Nobuyuki Ooike

Long-term survivor of a resected undifferentiated pancreatic carcinoma with osteoclast-like giant cells who underwent a second curative resection: A case report and review of the literature

An undifferentiated carcinoma with osteoclast-like giant cell tumors (UC-OGC) is a rare type of tumor, which predominantly occurs in the pancreas. Due to the rarity of UC-OGC, sufficient clinical data are not available and its prognosis following surgical resection remains unclear. In the current report the case of a 37-year-old female is presented, in whom an UC-OGC of the pancreas was removed and following this, a second carcinoma of the remnant pancreas was removed during a second surgical procedure. At the patient’s initial admission, the preoperative images demonstrated a well-demarcated mass with a marked cystic component at the pancreatic head. The patient underwent a pylorus-preserving pancreaticoduodenectomy. The final pathological diagnosis was UC-OGC of the pancreas and the tumor was considered to have been curatively resected based on the histopathological findings. Four years after the initial surgery, a small mass was detected in the remnant pancreas and a partial resection of the remnant pancreas was subsequently performed. Histopathologically, the tumor consisted of a poorly differentiated tubular adenocarcinoma. A retrospective pathological analysis showed a segment of a poorly differentiated tubular adenocarcinoma in the initial resected specimen. Therefore, the final diagnosis was considered to be an intra-pancreatic recurrence of UC-OGC. The patient survived 66 months following the initial surgery and 18 months since the second resection. A meta-analysis was performed in the current study by comparing UC-OGC patients who survived more than two years following surgical resection (long-term survivors) with those who succumbed less than one year following surgical resection (short-term survivors). The characteristics of the short-term survivors were patients of an older age, males, and those exhibiting smaller tumors, positive lymph node metastasis, and concomitant components of ductal adenocarcinoma, as well as pleomorphic giant cell carcinoma. The concomitant component of mucinous cystic neoplasm was not considered to be a prognostic factor. To the best of our knowledge, the patient in the current report is the first five-year survivor following a curative second resection.

RARE GASTRIC CALCIFYING FIBROUS PSEUDOTUMOR REMOVED BY ENDOSCOPIC SUBMUCOSAL DISSECTION

We report a case of a calcifying fibrous pseudotumor of the stomach that we resected using endoscopic submucosal dissection (ESD). A 61‐year‐old male with a gastric submucosal tumor was admitted to our hospital for treatment. By upper gastrointestinal tract endoscopy, a smooth‐surfaced submucosal tumor measuring 2 cm in diameter was observed in the anterior wall of the middle body of the stomach. By endoscopic ultrasonography, a mass was observed in the stomach submucosa; the mass had a well‐defined boundary. Internally, the mass was heterogeneous and hypoechoic; high spots were scattered throughout the mass. Continuity between the mass and the muscularis mucosae and muscularis propria was not observed. Strongly suspecting that this mass was a gastrointestinal stromal tumor arising from the stomach, we resected the mass by ESD for total biopsy. Histopathologically, the mass consisted of proliferation of eosinophilic collagen fibers with plasma cell infiltration and lymphoid follicle proliferation. Calcification was also observed in some parts of the mass. Thus, the mass was identified as calcifying fibrous pseudotumor. Calcifying fibrous pseudotumor of the stomach is extremely rare and its histogenesis remains unclear; however, its morphology became distinct by comparing endoscopic/radiological and histopathological findings.

A resected case of adenoneurofibroma of the colon: is it a new entity of the colonic tumor?

to achieve an activated PTT ratio of 2–3. A repeat sigmoidoscopy showed lack of endoscopic response after 3 days of heparin therapy. However, hematoxylin-eosin staining of the rectal and sigmoid biopsies revealed striking cytopathic changes of cytomegalovirus infection further confirmed on specific immunohistochemistry for cytomegalovirus antigen. Serum cytomegalovirus DNA also tested positive. Intravenous gancyclovir (5 mg/kg b.i.d.) was commenced. He failed to improve after 5 days of anticytomegalovirus therapy and underwent a subtotal colectomy with ileostomy on day 15 of his admission. The resection specimen confirmed active cytomegalovirus pancolitis on a background of chronic UC. Postoperative recovery was unremarkable, and the patient was offered the option of a pouch-anal anastomosis at a later date. Cytomegalovirus colitis is an opportunistic infection in the context of immunosuppression often seen in posttransplant and HIV-infected individuals. It is extremely rare in immunocompetent patients with IBD. Papadakis et al. (1) reported the largest cohort, 12 patients with IBD who developed cytomegalovirus colitis. Ten of these (83%) were receiving immunosuppressive agents including thiopurines and cyclosporin. However, i.v. steroid therapy alone may predispose to cytomegalovirus complicating a refractory acute colitis (5). In the current case report, the previous use of a specific cyclo-oxygenase-2 inhibitor may have triggered the relapse and altered the gut permeability to allow cytomegalovirus colonization and infection. Whether this association is causal or coincidental, the temporal relationship between a specific cyclo-oxygenase-2 inhibitor and cytomegalovirus infection in UC has not been described previously. A putative mechanism for an adverse effect of conventional nonsteroidal anti-inflammatory drugs and specific cyclo-oxygenase-2 inhibitors on IBD may be the inhibition of the prostaglandins (PGE2 and PGI2) that may have a major protective role in IBD mucosa (6). Unless future data indicate otherwise, it is probably wise to avoid the use of cyclo-oxygenase-2 inhibitors, specific or otherwise, in patients with IBD.

Subsequent loss of the proliferated bile ductules in fulminant hepatitis in childhood--isn't it caused by apoptosis?

positive predictive value in patients who have not received anyH. pylori eradication treatment. This means that patients testedH. pylori positive are reallyH. pylori positive. However, in the present study and in accordance with others (1, 4) we found the test not to be useful after eradication treatment, although other authors (3, 5, 6) found it to be reliable after eradication. We can not offer any plausible explanation for this discrepancy. It should be pointed out that we did not have any false positive results using the manufacturers’ recommended cutoff (0.140). Lowering the cutoff did not improve the results. In conclusion, we support the results of Forne et al. and think that the HpSA test is very useful for the primary diagnosis ofH. pylori infection but is not useful in confirming H. pylori eradication.

Periductal fibrosis of biliary atresia : Is it not caused by fat-storing cells?

diately after endoscopic detection by placement of a polyflex stent (diameter 18/23 mm, length 90 mm) covering the perforation and the stenosis (Fig. 1). The patient was allowed to eat and drink the following day and was discharged 7 days later without any symptoms. During the follow-up, no further complications occurred, and the patient had no dysphagia. In the endoscopy 4 months later, little granulation tissue at the proximal part of the stent was observed. By using atraumatic forceps (universal jaws), the stent could be removed under careful traction without any problems. The passage of gastrographin showed no leakage and no stenosis of the distal esophagus. In the 10-month follow-up, one argon plasma coagulation therapy to reduce granulation tissue was needed. The patient had no recurrent dysphagia during follow-up. By presenting this case, we would like to contribute a successful treatment scheme of benign esophageal stenoses and iatrogenic perforation by temporary use of a self-expanding plastic stent. This therapy was possible because of the design of the new stent, which was produced to be removable. It might not cause much granulation tissue in long-term use. This single case experience should be further evaluated in clinical trials.

Incomplete Annular Pancreas with Ectopic Opening of the Pancreatic and Bile Ducts into the Pyloric Ring: First Report of a Rare Anomaly

The patient was a 56-year-old woman who had experienced epigastralgia and dorsal pain several times over the last 20 years. She was admitted for a diagnosis of acute cholecystitis, and severe intra- and extrahepatic bile duct dilatation with inner air density was noted. No papilla of Vater was present in the descending duodenum, and 2 small holes were present in the pyloric ring. Bile excretion from one of the small holes was observed under forward-viewing endoscope. It was considered that the pancreatic and bile ducts separately opened into the pyloric ring. Based on these findings, malformation of the pancreaticobiliary duct was diagnosed. She did not wish treatment, but the obstruction associated with duodenal stenosis was noted after 2 years. Pancreatoduodenectomy was performed as curative treatment for duodenal stenosis and retrograde biliary infection through the bile duct opening in the pyloric ring. The ventral pancreas encompassed almost the entire circumference of the pyloric ring, suggesting a subtype of annular pancreas. Generally, lesions are present in the descending part of the duodenum in an annular pancreas, and the pancreatic and bile ducts join in the papillary region. However, in this patient, (1) the pancreas encompassed the pyloric ring, (2) the pancreatic and bile ducts opened separately, and (3) the openings of the pancreatic and bile ducts were present in the pyloric ring. The pancreas and biliary tract develop through a complex process, which may cause various types of malformation of the pancreaticobiliary system, but no similar case report was found on a literature search. This case was very rare and could not be classified in any type of congenital anomaly of the pancreas. We would classify it as a subtype of annular pancreas with separate ectopic opening of the pancreatic and bile ducts into the pyloric ring.

A resected case of pulmonary malignant lymphoma.

肺原発の悪性リンパ腫の一切除例を経験した.症例は59歳の女性で, 微熱と体重減少を主訴に来院した.胸部画像検査にて右肺上葉に内部が不均一な径5cm程度の腫瘤が確認され, 気管支内視鏡施行時に行われたブラッシング細胞診にて, リンパ球を背景に泡沫状の胞体を有する腫瘍細胞が散在性に認められた. 腫瘍細胞の核はきわめて大きく, 切れ込みを有しており, 大型の核小体が目立ち, クロマチン分布は不規則であった.これらの所見から腺癌が疑われ, 右肺全摘術が施行された.摘出標本の割面にて, B3に連続して径5cm程度の中心部壊死が著明な腫瘤が認められ, 組織学的にはびまん型, 大細胞型の悪性リンパ腫で, 免疫組織学的にはB細胞性と診断された.さらにin situ hybridizationにより腫瘍細胞へのEBウイルスの感染が確認された.

A case of malignant pancreatic endocrine tumor. Immunocytological and immunohistological report.

悪性膵内分泌腫瘍の一切除例を経験した.症例は47歳, 男性.1年半前より左季肋部痛が出現し, 1ヵ月前の人間ドックにて膵体尾部腫瘍と多発性の肝腫瘍が発見され当院受診となった.血液検査上は有意な所見を認めなかったが, 腹部画像診断にて膵体尾部に最大径7cm程度のhypervascularな腫瘍と多発性の肝腫瘍が確認された.肝腫瘍部位からの生検にて転移性の膵内分泌腫瘍が疑われたため, 膵体尾部切除術が施行された.切除材料の割面の擦過塗抹細胞標本では, 豊富な胞体を有する大型の腫瘍細胞がシート状あるいはリボン状に配列し, 核は大小不同で偏在傾向を示し, クロマチンパターンは顆粒状で, 大型の核小体を1～2個有するものが多くみられた.細胞学的には膵内分泌腫瘍と判定されたが, 明らかな悪性所見は確認されなかった.免疫細胞学的に, 大部分の腫瘍細胞はchromograninA, NSEに陽性で, 一部の細胞はsomatostatinとpancreatic polypeptideに陽性を示し, CEAやCA19-9に陽性の腫瘍細胞も確認された.組織学的に, chromograninA染色陽性の腫瘍細胞の脈管侵襲や神経周囲浸潤が確認され, 本症例は悪性膵内分泌腫瘍と診断された.

An autopsy case of mucin producing cancer of the pancreas.

粘液産生膵癌の1剖検例を経験した. 症例は84歳の男性で, 一年半前から上腹部鈍痛を自覚し, 近医にて慢性膵炎にて経過観察されていたが, 摂食困難に陥り当院受診となった. 血液検査にて貧血とCEA, CA19-9の上昇を認め, 腹部画像検査にて主膵管の瀰漫性拡張と膵頭部腫瘤が確認され, 粘液産生膵癌の診断で加療を行っていたが不帰の転機を辿り, 剖検がなされた. 剖検時, 主膵管や一次分岐膵管は粘液を内容し著明に拡張しており, 一部に乳頭状結節が確認された. 膵液細胞診にて, 粘液を背景に乳頭状構造を示す大型でN/Cが高く核小体が目立つ不整な核を有する高度異型の陽性細胞と, 中型でN/Cが高くなく核小体が目立たない楕円形の核を有する中等度異型の疑陽性細胞および異型性のない杯細胞化生細胞が認められた. 組織学的に乳頭状結節は膵管内乳頭腺癌-乳頭腺腫-乳頭状過形成が認められ, 膵液細胞所見は組織学的所見を反映したものと考えられた. また, 膵実質や腹膜には低分化型管状腺癌の著明な浸潤増殖がみられた.粘液産生膵癌にはさまざまな異型度の細胞がみられるが, 膵液細胞診では浸潤発育を示す場合の予後は不良であることを念頭に置いた対応が必要であることを示唆する症例と考えられた.