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DOI 10.3966/181020932016061402003 Case Report Lung Adenocarcinoma Metastatic to an Indirect Inguinal Hernia Sac: A Case Report and Literature Review Hui-Yu Lin1 Chun-Hou Liao1,2 Yu-Wei Chou1,* An inguinal hernia is a common medical condition in daily practice. Indeed, the lifetimeprevalence of inguinal hernias is 47% in patients > 75 years of age, but it is rare to have metastatic cancer of an inguinal hernia sac. Most cases of intraperitoneal malignant inguinal hernias arise from the gastrointestinal tract. Extraperitoneal inguinal metastases most commonly occur in males with testicular cancer; there have been no reported cases that have metastasized from lung adenocarcinoma. Here we report a 77-year-old male with possible cancerous peritonitis that led to the finding of metastases in a hernia sac. The patient presented with disease progression after chemo-radiation therapy with multiple metastases to the liver and bone. This report highlights the importance of a pathologic evaluation of the inguinal hernia sac in preventing misdiagnosis. Key words: Adenocarcinoma metastasis, Hernia sac, Inguinal hernia, Lung cancer INTRODUCTION Case report Inguinal hernias are one of the most common A 77-year-old Asian male with type II diabe- medical conditions in surgical practice, but it is tes mellitus and essential hypertension for decades rare to have metastatic cancer in an inguinal her- presented with a right inguinal painless mass for nia sac. Although few cases of metastatic cancer > 1 year. He reported no change in bowel habits. in an inguinal hernia sac have reported in review He also reported right groin pain and right lower articles, no metastases from lung adenocarcinoma abdomen tenderness, which first developed 1 week have been reported. Based on the report herein, prior to an evaluation by a urologist. The physi- lung adenocarcinoma metastasis should be con- cal examination revealed a painful, regularly- sidered when a patient has a clear history of lung shaped, fixed, non-reducible bulging mass in the cancer, even when the origin is distant from the right inguinal area. The tentative diagnosis was an inguinal area. inguinal hernia. To rule out an incarcerated inguinal hernia, an abdominal CT was arranged, which Division of Urology, Department of Surgery, Cardinal Tien Hospital, New Taipei, Taiwan1 School of Medicine, Fu-Jen Catholic University, New Taipei, Taiwan2 Submitted March, 18, 2014; final version accepted December, 01, 2015. *Correspondence author: Yu-Wei Chou ([email protected]) 輔仁醫學期刊 第 14 卷 第 2 期 2016 77 Hui-Yu Lin Chun-Hou Liao Yu-Wei Chou showed a hepatic hemangioma and right inguinal patient presented with disease progression after hernia with a small segment of small bowel in the chemo-radiation therapy with multiple metastases herniation sac (Fig. 1). Intra-operatively, indirect to the liver and bone. The patient died after 3 years hernia sac should be smooth in texture, but this of follow-up. case with multiple pieces of yellow granule-like tissue seeding the internal ring were noted, it feels grainy and which originated from the abdomen and was confirmed by opening the sac, The opened sac extruded ascites in the form of turbid, serous fluid. Surprisingly, special immunostaining of the sac showed the following: thyroid transcription factor (TTF)-1 and cytokeratin (CK)-7 were strongly positive; CK-20 was focally positive; Figure 1. Right inguinal hernia contents with small bowel loops (arrow). CDX-2 was negative; and PAS and mucin staining were also positive. Further study showed that among tumor markers, only CEA was elevated. Stool occult blood testing was negative. PSA was within the normal range. Plain chest films showed a patch with faint opacity over the right upper lung. Chest computed tomography revealed a 7 x 4.4 cm mass in the right lung. Bronchoscopy demonstrated stenosis of the orifice of the right B2 bronchus due to external compression. Biopsy and brushing were performed over the mucosa. The patient underwent video-as- Figure 2. Special staining of lung adenocarcinoma of inguinal metastases was positive for TTF-1. sisted thoracic surgery (VATS) decortication with a pleural biopsy, and the pathology showed poorly differentiated adenocarcinoma metastatic to the pleura. The pathologist reviewed the microscopic slides of the bronchial brushing and biopsy, and found extreme similarity between the morphology of adenocarcinoma and the ingunal hernia sac. Based on medical history of the patient and clinical findings, the metastatic adenocarcinoma was thought to originate from the right lung. Although peritoneal signs were limited to the right lower quadrant, the final diagnosis was right lung adeno- Figure 3. Special staining of lung adenocarcinoma metastatic to the inguinal area was CK 7-positive. carcinoma with metastasis (stage T4N0M1). The 78 Fu-Jen Journal of Medicine Vol.14 No.2 2016 Lung adenocarcinoma in inguinal hernia sac losa cell tumors, gastric cancer, and retroperitoneal liposarcomas have been diagnosed incidentally within the inguinal canal based on published reports[1,7,10]. Most intraperitoneal malignant inguinal hernias arise from cancers of the gastrointestinal tract; it is not uncommon to find advanced sigmoid colon cancer metastases within an inguinal hernia sac[2]. In extraperitoneal cancers, testicular cancer is the most common origin of extraperitoneal inFigure 4. Special staining of lung adenocarcinoma metastatic to the inguinal area was CDX-2negative (400x). guinal metastases in males, while ovarian cancer is the origin of most common extraperitoneal inguinal metastases in females[9]. Prostate adenocarcinoma with metastases to the inguinal sac after radical prostatectomy have also been reported[16]. These cancers have direct contact with the posterior wall of the inguinal canal, which made local tissue invasion more likely[5]. Distal lung metastases to the inguinal canal has not been reported, thus this is the first reported case of metastatic lung cancer to the inguinal canal. In this case, the use of CK7/ CK20 and TTF-1 may be helpful in distinguishing between primary and metastatic pulmonary adeno- Figure 5. Special staining of lung adenocarcinoma metastatic to the inguinal area was TTF-1positive (400x). Discussion carcinoma[11]. Primary pulmonary adenocarcinoma would be expected to show CK7+/CK20-/TTF1+[3,6]. TTF-1 is very important in distinguishing between primary and metastatic lung adenocarcinoma because most primary lung adenocarcinomas are TTF-1-positive[4,11-15]. An inguinal hernia is a common medical con- The patient might have had cancerous peri- dition with a lifetime prevalence as high as 47% tonitis, which would enable the cancer cells to [8] in patients > 75 years of age . Malignant masses spread to the hernia sac. To distinguish cancerous in the inguinal canal appear in < 0.5% of excised peritonitis from gastrointestinal adenocarcinoma, [1] sacs . Accordingly, the metastatic tumor described CDX-2 was stained and was shown to be negative. in this case can be classified as a saccular hernia Therefore, origin from the lung was a greater pos- [10] sac tumor . The most common cause of saccular tumors is metastases from abdominal malignan[1] sibility[6]. The systemic therapy is the primary approach cies . Malignant mesotheliomas, cecal adnocarci- to the management of metastatic lung cancer, and nomas, colon cancer, prostate cancer, adult granu- therapy directed against specific sites of disease 輔仁醫學期刊 第 14 卷 第 2 期 2016 79 Hui-Yu Lin Chun-Hou Liao Yu-Wei Chou may also play an important role. In this situation, immunostaining in separating pulmonary ad- surgical resection or definitive radiation therapy of enocarcinomas from colonic adenocarcinomas. the metastatic disease may produce durable ben- Am J Clin Pathol 1994; 102: 764–767. [17] efit . The most frequent sites of such metastases are the brain and the adrenal gland [18] 7. Wilson JM, Duncan AN, Ignjatovic A, et al. . The final Inguinal herniae: valuable clues to concurrent outcomes in the treatment of patients with lung abdominal pathology. A series of case stud- cancer depends upon the cell type (non-small cell ies describing unusual findings in “routine” versus small cell), tumor stage, molecular charac- hernia operations which demonstrate the need teristics, and an assessment of the patient's overall for thorough surgical training. Intl J Surg Case medical condition [19] . This report highlights the Reports 2011;2:198-200. importance of evaluation of inguinal hernia sacs in 8. Kingsnorth A, LeBlanc K. Hernias: inguinal aiding the correct diagnosis and preventing misdi- and incisional. Lancet 2003;362:1561-1571. agnosis. 9. Yang XJ, Zheng FY, Xu YS, et al. Ovarian cancer initially presenting with isolated ipsilateral Reference superficial inguinal lymph node metastasis: a case study and review of the literature. J Ovar- 1. Nicholson CP, Donohue JH, Thompson GB, et ian Res 2014;7:20. al. A study of metastatic cancer found during 10.Yokoyama N, Hidehiro YS, Hatakeyama YK: inguinal hernia repair. Cancer 1992; 69:3008- An inguinal hernia sac tumor of extrahepatic 3011. cholangiocarcinoma origin. World J Surg On- 2. Tan GY, Guy RJ, Eu KW. Obstructing sigmoid cancer with local invasion in an incarcerated col 2006; 4: 13. 11.Chhieng DC, Cangiarella JF, Zakowski MF, et inguinal hernia. ANZ J Surg 2003;73:80-82. al. Use of thyroid transcription factor 1, PE-10, 3. Lau SK, Desrochers MJ, Luthringer DJ. and cytokeratins 7 and 20 in discriminating be- Expression of thyroid transcription factor-1, tween primary lung carcinomas and metastatic cytokeratin 7, and cytokeratin 20 in bron- lesions in fine-needle aspiration biopsy speci- chioloalveolar carcinomas: an immunohisto- mens. Cancer 2001; 93: 330-336. chemical evaluation of 67 cases. Mod Pathol 2002;15:538-542. 12.Holzinger A, Dingle S, Bejarano PA, et al. Monoclonal antibody to thyroid transcription 4. Anderson SS, Langel DJ, Gown AM, et al. factor-1: production, characterization, and use- Thyroid transcription factor-1 (TTF-1) is a fulness in tumor diagnosis. Hybridoma 1996; more sensitive lung carcinoma marker than 15: 49–53. surfactant APOA1 [abstract]. Lab Invest 1998; 78: 171. 5. Vagnoni V1, Brunocilla E, Schiavina R, et al. Inguinal canal tumors of adulthood. Anticancer Res 2013;33:2361-2368. 6. Loy TS, Calaluce RD. Utility of cytokeratin 80 13.Fabbro D, Di Loreto C, Stamerra O, et al. TTF1 gene expression in human lung tumors. Eur J Cancer 1996; 32A: 512–517. 14.Bejarano PA, Baughman RP, Biddinger PW, et al. Surfactant proteins and thyroid transcription factor-1 in pulmonary and breast carcinomas. Fu-Jen Journal of Medicine Vol.14 No.2 2016 Lung adenocarcinoma in inguinal hernia sac Mod Pathol 1996; 9: 445–452. 18.Howell GM, Carty SE, Armstrong MJ, et al. 15.Di Loreto C, Di Lauro V, Puglisi F, et al. Im- Outcome and prognostic factors after adrenal- munocytochemical expression of tissue spe- ectomy for patients with distant adrenal metas- cific transcription factor-1 in lung carcinoma. J tasis. Ann Surg Oncol 2013; 20:3491. Clin Pathol 1997; 50: 30–32. 19.Blackstock AW, Herndon JE 2nd, Paskett ED, 16.Chung SD, Yu HJ, Lin WC. Metastatic pros- et al. Outcomes among African-American/ tatic adenocarcinoma in an inguinal hernia sac non-African-American patients with advanced in a patient with undetectable serum prostate non-small-cell lung carcinoma: report from the specific antigen level. J Formos Med Assoc Cancer and Leukemia Group B. J Natl Cancer 2007; 106:397-399. Inst 2002; 94:284. 17.NCCN guidelines http://www.nccn.org/professionals/physician_gls/f_guidelines.asp#site. 輔仁醫學期刊 第 14 卷 第 2 期 2016 81 Hui-Yu Lin Chun-Hou Liao Yu-Wei Chou 肺腺癌轉移至腹股溝疝氣:病例報告及文獻回顧 林惠鈺 1 廖俊厚 1,2 周有偉 1,* 腹股溝疝是在我們醫療工作中,是一種相當常見的的疾病。在患者的年齡超過了 75 歲的族群中,其終生患病率約為 47%,但其病因非常罕見的是由癌症轉移所致。而大多 數腹膜惡性腫瘤轉移至腹股溝疝多是來自胃腸道。在非腹膜惡性腫瘤轉移中,睾丸癌是最 常見的腹膜外腹股溝轉移,但是從來沒有一例報告是從肺腺癌轉移。患者可能有惡性腫瘤 轉移而引發腹股溝疝氣。這份報告提醒我們的腹股溝疝病理評價的重要性。否則,潛在的 病理可能被錯過。 關鍵字:肺癌轉移,腹股溝疝氣 天主教耕莘醫院外科部泌尿科 1 輔仁大學醫學系 2 投稿日期:2014 年 03 月 18 日 接受日期:2015 年 12 月 01 日 * 通訊作者:周有偉 電子信箱:[email protected] 82 Fu-Jen Journal of Medicine Vol.14 No.2 2016