AOKI, YOZO

KAWAGUCHI, TOMIJI

KAWASHIMA, HIROAKI

KODAMA, ETSUO

MORI, KAZUNARI

SAKAMOTO, YUKITOMO

SASAKI, MASAKAZU

SUGIMOTO, YOSHIHIRO

TANIMURA, HIROSHI

UCHIYAMA, KAZUHISA

UEDA, KOSHIN

UESAKA, KAZUNOBU

Kyoto University Research Information Repository

Title Osteoclast-like Giant Cell Tumor of the Pancreas Associatedwith CystadenocarcinomaAuthor(s)AOKI, YOZO; TANIMURA, HIROSHI; MORI, KAZUNARI;KODAMA, ETSUO; UESAKA, KAZUNOBU;KAWAGUCHI, TOMIJI; SUGIMOTO, YOSHIHIRO;SAKAMOTO, YUKITOMO; UCHIYAMA, KAZUHISA;SASAKI, MASAKAZU; UEDA, KOSHIN; KAWASHIMA,HIROAKICitation日本外科宝函 (1989), 58(5): 452-460Issue Date1989-09-01URL http://hdl.handle.net/2433/203896RightType Departmental Bulletin PaperTextversionpublisherKyoto UniversityArch Jpn Chir 58(5), 452～460, Sept., 1989 臨床Osteoclast-like Giant Cell Tumor of the Pancreas Associated with Cystadenocarcinoma Yozo AOKI, HrnosHI TANIMURA, KAzuNARI MoRI, ETsuo KODAMA, KAZUNOBU UESAKA, TOMIJI KAWAGUCHI, YosHIHIRO SUGIMOTO, YUKITOMO SAKAMOTO KAZUHISA UCHIYAMA, MASAKAZU SASAKI, KosHIN UEDA and HIROAKI KAWASHIMA Department of Gastroenterological Surgery, Wakayama Medical College Received for Publication, May 18, 1989. Summary （れteoclast-likegiant cell tumor is a very rare type of pancreatic malignancy, and no more than 20 cases have been reported in the English literatures. A case of this tumor associated with cystadenocarcinoma is reported. This giant cell is undistinguishable on light microscopy from osteoclast of bone and correct diagnosis of this unusual tumor is important because of its relatively poor prognosis. Introduction The incidence of pancreatic cancer is increasing in Japan 12>. Among the pancreatic cancer, ductal adenocarcinoma represents 81 % of the autopsy series and 80% of the surgical material14>. This paper reports a case of primary osteoclast like giant cell tumor of the pancreas associated with cystadenocarcinoma with a brief survey of the literature. Case Report A 44ぅ7四 rold female was admitted on September 2, 1986 with a chief complaint of palpable tumor in her left upper quadrant of the abdomen. She had experienced her s戸nptomfor last one year. On physical examination, it was found that the abdomen was soft, but a smooth, partly lobulated, child-head sized round mass was palpable in the left upper quadrant. No tenderness was elicited with palpation. Her past history included appendectomy and hypertension 20 and 2 years before her ad-Key words司 Carcinomaof pancreas, Osteoclast-like giant cel carcinoma of pancreas, Pleomorphic giant cel carcinoma of pancreas, Cystadenocarcinoma of pancreas, Carbohydrate antigen 19-9. 索引語：勝癌， E率破骨細胞型巨細胞癌，勝多形細胞型巨細胞癌，勝強胞腺癌， CA199. Present address: Department of Ga5troenterological Surgery, Wakayama Medical College, ¥Vakayama City 640. Japan GIANT CELL Tl' ¥!UR OF PA¥'< 'REAS 453 Fi量・ 1. An upper GI series showing evidence of cephalo-ventral displacement of the stomach. mission, respectively. Medication was reserpine. Laboratory results were as follows; REC count 458×104/mm3, ¥VBC count 7,400/mm3, Hb 14.8 g/dl, hematocrit 41.2%, platelet count 41.2×104/mm3, amylase 62 IU/l, lactate de-hydrogenase 96 U /I,SGOT 21じ／1,SGPT 15じ／I,alkaline phosphatase 96 U/l, y-GTP 21 U/l, total bilirubin 0.5 mg/di, ferritin level 273.3 ng/ml, carcinoembryonic antigen (CE:¥) level 1.6 ng/ml, a長toproteinlevel 0.3 ng/ml, tissue polypeptide antigen (TP A) level 36.5 U /1, and carbohydrate antigen (CA) 19-9 level 10.2 U /ml. An upper GI series showed evidence of cephalo-ventral displacement of the stomach (Fig. 1). An ultrasonography revealed a multiloculated mass with a strong posterior wall echo. An endoscopic retrograde cholangiopancreatography (ERCP) demonstrated narrowing and stretching of the main pancreatic duct at the tail of pancreas and no communication between the ductal system and C)'stic lesion. A CT scan showed a multilocular cvstic mass suspected of the pancreas in origin (Fig. 2).At laparotomy, a child-head-sized lobulated cystic mass was found arising from the tail of the n ~ P n r s. Xo liver metastases were recognized. Distal pancreatectomy was done. The resected specimen weighed 1,400 g and showed macroscopicaly a globular form with a conical protrusion (Fig. 3). Cut section examination showed various size of cyはiccavities (Fig. 3). The contents of these cysts were thick and mucoid, and brownish in colour. In one of these mass, which corresponded to th巴neckof the protrusion shown in Fig. 3. a solid focus 2 cm in diameter ~as. present. The content obtained from the くystswas analysed as follows：則00附 mlfor rern U/ml for CA 19一9.454 日外宝第58巻第5号（平成元年9月）Fig. 2. A CT scan suggesting a multilocular cystic mass originating from the pancreas. ??????????』 ?????? ? ……?、??『?，??????????? ??? ?．?，???????????????????????』 ??．?????? ? ??， ? ?????????、??????????? 、?????、??????????? ??Th巴patientrecovered uneventful and was discharged from the hospital on the 23th post-operative day. She has been doing well 3 years after surgery. Microscopical Findings In large part of the cystic wall、thecolumnar epitherial lining cells were well oriented to their basement membrane, were regularly aligned、andhad no evidence of anaplasia, piling up, or invasiveness (Fig. 4). These cells were secreting mucus, which stained positively with periodic acid Schiff stain and positively with alcian blue. In some areas, the epithelium merged with a more atypical epithelium with nuclear stratification (Fig. 4). The solid part had the appear-叫lJO ,(;)U官ull)]BUI;np‘9 'Jl).'I U) UJ¥主oqs"¥' ・urs)qd10uro;:i1d Ju;:ipnu JO wnour官d］甲）l'BA官P拙oqspu官＇urs官jdOlAココ）j)l{dOU)SO;Jpu喧）;:ipnuI官jnJ)S;JAI官AOOl puno1 pu可ゐl{ょ・p;:id官qs叶pu)dS10 1官AO;)l;)A¥ sndJ 1官山OllSa可よ'Sl!;JJ lU官）ldl工IOSU) p;:il哲］OnJ官Apu官《J）日刊dOU)SO;Jpuu lU官punq官叩M.UISBjd Ol心3可よL・);:ipnu j'BllUdJ ，山菅川p;:iU)UlUOJA;Jl{J, ・可jd:)1官UIOlOlS;Jl{l l{l)A¥ p;:ilB)JOSS官A:pi官山）JU!;:i1;:iA¥ pu官＇uil)u;:iqp;:i1四dd官SjjdJlU官）l;)l四pnU)l]l1山;Jl{J,. sndJ 1官UIOllSrn;:ipnuouorn pUB 可j;JJ;J}j)j-lS官jJO;JlSO~ JFD JO s;:idムOA¥l10 p;:ils!suw l r ・Cs・l）丘）;:iuoq JO 10山町jd:llU官）lJO <):JUB SSr :-;v:’•[}[.)'.\:\'d ：［（）迂cm:.1iTl3:::> J,NVI'.J 456 日外宝第58巻第5号（平成元年9月）Fi邑.6. CEA tisue staining showing positivity for some stromal cels (arrows，×300）・stromal cells was partly demonstrated by the results of immunohistochemical staining of CEA act1vity13J From the above findings, the diagnosis of osteoclast-like giant cel tumor of the pancreas associated with cvstadl~nocarcinoma was made. Discussion There are two type' of giant cel tumor of the pancreas1>. One is pleomorphic carcinoma, a varient of adenocarcinoma, first reported by SOMMER ct al21>, and the other giant cel tumor reported by RosAI17) The former is a pleomorphic giant cell carcinoma composed of malignant mononudeated and multinucleated giant cells with sarcomatous stroma and usually showing mucin secretion. The latter is a more benign appearing giant cell carcinoma composed of osteoclast-like giant cells and malignant stroma. These two types should be strictly distinguished because the nomenclature remains nonstandardized and considerable confusion stil exists. Tumor containing osteoclast like giant cells have been reported to occur in the thyroid20>, subcutaneous tissue18>, ovary23J, breast7人andskin2> as well as in the pancreas. These giant cells are indistinguishable from osteoclasts of bone2 ') Some authors3, 9J state that osteoclast-like giant cell tumor of the pancreas is apt to present as a large mass, and metastatic spread is slow, and that the prognosis 'ccms to be musch better than in ordinary carcinoma or pleomorphic、giantcel丸GIANT CELL TU'YIOR OF PANCREAS 457 Table 1. Literature review of osteoclast-like giant cel carcinoma of the pancreas. Case Authors Age(yrs) Symptoms Duration Location Size Treatmゼnt Survival and sex of (cm) symptoms 1 Shamblin, et al19' 49, M Peri umbilical S"veral Hea<l Man’s五st Total Alive, (1966) cramping pain, weeks pancreatectomy 15 yrs later Anorexia 2 Rosai口九（1968) 82, F Anorexia, 1 mo目 Tail 13×12×8 Subtotal 人live,Fatigue, pancreatectonw, 4 mo. later Weight loss, Partial Left upper abd. gastrectomy, mass Splenectomy, Removal of tumor thrombi from portal vein 3 Rosai1η’（1968) 74, F Anorexia, 5 mo. Head 10.5×7.5×4 Whipple Alive, Weight loss, lOmo. later Melena 4 Kay, et aJI"' (1969) 43, M Weakness 3 mo. lfrad 6×5×5 Whipple Ahve, 2lmo. いter5 Freud'', (1973) 32, F Nausea, 3 mo. Head 15 in Whipple 1、rVomiting, diameter Right upper abd. pain, Jaundice 6 Alguacil-Garcia, 93, F Large Abdominal 10 mo. et al" (1977) explorat1on, Biopsy 7 Robinson, et al即日， M 15 in Radiation 4.5 mo. (1977) diameter 8 Cubilla, et al" 45, M H"ad 7×6 ＼・hippie 4 yrs (1979) 9 Posen山，（1981) 45, F Menorrhagia, Body 14 in 九＇eakness, diameter Lower abd. pain, Nausea, Vomiting 10 Trepeta, et aJ2》 68, M Back and abd. 2 mo. Tail B×8x6 No (Radiation 3 mo. (1981) pain, planned) Intermittent mental confusion 1 Yamashita, et al2" 70, M Constipation, 2 yr. Body Thumb-tip No 7 wk. (1982) Abd. fullness 12 Walts2ぺ（1983) 59, F Epigastric pain 3 mo. Head Large （、holecysto- 2.5 yrs and bodv jejunostor町，Radiation, Chemotherapy 13 Jefrey, et al" 55, M Jaundice, Fever, 2 wk. Head 10 in l'holec、・sto- 7 mo. (1983) Weight loss diameter jejunostomy 458 日外宝第58巻第5号（平成元年9月）14 Jaloh., (1983) 72, F Upper abd. and 6-8 mo. Head 6.5×6×5 No 10 days back pain 15 Kodama, et al1》 59,M Body 4 mo. (1983) 16 Kodama, et aJl> 65, :¥! Bod、， 5 mo. (1983) 17 Drexler, et al" 75, M Anorexia, 4 mo. Head 12×10 De bulking 5 mo. (1986) Weight los (242g), Cholecysto-jejunostomy, Castro-jejunostomy, Radiation, Chemotherapy 18 Baniel, et al'》 65, F Abd. pain, 3 yr. Body 23×16×9 Local resection, Ahve, (1987) Epigastric Subtotal 6 yrs later fulnes, gastrectomy, Constipation メplenectomy19 Authors, (1989) 4, F Abd. mass 1 yr. Tail 15×15×11 Distal Ahve, pancreatectomy, 3 yrs later Splenectomy A、listedin Table 1, up to 1988, 18 cases of this type of tumor have been reported in English literaturet, 3 6,s-11, 15-17, 2, 2ペ25>. There were 10 men and 9 women ranging in age from 32 to 93 with a median age of 60.9. :¥lain clinical symptoms before admission included abdominal pain in 7, weight loss in 5, anorexia in 4 etc. Duration of these symptoms ranged from 2 weeks to 3 years (mean 8.4 months). The tumor arose from the head of the pancreas in 8, body in 5, tail in 3 and head and body in 1. The lesions were resected in only half of the patients. The average survival w出 only22.3 months. Distant metastase日arereported to occur in livers,9,21, lung引16> and adrenal gland8>, and the patient died, extremely speaking, 10 days after presentation8> From these facts, it is dif五cultto say the tumor slow growing or unlikely to metastasize. Even in the resected cases4人10>,which were pancreas head in origin, there were no obvious characteristics in clinical symptoms and macroscopic findings of the tumors compared with an ordinary adenocarcinoma of the pancreas. Thus, an early diagnosis of this type of tumor seemed to be very di伍cultand development of some newer diagnostic tools including CT scan and magnetic resonance imaging (¥IR-I) are desirable. Immunohistochemical staining of CEA activity was partly positive in our case目 Ingeneral, clear demonstration of CEA activity is highly suggestive of malignancy or, possibly, premalignant change, however, this i討notspecific for osteoclast like giant cell tumor of the pancreas. Re＜、enthistological studies have revealed that these osteoclast like cells are epithelial in origin3, l川 althoughsome arthors consider as acinar cel11?> or stromal cels>. As PosEN15> proposed, the association of a cystadenocarcinoma with a giant cell tumor of the pancreas suggests that both these tumors arose from pancreatic ductal epithelium, a theory that appears to be borne GIANT CELL Tl'MOR OF PANCREAS 459 out by the fact that these two tumors were not distinctly separate but appeared to merge. Our case may support the PosEN’s theory. 日urgeryhas been used for treatment. In fact, surgical resection in toto is the treatment of choice. When the tumor i、unresectable,only bypass operation is performed as prophylaxis against intestinal obstruction or obstructive jaundice. This palliative surgery is folio鴨・edby cl削釘Tof radiation. DREXLER et al 5i stated that a combination of 5-fluorouracil and radiation therapy was the first medical treatment with a document respomc. On the other hand, ROBI＇＼、o:-;et al16> reported that the lesion had not responded to radiation therapy but had spread diffusely. Thus, accurate histological recognition of the osteoclast-like tumor encourages an agressive surgical approach in view of the expectation of better prognosis. References 1) Alguacil-Garcia A, ¥' eiland L: The histologic spectrum. prognosis, and histogenesis of the sarcomatoid carcinoma of the pancreas. Cancer 39: 1181 1189, 1977目2) Andreev ＼＂仁、 RaitchevR, Nicolova D: l¥lalignant osteoclastoma of the skin. Br J Derma to! 76: 40-44, 1964. 3) Baniel J, Konichezky M, Wolloch ¥" Osteoclast type giant cel tumor of the pancreas. Case report. Acta Chir Scand 153: 67 69, 1987. 4) Cubilla AL, Fitzgerald PJ: Cancer of the pancreas (non-endocrine): A suggested morphologic classification. Semin Oncol 6: 285-297, 1979. 5) Drexler LJ, l¥litre RJ, Maden E, et al: Pancreatic giant cel carcinoma of the osteoclastic type: Response to 5-fluorouracil and radiation. Am J Gastroenterol 81: 1093-1097, 1986. 6) Freund U Pleomorphic giant cel tumor of the pancreas. A case report. br<1eリMedSci 9・84-88,1973. 7) Fry HJB: Osteoclastoma (myeloid sarcoma) of the human female breast. J Path Bact 30: 529-536, 1927. 8) Jalloh SS: Giant cel tumour (osteoclastoma) of the pancreas an epithelial tumour probably of pancreatic acinar origin. J Clin Pathol 36: 1171-1175, 1983目9) Jefrey I. Crow J, Ellis B¥¥': （）、teoclast-typegiant cel tumour of the pancreas. J Cl in Pathol 36: 1165 1170, 1983. 10) Kay S. Harrison J :¥I Unusual pleomorphic carcinoma of the pancreas featuring production of osteoid. Cancer 23: 1158-1162, 1969. 1) Kodama T, i¥lori ¥': Morphological behavior of carcinoma of the pancreas. 1. Histological classi五cationand electron microscopical observation. Acta Pa tho! Jpn 33: 467 481, 1983. 12) Krain LS: The rising incidence of cancer of the pancreas-Further epidemiologic studies. J Chronic Dis 23: 685 690, 1975目13) ~Iason TE, Phifer RE, Spicer SS, et al: An immunoglobulin enzyme bridge method for localizing tissue antigens. J Histochem Cytochem 17: 563 569, 1969. 14) :.Iorohoshi T, Held G, Kloppcl G: Exocrine pancreatic tumors and their histological classi凸cation. A study based on 167 autopsy and 97 surgical cases. Histopathology 7: 645 661, 1983. 15) Posen JA: Giant cel tumor of the pancreas of the osteoclastic type associated川tha mucous secreting cystadenocarcinoma. Hum Pathol 12: 944-947, 1981. 16) Robinson L, Damjenov I, Brezina p: ;.1 ultinucleated giant cel neoplasm of pancreas. Light and electron microscopy features. Arch Pathol Lab Med 101: 590 593, 1977. lηRosai J: Carcinoma of the pancreas simulating giant cel tumor of bone. Electron microscopic れ吋enceof its acinar cel origin. Cancer 22: 333 344, 1968. 18) Schmaman A, Penn I, Sorour y, Giant cel tumours of the soft tissues. S Afr :¥led J 37: 819-820, 1963. 19) Shamblin WR, Priestley JT, Sprague RG, et al: Total pancreatectomy for pleomorphic carcinoma. A五ve-year cure. Arch Surg 92: 315 317, 1966. 20) Sieberberg SG, DeGiorgi LS Osteoclastoma-like giant cel tumour of the thyroid. Cancer 31: 621-625, 1973. 460 日外宝第58巻第5号（平成元年9月）21) Sommers SC. :¥Ieissner WA: l1nu吋 alcarcinomas of the pancreas. Arch Pathol 58: 101 111, 1954. 22) Trepeta R¥¥', ¥lathur B, Laginメ etal: Giant cel tumor （“Osteoclatoma・＇） of the pancreas: A tumor of epithelial origin. Cancer 48: 2022 2028, 1981. 23) Veliath AJ, Sankaran V、AuroraAL. Ovarian giant cell tumour with cystadenocarcinoma. Arch Pathol 99: 488 491, 1975. 24) ¥¥'alts A E Osteocla,t-tq1c giant cel tumor of the pancrea,. ..¥eta Cytol 27: 500-504, 1983. 25) Yamashita H, l¥akayama I. ¥foriuchiλet al: A giant cel tumor円fthe pancreas: A tumor of acinar cel origin. Trop ¥led 24: 203-208, 1982. 和文抄録日華嚢胞腺癌に合併した破骨細胞型巨細胞癌和歌山県立医科大学消化器外科教室青木洋三，谷村 弘，森 一成，児玉悦男，植阪和修，川口富司，杉本恵洋，内山和久，佐々木政一，上田耕臣，川嶋寛昭勝悪性腫場のうちでも破骨細胞型巨細胞癌は極めて れているが，後者も，今回著者らが調べ得た範囲では，稀で， 1988年までの報告例は20例lζ満たない．我々は 診断確定後，あるいは術後の平均生存は約22カ月に過44歳，女性で，勝嚢胞腺痛に合併した本症を経験した． ぎず，必ずしも良好とはいえない．本症の組織発生に勝尾側切除術を施行し，術後約3年を経た現在，再発 ついては，腺房細胞説と腺管上皮細胞説が挙げられてなく社会復帰している． いるが，本例が嚢胞腺癌組織中lこ混在・発生していた勝巨細胞癌は，病理組織学的には多形細胞型と破骨 ζ とから，我々は後者を支持したい．細胞型l己分類され，前者の予後は極めて悪いと報告さ

膵囊胞腺癌に合併した破骨細胞型巨細胞癌

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膵囊胞腺癌に合併した破骨細胞型巨細胞癌

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