CASE REPORT – OPEN ACCESS International Journal of Surgery Case Reports 3 (2012) 516–519
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Gastric carcinoma with osteoblastic differentiation Fatih Selcukbiricik a , Deniz Tural a,∗ , Elif Tuba Senel a , Sergülen Dervisoglu b , Süheyla Serdengecti a a b
Istanbul University, Cerrahpasa Medical Faculty, Department of Internal Medicine, Division of Medical Oncology, TR-34098 Istanbul, Turkey Istanbul University, Cerrahpasa Medical Faculty, Department of Pathology, Istanbul, Turkey
a r t i c l e
i n f o
Article history: Received 8 March 2012 Received in revised form 4 July 2012 Accepted 6 July 2012 Available online 20 July 2012 Keywords: Gastric osteosarcoma Early recurrence Poor prognosis
a b s t r a c t INTRODUCTION: Carcinosarcoma is a rare malignant biphasic tumor which has sarcomatous and carcinomatous components. Stomach localization is very rare. We discuss the diagnosis, follow-up and treatment of patients diagnosed with gastric carcinosarcoma in company with the literature review. PRESENTATION OF CASE: A 73-year-old white male patient applied to hospital with dyspeptic complaints lasting for 2 months. His endoscopic examination revealed an ulcero-vegetating mass in the cardiac region of his stomach. Total gastrectomy and D2 lymph node dissection were performed for the patient. In the pathologic evaluation, the tumor was found consistent with Stage IIA stomach adenocarcinoma in accordance with AJCC (7edt, 2010) classification. Pathologic specimen was reevaluated by an expert pathologist for the patient with progression and liver metastasis under adjuvant chemotherapy and concomitant radiotherapy. The new pathology was consistent with gastric carcinosarcoma, and 90% of the tumor was identified as osteosarcoma whereas 10% was identified as carcinoma. Cisplatin doxorubicinebased chemotherapy was given considering the fact that sarcomatous component was dominant.1 The patient was given 3 courses of chemotherapy. However, as the patient showed progression under therapy, he died after 14 months of the diagnosis. DISCUSSION: Gastric carcinosarcoma is a very rare and clinically aggressive malignancy. Recurrence is likely to occur with a rate of more than 50% in patients who have undergone resection within the first year following surgery, and overall survival time is 10–15 months. CONCLUSION: In refractory gastric carcinoma cases with rapid progression, we suggest that gastric carcinosarcoma with biphasic component should be taken into consideration and the pathological evaluation should be performed by an expert pathologist. © 2012 Surgical Associates Ltd. Published by Elsevier Ltd. Open access under CC BY-NC-ND license.
1. Introduction Carcinosarcoma is a rare malignant biphasic tumor which has sarcomatous and carcinomatous components together.2 It typically develops in the uterus, eosophagus, and breast while stomach localization is very rarely seen in carcinosarcoma cases.3–6 Although the tumor may be polypoid, exophytic and endophytic it is generally ulceroinfiltrative in structure and large in size.5,7,8 It is diagnosed immunohistochemically.9,10 Its treatment involves radical, subtotal or total gastrectomy.11,12 2. Case A 73-year-old white male patient applied to hospital with dyspeptic complaints lasting for 2 months. His endoscopic examination revealed an ulcero-vegetating mass of 5–6 cm in the cardiac region filling the fundus. Evaluation of preoperative thoracal CT revealed a mass lesion causing an asymmetrical thickening of the gastric wall without any detected distant metastasis. Fine needle biopsy of the mass conducted under the guidance of upper tract gastroscopy
∗ Corresponding author. Tel.: +90 212 4143000; fax: +90 212 4143017. E-mail address:
[email protected] (D. Tural).
suggested the diagnosis of gastric adenocarcinoma. Total gastrectomy and D2 lymph node dissection were performed for the patient. In his pathological examination, a nonmetastatic subserosal tumor was detected during the sampling of 23 lymph nodes. The tumor was found consistent with T3 N0 (Stage IIA) stomach adenocarcinoma in accordance with AJCC (7edt, 2010) classification. Tumor marker level was below CEA 3.78, CA 19.9 2. Thoracic, abdominal, and pelvic computed tomography revealed no distant metastasis. The patient diagnosed with Stage II stomach adenocarcinoma was given adjuvant 5 fluorouracil, calcium folinate chemotherapy and concomitant radiotherapy in the Macdonald protocol.13 The abdominal computed tomography showed that the size of the liver was above the normal, and revealed multiple metastatic lesions that were in affinity with each other and diffuse in the left lobe of the liver in patient who developed left side pain (Fig. 1). Upon progression under treatment, revision was required for pathological representations. Pathologic specimen was reevaluated by an expert pathologist. The tumor was located in the cardia; had a size of 12 cm × 9.5 cm × 5 cm; was fungative, expansive with 20% massive necrosis area in the center (Fig. 2a–c). It had totally two components which were epithelial and mesenchymal. The carcinomatous component was reported as tubular and indifferential type adenocarcinoma; whereas, the sarcomatous component was reported as osterosarcoma. Ninety percent of the tumor was
2210-2612 © 2012 Surgical Associates Ltd. Published by Elsevier Ltd. Open access under CC BY-NC-ND license. http://dx.doi.org/10.1016/j.ijscr.2012.07.001
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criteria, inability to evaluate field of the tumoral area completely, and misleading impact of histopathologic examination of the fine needle biopsy material were suggested as the reasons for the misdiagnosis made after the first evaluation (see Table 1). Considering the fact that sarcomatous component was dominant, metastatic osteosarcoma therapy was planned and cisplatin doxorubicine-based chemotherapy was given to the patient diagnosed with metastatic stomach carcinosarcoma.1 However, the patient died after 14 months of diagnosis in the 3rd month of the therapy. 3. Discussion
Fig. 1. Multiple liver metastasis.
identified as osteosarcoma whereas 10% was identified as carcinoma (Fig. 3a–c). The sarcomatous component stained positively for vimentin; further, an intermediate component was also available that was negative for keratin and vimentin. Lymphovascular invasion was positive. Assessment using only light microscopic
Carcinosarcoma is a rare biphasic tumor which has epithelial and mesenchymal malignant tumors together.6,12 The majority of gastric carcinosarcoma cases is comprised male population from Japan and who are above 60-years-old.2,7,9 It typically develops in the uterus, eosophagus, and breast while stomach localization is very rarely seen in carcinosarcoma cases.3–6 Although the tumor may be polypoid, exophytic and endophytic it is generally ulceroinfiltrative in structure and large in size.5,7,8 Although the carcinomatous component usually consists of intestinal adenocarcinoma, there may be endocrine and neuroendocrine components. The sarcomatous component may present myeloblastic, condroblastic, lipoid or osteoblastic differentiation.2,5,9,10,15 The gold standard of diagnosis is immunohistochemical analysis. The major specific markers used in the diagnosis are CEA, EMA, chromogranin A, pancreozymin, CD 56, and synaptophysin for the carcinomatous component; demsin and vimentin for the sarcomatous component.9,10 Carcinosarcoma
Fig. 2. (a–c) Macroscopic appearance of the tumor.
Fig. 3. (a) 4785/09-HEx100, (b) 4875/09-HEx200, (c) 4875/09-HEx200. Malign osteoid production by tumoral osteoblasts in osteosarcomatous area next to moderately differentiated adenocarcinoma component of tumor.
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Table 1 Prior reported cases gastrit carcinosarcoma. No.
Author
Year
Age
Sex
Size (cm)
Treatment
Outcome
1 2 3 4 5 6 7 8 9 10 11
Ashide4 Nakayama et al.2 Matsukuma24 Inoue25 Tsuneyama13 Sato et al.10 Teramachi et al.9 Yamazaki5 Kuroda et al.15 Yoshıyukı22 Current study
1995 1997 1997 1998 1999 2001 2003 2003 2006 2007 2012
74 69 74 74 63 67 62 56 59 70 73
M M F F M F M M M F M
12 20 15 7.8 7 8 10 9 9.2 21 12
Resection Resection Resection Resection Resection Resection Resection Resection Resection Palliative care Resection
NED (7 year) DOD (1 month) DOD (5.5 month) DOD (10 month) NED (10 month) NED (11 month) NED (20 month) DOD (2 month) ND DED (16 day) DOD (14 month)
DOD: died of disease, ND: not described, NED: alive no evidence disease.
metastasis may consist of only the carcinomatous or only the sarcomatous component as well as carcinosarcoma.7 The surgical treatment of gastric cancer has been a matter of extensive research. Carcinosarcoma treatment consists of radical, subtotal or total gastrectomy11,12 and in the current era D2 gastrectomy is accepted as the standard operation, with an excellent cure rate and low loco regional recurrence.16 More aggressive extended D3 gastrectomy has been practiced in certain patients with survival and diseasefree benefits, especially for advanced gastric cancer.17,18 On the contrary, D1 gastrectomy has been considered to be sufficient for mucosal cancer.19 Chemotherapy and radiotherapy have not been defined yet. Although uncertain, methionine/valine-depleted enteral nutrition has been reported to contribute to tumor reduction.20 Although the prognosis of gastric carcinosarcoma is more poor compared to that of other gastric carcinomas, the overall survival time is 10–15 months with a possibility of relapse over 50% within the first year following surgery in gastric carcinosarcoma cases.9,10,21 Gastric carcinosarcoma is a very rare and clinically aggressive malignancy. Recurrence is likely to occur with a rate of more than 50% in patients who have undergone gastric resection within the first year following surgery with an overall survival time of 10–15 months.9,10,21 In the differential diagnosis of refractory gastric carcinoma cases with rapid progression, we suggest that gastric carcinosarcoma should be taken into consideration; the pathological evaluation should be performed by an expert pathologist; and tumor specimen should be evaluated in details. Conflict of interest None. Funding None. Ethical approval Written informed consent was obtained from the patient for publication of this manuscript and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal Author contributions All the authors have contributed to this case report, and to its design. Imaging and pathology studies were performed by the corresponding author and the last author. Analysis and interpretation of the data were performed by all the authors.
References 1. Souhami RL, Craft AW, Van der Eijken JW. Randomised trial of two regimens of chemotherapy in operable osteosarcoma: a study of the European Osteosarcoma Intergroup. Lancet 1997;2735:911–7. 2. Nakayama Y, Murayama H, Iwasaki H, Iwanaga S, Kikuchi M, Ikeda S, et al. Gastric carcinosarcoma (sarcomatoid carcinoma) with rhabdomyoblastic and osteoblastic differentiation. Pathology International 1997;47: 557–63. 3. Tanimura H, Furuta M. Carcinosarcoma of the stomach. American Journal of Surgery 1967;113:702–9. 4. Hanada M, Nakano K, Ii Y, Takami M. Carcinosarcoma of the stomach. A case report with light microscopic, immunohistochemical and electron microscopic study. Acta Pathologica Japonica 1985;35:951–9. 5. Yamazaki K. A gastric carcinosarcoma with neuroendocrine cell differentiation and undifferentiated spindle-shaped sarcomacomponent possibly progressing from the conventional tubular adenocarcinoma; an immunohistochemical and ultra structural study. Virchows Archiv: An International Journal of Pathology 2003;442:77–81. 6. Insabato L, Di Vizio D, Ciancia G, Pettinato G, Tornillo L, Terracciano L. Malignant gastrointestinal leiomyosarcoma and gastrointestinal stromal tumor with prominent osteoclast-like giant cells. Archives of Pathology and Laboratory Medicine 2004;128:440–3. 7. Kayaselcuk F, Tuncer I, Toyganozu Y, Bal N, Ozgur G. Carcinosarcoma of the stomach. Pathology Oncology Research 2002;8:275–7. 8. 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. Archives of Pathology and Laboratory Medicine 2000;124:682–6. 9. Teramachi K, Kanomata N, Hasebe T, Ishii G, Sugito M, Ochiai A. Carcinosarcoma (pure endocrine cell carcinoma with sarcoma components) of the stomach. Pathology International 2003;53:552–6. 10. Sato Y, Shimozono T, Kawano S, Toyoda K, Onoe K, Asada Y, et al. Gastric carcinosarcoma, coexistence of adenosquamous carcinoma and rhabdomyosarcoma: a case report. Histopathology 2001;39:543–4. 11. Chen YP, Yang JS, Liu DT, Chen YQ, Yang WP. Long term effect on carcinoma of esophagus of distal subtotal gastrectomy. World Journal of Gastroenterology 2004;10:626–9. 12. Liu SW, Chen GH, Hsieh PP. Collision tumor of the stomach: a case report of mixed gastrointestinal stromal tumor and adenocarcinoma. Journal of Clinical Gastroenterology 2002;5:332–4. 13. Macdonald JS, Benedetti J, Smalley S, Haller D, Hundahl S, Jessup J, et al. Chemoradiation of resected gastric cancer: a 10-year follow-up of the phase III trial INT0116 (SWOG 9008). Journal of Clinical Oncology (Meeting Abstracts) 2009;2715:4515. 15. Kuroda N, Oonishi K, Iwamura S, Ohara M, Hirouchi T, Mizumo K, et al. Gastric carcinosarcoma with neuroendocrine differentiation as and leiomyosarcomatous and myofibroblastic differentiation as the sarcomatous component. APMIS 2006;114:234–8. 16. Songun I, Putter H, Kranenbarg EM, et al. Surgical treatment of gastric cancer: 15year follow-up results of the randomised nationwide Dutch D1D2 trial. Lancet Oncology 2010;11:439–49. 17. Roviello F, Pedrazzani C, Marrelli D, et al. Super-extended (D3) lymphadenectomy in advanced gastric cancer. European Journal of Surgical Oncology 2010;36:439–46. 18. Zhang H, Liu C, Wu D, et al. Does D3 surgery offer a better survival outcome compared to D1 surgery for gastric cancer? A result based on a hospital population of two decades as taking D2 surgery for reference. BMC Cancer 2010; 10:308. 19. Kong SH, Yoo MW, Kim JW, et al. Validation of limited lymphadenectomy for lower-third gastric cancer based on depth of tumour invasion. British Journal of Surgery 2011;98:65–72. 20. He YC, Cao J, Chen JW, Pan DY, Zhou YK. Influence of methionine/valine depleted enteral nutrition on nucleicacid and protein metabolism in tumor-bearing rats. World Journal of Gastroenterology 2003;9:771–4.
CASE REPORT – OPEN ACCESS F. Selcukbiricik et al. / International Journal of Surgery Case Reports 3 (2012) 516–519 21. Matsui K, Jin XM, Kitagawa M, Miwa A. Clinicopathologic features of neuroendocrine carcinomas of the stomach: appraisal of small cell and large cell variants. Archives of Pathology & Laboratory Medicine 1998;122:1010–7. 22. Ikeda Y, Kosugi S, Nishikura K, Ohashi M, Kanda T, Kobayashi T, et al. Gastric carcinosarcoma presenting as a huge epigastric mass. Gastric Cancer: Official Journal of the International Gastric Cancer Association and the Japanese Gastric Cancer Association 2007;10:63–8.
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24. Inoue S, Yoshimi F, Tonouchi H, Ono H, Amemiya R, Koizumi S, et al. A carcinosarcoma of the stomach. The Japanese Journal of Gastroenterological Surgery 1998;31:945–9 [in Japanese]. 25. Matsukuma S, Wada R, Hase K, Sakai Y, Ogata S, Kuwabara N. Gastric stump carcinosarcoma with rhabdomyosarcomatous differentiation. Pathology International 1997;47:73–7.
