunately died 3 months after the diagnosis. This report illustrates an unusual metastasis of GBC to the skeletal muscle, which to the best of our knowledge has not yet been reported in the English literature. 病例報告 Keywords : gallbladder cancer, metastasis, muscle 膽囊癌合併骨骼肌轉移 張嘉麟 1 葉士芃 1,2 邱昌芳 1,2 白禮源 1,2 * 1 中國醫藥大學附設醫院血液腫瘤科 2 中國醫藥大學醫學系 中文摘要膽囊癌在被診斷時通常已進行至晚期, 且預後不佳 肝臟和區域淋巴結是最常見被侵犯的器官 在此, 我們報告一位 69 歲女性病患膽囊癌合併遠處骨骼肌肉轉移, 此位病 人對化學治療反應不佳, 不幸在診斷三個月後去世 這個病例描述了一個不常見的膽囊癌合併遠處骨骼肌轉移, 此種轉移在英文文獻中, 據吾人所知, 尚未被報告過 關鍵字 : 膽囊癌 轉移 肌肉 INTRODUCTION Gallbladder cancer (GBC) is a relatively uncommon disease with a poor prognosis. Approximately one third of patients have metastatic disease at the time of diagnosis, with the liver and regional lymph nodes most frequently affected. There have been a few reports describing cutaneous metastases from gallbladder cancer, but to the best of our knowledge, gallbladder cancer metastasizing to skeletal muscle has never been reported. Therefore we present a patient with gallbladder cancer who had skeletal muscle metastasis. CASE REPORT A 69-year-old woman presented with a painful, progressively enlarging mass in her left lower neck.

2 264 C. L. Chang et al./jcrp 27(2011) She also complained of bilateral leg edema and a 9 kg body weight loss during the past 3 months. The patient reported neither abdominal pain nor history of abdominal surgery. On physical examination, a 1.5 cm-sized, firm, fixed, tender mass was noted on the left lower neck. Laboratory analysis showed the following results: white blood cell count 7,580/mm 3, hemoglobin 11.3 mg/dl, total bilirubin 0.5 mg/dl, aspartate transaminase 39 IU/L, alanine aminotransferase 18 IU/L, lactate dehydrogenase 257 IU/L, carbohydrate antigen 19-9 (CA 19-9) 35.8 ng/ml and carcinoembryonic antigen (CEA) ng/ml. Pathological examination of the left neck mass revealed metastatic adenocarcinoma of the lymph node composed of tumor cells with trabecular configuration, intracellular mucin content and extracellular mucin production. The tumor cells had focal positivity for cytokeratin (CK) 7, CEA and no expression of the estrogen receptor, naspsin A, CK 20, or thyroid transcription factor 1 (TTF-1) (Figure 1). Abdominal sonography showed one mixed echoic tumor, 4.0 cm 3.5 cm in diameter, within the gallbladder. A contrast-enhanced computed tomography (CT) scan revealed multiple enlarged lymph nodes in the left neck, left supraclavicular fossa, left axilla, retrocrural areas on both sides, paraaortic, paracaval, periportal areas and left common iliac region. After admission, the patient received chemotherapy with biweekly gemcitabine 800 mg/m 2 /day and oxaliplatin 85 mg/m 2 /day (GEMOX) under the diagnosis of gallbladder adenocarcinoma with multiple lymph node metastases. One day before the second course of GEMOX, she complained of right lower abdominal wall pain. Upon examination, a 2.0 *Corresponding author: Li-Yuan Bai M.D. * 通訊作者 : 白禮源醫師 Tel: Fax: lybai6@gmail.com cm-sized tender mass was vaguely-palpated in her right lower abdominal wall. The mass was fixed to the muscle, and the overlying skin was free. The main findings of another CT scan consisted of diffuse swelling of muscle in right lower abdominal wall with one enhancing metastatic lesion of 2.0 cm 2.3 cm in the muscular layer (Figure 2). Fine needle aspiration cytology from the mass revealed cohesive clusters of tumor cells with enlarged, hyperchromatic, eccentric nuclei, irregular nuclear membrane, and prominent nucleoli (Figure 3). The patient s disease did not respond to chemotherapy and she suffered from rapid disease deterioration with pleural metastasis causing pleural effusion, obstructive jaundice and massive ascites. She died 3 months after the original diagnosis. DISCUSSION GBC is a relatively uncommon disease. The incidence of gallbladder cancer in the United States in 2008 was 1.2/100,000 [1]. The leading risk factors for GBC include gallstones, female sex, advancing age, chronic inflammation and porcelain gallbladder [2]. The symptoms of GBC are usually nonspecific. At least 20% of patients of GBC are diagnosed at the time of cholecystectomy for biliary colic or cholelithiasis. Abdominal pain is the most common symptom of GBC, followed by nausea and vomiting, jaundice, anorexia, and body weight loss. Constitutional symptoms, ascites, and a palpable mass are all indicative of advanced disease status and carry poor prognosis. The primary histologic type in most cases of GBC is adenocarcinoma [3]. GBC has traditionally been associated with a poor prognosis, with a reported 5-year survival of 5-10% and a median survival of only 3-6 months from the time of diagnosis [4]. The unfortunate outcome of patients with GBC is thought to be related to the advanced stage at diagnosis, which is due both to the anatomic position of the gallbladder, and the vagueness and non-specificity of symptoms. Metastasis from

3 C. L. Chang et al./jcrp 27(2011) Figure 1. Histological findings of the neck lymph node. (A) Hematoxylin and eosin staining. Neoplastic glandular infiltration in the lymph node, suggesting metastatic adenocarcinoma. Neoplastic glands show positive staining with CK 7 (B) and CEA (C), but no staining for CK 20 (D) (100 x in A-D) GBC occurs most frequently to the liver (76% - 86%), due to lactic acid and other metabolites have also been followed by regional lymph nodes [5]. Rare sites of implicated as an explanation. It is thought that lactic metastases include the heart, orbit, central nervous acid production by skeletal muscles may prohibit the system, skin, bone, and scalp [6]. implantation and growth of tumor cells [8]. The Although more than half of our body mass con- prognosis for patients with soft tissue metastasis is sists of soft tissues, the chance of metastasis from poor with survival time ranging from 1 to 19 months other primary sites to these regions is relatively slim (mean 5.4 months) after diagnosis of the metastasis [7]. Several theories have been postulated for this ob- [9]. servation. The lung, liver and bone are common sites Malignancies most apt to metastasize to the mus- for metastatic disease and have a constant steady cle are melanoma, and cancer of the kidney, lung and blood flow, whereas skeletal muscles have a variable thyroid. Other malignancies reported include gastro- blood flow subject to changing tissue pressure, which intestinal tract cancer, breast cancer, head and neck may hinder tumor cell implantation. Changes in ph cancer, and cancer of an unknown primary origin.

4 266 C. L. Chang et al./jcrp 27(2011) Figure 2. CT scan revealed one 2.0 cm x 2.3 cm enhancing mass lesion (arrow) in the muscle layer of the right lower abdominal wall Figure 3. Aspiration cytology from the abdominal mass (Liu s stain, 1000x) There have been a few articles reporting cholangiocarcinoma with skeletal muscle metastases [10-12]. A 72-year-old man with cholangiocarcinoma had metastasis to his calf muscle [10], and a 44-year-old man presenting as Budd-Chiari syndrome had disseminated venous thrombosis and multiple skeletal muscle metastases, bilateral to the buttocks and erector spinal muscles [11]. A third case was a 45-year-old man complaining of right upper quadrat abdominal pain from intrahepatic cholangiocarcinoma. He had distant skeletal muscle metastases in the thorax and buttock noted in a 2-[18F] fluro-2-deoxy-d-glucose positron-emission tomography scan [12]. GBC with distant skeletal muscle involvement must be even rare, and has never been reported. Trocar site metastasis, however has been reported in patients who underwent laparoscopic cholecystectomy for preoperatively undiagnosed GBC [13,14]. In another report, GBC directly invading the abdominal wall was noted in an 83-year-old man [15]. Neither of such invasions belonged to hematogenous spreading to skeletal muscle from a GBC. Thus, the best of our knowledge, our present paper is the first one reporting a GBC with distant skeletal muscle metastasis. In conclusion, this case illustrates an unusual presentation of GBC. Clinicians should be aware of the possibility of such rare metastases from malignancies, and should then investigate properly with imaging and histologic verification. Distant skeletal muscle

5 C. L. Chang et al./jcrp 27(2011) metastasis from GBC indicates the presence of advanced disease and is associated with a very poor prognosis. REFERENCES 1. Duffy A, Capanu M, Abou-Alfa GK, et al. Gallbladder cancer (GBC): 10-year experience at Memorial Sloan-Kettering Cancer Centre (MSKCC). J Surg Oncol 98: , Rustagi T, Dasanu CA. Risk factors for gallbladder cancer and cholangiocarcinoma: similarities, differences and updates. J Gastrointest Cancer 43: , Reid KM, Medina ARD, Donohue JH. Diagnosis and surgical management of gallbladder cancer: a review. J Gastrointest Surg 11: , Hueman MT, Vollmer CM Jr, Pawlik TM. Evolving treatment strategies for gallbladder cancer. Ann Surg Oncol 16: , Kokudo N, Makuuchi M, Natori T, et al. Strategies for surgical treatment of gallbladder carcinoma based on information available before resection. Arch Surg 138: , Kaur J, Puri T, Julka PK. Adenocarcinoma of the gall bladder presenting with a cutaneous metastasis. Indian J Dermatol Venereol Leprol 72: 64-66, Plaza JA, Perez-Montiel D, Mayerson J, et al. Metastases to soft tissue: a review of 118 cases over a 30-year period. Cancer 112: , Abed R, Grimer RJ, Carter SR, et al. Soft-tissue metastases: their presentation and origin. J Bone Joint Surg 91: , Damron TA, Heiner J. Distant soft tissue metastases: a series of 30 new patients and 91 cases from the literature. Ann Surg Oncol 7: , Yoshimura Y, Isobe K, Koike T, et al. Metastatic carcinoma to subcutaneous tissue and skeletal muscle: clinicopathological features in 11 cases. Jpn J Clin Oncol 41: , Kwon OS, Jun DW, Kim SH, et al. Distant skeletal muscle metastasis from intrahepatic cholangiocarcinoma presenting as Budd-Chiari syndrome. World J Gastroenterol 13: , Park SK, Kim YS, Kim SG, et al. Detection of distant metastasis to skeletal muscle by 18F-FDG-PET in a case of intrahepatic cholangiocarcinoma. Korean J Hepatol 16: , Yoshida T, Matsumoto T, Sasaki A, et al. Laparoscopic cholecystectomy in the treatment of patients with gallbladder cancer. J Am Coll Surg 191: , Steinert R, Nestler G, Sagynaliev E, et al. Laparoscopic cholecystectomy and gallbladder cancer. J Surg Oncol 93: , Rajagopalan S, Loudon M. Abdominal-wall abscess. N Engl J Med 362: 8, 2010.