Clin J Gastroenterol (2009) 2:394–397 DOI 10.1007/s12328-009-0109-5

CASE REPORT

Simultaneous adenocarcinoma and leiomyoma of the stomach presenting as a collision tumor Masanori Tokunaga Æ Shigekazu Ohyama Æ Yoshiya Fujimoto Æ Naoki Hiki Æ Tetsu Fukunaga Æ Noriko Yamamoto Æ Toshiharu Yamaguchi

Received: 3 January 2009 / Accepted: 7 September 2009 / Published online: 2 October 2009 Ó Springer 2009

Abstract A 68-year old Japanese man was referred to the Cancer Institute Hospital for the treatment of a rectal cancer. Preoperative esophagogastroduodenoscopy revealed a submucosal tumor with ulcer formation near the esophagogastric junction of the stomach. The initial treatment strategy was to perform a proximal gastrectomy with limited lymph node dissection as well as a simultaneous anterior resection. However, histopathological examination of a biopsy specimen of the submucosal tumor revealed a moderately differentiated adenocarcinoma, and therefore a total gastrectomy with D2 lymph node dissection was performed. Histopathological examination of the resected specimen revealed both adenocarcinoma and leiomyoma presenting as a collision tumor. Preoperative accurate histopathological diagnosis of a gastric submucosal tumor is generally difficult. Consequently, surgical resection of large gastric submucosal tumors may be indicated in the absence of histopathological confirmation of the diagnosis. However, in this case preoperative histopathological diagnosis enabled surgeons to perform the appropriate surgery. We conclude that to determine the appropriate treatment strategy, preoperative histopathological examination of tumors should be performed even in a case of

M. Tokunaga  S. Ohyama (&)  Y. Fujimoto  N. Hiki  T. Fukunaga  T. Yamaguchi Department of Gastroenterological Surgery, Cancer Institute Hospital, Japanese Foundation for Cancer Research, 3-10-6 Ariake, Koto-ku, Tokyo 135-8550, Japan e-mail: [email protected] N. Yamamoto Department of Pathology, Cancer Institute Hospital, Japanese Foundation for Cancer Research, Tokyo, Japan

123

suspected submucosal tumor, particularly if mucosal ulceration is present. Keywords Collision tumor  Gastric cancer  Leiomyoma  Adenocarcinoma

Introduction Submucosal tumor (SMT) is a relatively rare gastrointestinal neoplasm and accounts for no more than 1–2% of all gastrointestinal malignancies [1–3]. Gastric SMT includes gastrointestinal stromal tumor (GIST), leiomyoma, and leiomyosarcoma or schwannoma. Obtaining an accurate preoperative histopathological diagnosis is generally difficult [4, 5]. As a result, surgical resection of a large SMT (C20 mm diameter) is usually performed for the dual purpose of achieving a cure and obtaining an accurate histopathological diagnosis [1, 6–8]. The incidence of lymph node metastasis is rare in gastric SMT; therefore standard treatment for SMT is a local resection of the stomach without lymph node dissection, even if the tumor is large. A collision tumor is considered as double tumor that morphologically appears as a ‘side by side’ or ‘one upon another’ pattern, and reports concerning collision tumors of adenocarcinoma and SMT are anecdotal. Theoretically, gastrectomy with lymph node dissection is an essential treatment [9, 10]. However, lymphadenectomy may be omitted if preoperative examination failed to detect adenocarcinoma and diagnosed the tumor as SMT. We present a case of a collision tumor of SMT and adenocarcinoma originating in the stomach which presented macroscopically as an SMT with mucosal ulceration. Preoperative histopathological examination enabled surgeons to implement an appropriate treatment strategy.

Clin J Gastroenterol (2009) 2:394–397

395

Fig. 1 a Esophagogastroduodenoscopy performed prior to surgery revealed a submucosal tumor with ulcer formation. b Computed tomography identified a gastric submucosal tumor near the esophagogastric junction. No lymph node swelling, invasion to adjacent structure, or metastasis to other organs was evident

Case report A 68-year old Japanese man was referred to the Cancer Institute Hospital for further examination of a rectal cancer. The rectal cancer was a stage IIIa advanced cancer, and curative treatment required a laparoscopic low anterior resection [11]. Preoperative routine examination using esophagogastroduodenoscopy and computed tomography also revealed a 50 mm diameter SMT with ulcer formation near the esophagogastric junction of the stomach (Fig. 1a, b). The initial diagnosis was of a gastric SMT and a proximal gastrectomy with limited lymph node dissection was recommended. However, histopathological examination of the SMT biopsy indicated moderately differentiated adenocarcinoma (Fig. 2). In view of this result, a total gastrectomy with D2 lymph node dissection was performed as well as laparoscopic low anterior resection of the rectal tumor. The postoperative clinical course was uneventful and the patient was discharged 19 days post-surgery.

Fig. 2 Histopathological examination prior to the surgery indicated a moderately differentiated adenocarcinoma

Results of pathological examination Gastric cancer A tumor was located in the posterior wall of the upper third of the stomach (Fig. 3). Pathological examination of the stomach revealed moderately differentiated adenocarcinoma which had infiltrated the proper muscle layer. Moreover, SMT was also present immediately beneath the adenocarcinoma in a collision tumor (Fig. 4a–c). Immunohistochemical staining revealed the SMT to be negative for c-KIT and CD34, but positive for Desmin and SMA, indicating that it was a leiomyoma. Six of thirty-seven retrieved lymph nodes were adenocarcinoma positive and some of these were located in the extraperigastric area along with the splenic artery [Station 11p lymph node according to the Japanese

Fig. 3 Submucosal tumor was located in the posterior wall in the upper third of the stomach. The tumor displayed mucosal ulceration

classification of gastric carcinoma (JCGC)] [12]. The tumor was classified as a stage IIIa advanced gastric cancer according to the JCGC.

123

396

Clin J Gastroenterol (2009) 2:394–397

submucosal layer (T1). Two of fifteen retrieved lymph nodes were positive for adenocarcinoma and therefore the tumor was classified as a stage IIIa rectal cancer according to the Japanese classification of colorectal carcinoma [11]. The patient began taking S-1 1 month after surgery as adjuvant chemotherapy for the gastric cancer.

Discussion

Fig. 4 a Mapping of both components of the collision tumor using a vertical section of the specimen; White arrows indicates an area of adenocarcinoma, and the white arrowheads indicates an area of leiomyoma. b, c Microscopic examination of the specimen indicated adenocarcinoma and leiomyoma present in a collision tumor. Adenocarcinoma had invaded the proper muscle layer

Rectal cancer Pathological examination of the rectal cancer revealed a well differentiated adenocarcinoma infiltrating within the

123

Adenocarcinoma is the most frequently observed neoplasm of the alimentary tract, whereas SMT is relatively rare, accounting for only 1–2% of all gastrointestinal malignancies [1–3]. Simultaneous occurrence of adenocarcinoma and SMT has occasionally been observed and reported [13–19] but they usually originate at different sites within the stomach [13–15, 17]. Although accurate incidence of collision tumors of adenocarcinoma and SMT is not yet determined, it is considered to be extremely rare and is usually reported as case reports. Lee et al. [18] and Trabelsi et al. [19] reported a collision tumor of signet ring cell carcinoma and CD 117 positive GIST. On the other hand, Liu et al. [16] reported a collision tumor of intestinal type adenocarcinoma and CD117 positive GIST. In the present study, the histological type of the adenocarcinoma component was intestinal type and immunohistochemical examination revealed SMT as a leiomyoma. A collision tumor is considered as double tumor that morphologically appears as a ‘side by side’ or ‘one upon another’ pattern. On the other hand, in a composite tumor, which also includes two different histologies, both components appear to be mixed haphazardly [20, 21]. It is difficult to distinguish a collision tumor from a composite tumor if both tumors are of epithelial origin [20]. In our patient, on the other hand, both components had different origins (adenocarcinoma is epithelial origin, while SMT is non-epithelial origin), and appeared as a ‘one upon another’ pattern; thus it was considered as a collision tumor. Submucosal tumor is usually covered with normal mucosal membrane, which limits the accuracy of preoperative histopathological examination [4, 5]. Therefore, large SMTs (C2 cm in diameter) are usually treated surgically to achieve an accurate histopathological diagnosis as well as a cure [1, 6–8]. Moreover, the presence of ulcer formation raises the probability of malignant potential and this also influences the decision to perform surgery prior to obtaining a histopathological diagnosis. In the present case, esophagogastroduodenoscopy revealed an SMT-like tumor with ulcer formation. Since the tumor was located very close to the esophagogastric junction, we attempted a proximal gastrectomy with limited lymph node dissection. However, histopathological

Clin J Gastroenterol (2009) 2:394–397

examination of the biopsy specimen revealed moderately differentiated adenocarcinoma and therefore a total gastrectomy with D2 lymph node dissection was performed. If the diagnosis of moderately differentiated adenocarcinoma had not been confirmed prior to the surgery, only a proximal gastrectomy with limited lymph node dissection would have been performed. This would have been insufficient treatment, leaving metastases in lymph nodes along with splenic artery (Station 11p). Today, narrow-band imaging endoscopy or pigment endoscopy enables us to detect minute early gastric cancers or to distinguish adenoma and carcinoma [22, 23]. These modalities may be useful in finding adenocarcinoma components in patients with collision tumors of adenocarcinoma and SMT, although they were not applicable in this case. However, pathological confirmation is inevitable to perform extended lymphadenectomy. We consider biopsy to be necessary in cases with SMT having ulcer formation while narrow-band imaging endoscopy and pigment endoscopy are promising options. In the present case, both adenocarcinoma and leiomyoma originated in the same site and presented as a collision tumor. Lee et al. [18] reported a similar case of large GIST in which simultaneous signet ring cell carcinoma was detected by pathological examination prior to surgery. In their case, since curative resection could not be achieved because of the advanced stage of the gastric cancer including multiple peritoneal seeding, the histopathological diagnosis prior to surgery did not have an impact on their treatment strategy. In contrast, our patient obtained appropriate treatment because of the preoperative histopathological diagnosis of adenocarcinoma. Therefore, we believe preoperative histopathological examination should be performed to select an appropriate treatment strategy even in cases of SMT, particularly those with ulcer formation.

References 1. Otani Y, Furukawa T, Yoshida M, Saikawa Y, Wada N, Ueda M, et al. Operative indications for relatively small (2–5 cm) gastrointestinal stromal tumor of the stomach based on analysis of 60 operated cases. Surgery. 2006;139:484–92. 2. DeMatteo RP, Lewis JJ, Leung D, Mudan SS, Woodruff JM, Brennan MF. Two hundred gastrointestinal stromal tumors: recurrence patterns and prognostic factors for survival. Ann Surg. 2000;231:51–8. 3. Shiu MH, Farr GH, Papachristou DN, Hajdu SI. Myosarcomas of the stomach: natural history, prognostic factors and management. Cancer. 1982;49:177–87. 4. Shinomura Y, Kinoshita K, Tsutsui S, Hirota S. Pathophysiology, diagnosis, and treatment of gastrointestinal stromal tumors. J Gastroenterol. 2005;40:775–80.

397 5. Okubo K, Yamao K, Nakamura T, Tajika M, Sawaki A, Hara K, et al. Endoscopic ultrasound-guided fine-needle aspiration biopsy for the diagnosis of gastrointestinal stromal tumors in the stomach. J Gastroenterol. 2004;39:747–53. 6. Rivera RE, Eagon JC, Soper NJ, Klingensmith ME, Brunt LM. Experience with laparoscopic gastric resection: results and outcomes for 37 cases. Surg Endosc. 2005;19:1622–6. 7. Iwahashi M, Takifuji K, Ojima T, Nakamura M, Nakamori M, Nakatani Y, et al. Surgical management of small gastrointestinal stromal tumors of the stomach. World J Surg. 2006;30:28–35. 8. Hiki N, Yamamoto Y, Fukunaga T, Yamaguchi T, Nunobe S, Tokunaga M, et al. Laparoscopic and endoscopic cooperative surgery for gastrointestinal stromal tumor dissection. Surg Endosc. 2008;22:1729–35. 9. Kurihara N, Kubota T, Otani Y, Ohgami M, Kumai K, Sugiura H, et al. Lymph node metastasis of early gastric cancer with submucosal invasion. Br J Surg. 1998;85:835–9. 10. Seto Y, Shimoyama S, Kitayama J, Mafune K, Kaminishi M, Aikou T, et al. Lymph node metastasis and preoperative diagnosis of depth of invasion in early gastric cancer. Gastric Cancer. 2001;4:34–8. 11. Japanese Society for Cancer of the Colon, Rectum. Japanese classification of colorectal carcinoma. 2nd ed. Tokyo: Kanehara; 2009. 12. Japanese Gastric Cancer A. Japanese classification of gastric carcinoma—2nd English edition. Gastric Cancer. 1998;1:10–24. 13. Uchiyama S, Nagano M, Takahashi N, Hidaka H, Matsuda H, Nagaike K, et al. Synchronous adenocarcinoma and gastrointestinal stromal tumors of the stomach treated laparoscopically. Int J Clin Oncol. 2007;12:478–81. 14. Salemis NS, Gourgiotis S, Tsiambas E, Karameris A, Tsohataridis E. Synchronous occurrence of advanced adenocarcinoma with a stromal tumor in the stomach: a case report. J Gastrointest Liver Dis. 2008;17:213–5. 15. Pamukcuoglu M, Budakoglu B, Han O, Tad M, Oksuzoglu B, Abali H, et al. An extraordinary case in whom gastrointestinal stromal tumor and low-grade malignant lymphoma are seen together in the stomach. Med Oncol. 2007;24:351–3. 16. Liu SW, Chen GH, Hsieh PP. Collision tumor of the stomach: a case report of mixed gastrointestinal stromal tumor and adenocarcinoma. J Clin Gastroenterol. 2002;35:332–4. 17. Maiorana A, Fante R, Maria Cesinaro A, Adriana Fano R. Synchronous occurrence of epithelial and stromal tumors in the stomach: a report of 6 cases. Arch Pathol Lab Med. 2000; 124:682–6. 18. Lee FY, Jan YJ, Wang J, Yu CC, Wu CC. Synchronous gastric gastrointestinal stromal tumor and signet-ring cell adenocarcinoma: a case report. Int J Surg Pathol. 2007;15:397–400. 19. Trabelsi A, Stita W, Mokni M, Yacoubi T, Mestiri S, Korbi SY. Collision epithelial and stromal tumours of the stomach: a case report. Pathologica. 2008;100:18–20. 20. Fukui H, Takada M, Chiba T, Kashiwagi R, Sakane M, Tabata F, et al. Concurrent occurrence of gastric adenocarcinoma and duodenal neuroendocrine cell carcinoma: a composite tumour or collision tumours? Gut. 2001;48:853–6. 21. Chodankar CM, Pandit SP, Motiwale SS, Deodhar KP. Collision tumour of stomach. Indian J Gastroenterol. 1989;8:297–8. 22. Gheorghe C. Narrow-band imaging endoscopy for diagnosis of malignant and premalignant gastrointestinal lesions. J Gastrointest Liver Dis. 2006;15:77–82. 23. Yao K, Iwashita A, Tanabe H, Nishimata N, Nagahama T, Maki S, et al. White opaque substance within superficial elevated gastric neoplasia as visualized by magnification endoscopy with narrowband imaging: a new optical sign for differentiating between adenoma and carcinoma. Gastrointest Endosc. 2008;68:574–80.

123

Simultaneous adenocarcinoma and leiomyoma of the stomach presenting as a collision tumor.

A 68-year old Japanese man was referred to the Cancer Institute Hospital for the treatment of a rectal cancer. Preoperative esophagogastroduodenoscopy...
345KB Sizes 0 Downloads 13 Views