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Stomach and Intestine(Tokyo) Volume 50, Issue 7 （June 2015）

胃と腸 50巻7号（2015年6月発行）

Japanese English

今月の主題診断困難な炎症性腸疾患

主題研究

臨床病理学的所見からみた非特異性多発性小腸潰瘍症─慢性出血性小腸潰瘍症 Clinicopathological Findings of Chronic Nonspecific Multiple Ulcers of the Small Intestine（Chronic Hemorrhagic Ulcers of the Small Intestine）八尾恒良 ^1,2,3 , 岩下明德 ^4,5 , 平橋美奈子 ⁶ , 二村聡 ⁷ , 松本主之 ^2,8,9 , 江㟢幹宏 ⁸ , 飯田三雄 ^2,10 , 松井敏幸 ^2,3 , 田中雅夫 ¹¹ , 二見喜太郎 ¹² , 八尾隆史 ^5,13 , 渡辺英伸 ^5,14 , 崎村正弘 ^2,15 , 岡部治弥 ² Tsuneyoshi Yao ^1,2,3 , Akinori Iwashita ^4,5 , Minako Hirahashi ⁶ , Satoshi Nimura ⁷ , Takayuki Matsumoto ^2,8,9 , Motohiro Esaki ⁸ , Mitsuo Iida ^2,10 , Toshiyuki Matsui ^2,3 , Masao Tanaka ¹¹ , Kitaro Futami ¹² , Kenshi Yao ^5,13 , Hidenobu Watanabe ^5,14 , Masahiro Sakimura ^2,15 , Haruya Okabe ² ¹佐田病院 ²元九州大学第2内科 ³福岡大学筑紫病院消化器科 ⁴福岡大学筑紫病院病理部 ⁵元九州大学第2病理学教室 ⁶九州大学臨床研究院保健学部門病態制御学 ⁷福岡大学病理学講座 ⁸九州大学病態機能内科 ⁹岩手医科大学消化器内科 ¹⁰九州中央病院 ¹¹元九州大学臨床・腫瘍外科学 ¹²福岡大学筑紫病院外科 ¹³順天堂大学人体病理病態学 ¹⁴PCLジャパン病理・細胞診センター ¹⁵崎村医院 ¹Division of Gastroenterology, Sada hospital, Fukuoka, Japan ²Secound Department of Internal Medicine, Kyushu University Hospital, Fukuoka, Japan ³Department of Gastroenterology, Fukuoka University Chikushi Hospital, Chikushino, Japan ⁴Department of Pathology, Fukuoka University, Chikushi Hospital, Chikushino, Japan ⁵The Second Department of Pathology, Faculty, Faculty of Medicine, Kyushu University, Fukuoka, Japan ⁶Department of Anatomic Pathology, Pathological Sciences, Graduate School of Medical Sciences, Kyushu University, Fukuoka, Japan ⁷Department of Pathology, Fukuoka University School of Medicine, Fukuoka, Japan ⁸Departments of Medicine and Clinical Science, Graduate School of Medical Sciences, Kyushu University, Fukuoka, Japan ⁹Division of Gastroenterology, Department of Internal Medicine, School of Medicine, Iwate Medical University, Morioka, Japan ¹⁰Department of Internal Medicine, Kyushu Central Hospital, Fukuoka, Japan ¹¹Department of Surgery and Oncology Graduate School of Medical Sciences Kyushu University, Fukuoka, Japan ¹²Department of Surgery, Fukuoka University Chikushi Hospital, Chikushino, Japan ¹³Department of Human Pathology, Juntendo University Graduate School of Medicine, Tokyo ¹⁴PCL Japan, Pathology & Cytology Center, Tokyo ¹⁵Sakimura Clinic, Fukuoka, Japan キーワード：非特異性多発性小腸潰瘍症 , 慢性出血性小腸潰瘍症 , デスミン染色 , CMUSE , CNSU Keyword: 非特異性多発性小腸潰瘍症 , 慢性出血性小腸潰瘍症 , デスミン染色 , CMUSE , CNSU pp.917-931

発行日 2015年6月25日 Published Date 2015/6/25

DOI https://doi.org/10.11477/mf.1403200347

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要旨●2004年までに切除され九州大学形態機能病理，福岡大学筑紫病院病理部，福岡大学病理学教室に保存されていた非特異性多発性小腸潰瘍症（CNSU）8症例，11切除標本とその臨床情報を検討し考察を加えた．1．標本；（1）小腸病変範囲：Bauhin弁の口側40〜165cm，（2）病変間距離：5cm以下，（3）敷石像，炎症性ポリープ：なし，2．潰瘍の肉眼型：CU（curved ulcer），IU（irregular ulcer）に大別され，前者が全体の64.6％を占めた．CUは本症に特異的であった．3．顕微鏡所見：病変の97.5％は粘膜下層に留まる炎症であった．デスミン染色標本上，粘膜下層に粘膜筋板と並行または集中様に走行する複層化した筋線維を認める症例があった．4．TPN繰り返し施行群（C群）では，術前1回のみ施行群（B群）に比べ，切除標本上有意に潰瘍の治癒率が低く，未施行群（A群）とB群に比べ有意に腸壁が厚かった．本症のイレウスはCMUSEと異なり，C群の長い経過中の再発再燃，腸壁の肥厚が原因と推測され，基本的な病像ではない．結論1．CNSUの小腸標本の肉眼所見は特異的である．2．本邦や欧米の非特異性多発性小腸潰瘍（症）およびCMUSEは種々雑多で，疾患単位としての概念ではない．CNSUは原因遺伝子解析の結果をまって適切な名称をつけ，1疾患単位として独立する必要がある．肉眼所見が適切に表現されたCNSU症例の蓄積が必要である．

　We investigated and discussed 11 resected specimens（lesions identified by gross examination, 164 ; lesions identified by microscopic examination, 119）that were excised by 2004 from 8 patients with chronic nonspecific multiple ulcers of the small intestine（CNSU）and stored at the Department of Anatomic Pathology of the Kyushu University, the Department of Pathology of the Fukuoka University Chikushi Hospital, or the Department of Pathology of the Fukuoka University, along with associated clinical information.

　1. Specimens :（1）extent of small intestinal lesions : 40-165cm proximal to Bauhin's valve,（2）inter-lesion distance : up to 5cm,（3）cobblestone appearance, inflammatory polyps ; none, 2. Macroscopic classification of ulcers : Ulcers were classified into two major types, CU（curved ulcer）and IU（irregular ulcer）, with the former accounting for 64.6% of the total. CU was specific to this disease. 3. Microscopic findings : 97.5% of the lesions had nonspecific inflammation, with inflammation restricted within the submucosal layer. Stratified muscle fibers running in parallel with the muscularis mucosa or in a convergence-like manner were observed in some desmin-stained specimens. This made it difficult to discriminate UL I and II ulcers. 4. Based on TPN treatment, the cases were divided into an untreated group（Group A）, a single preoperative treatment group（Group B）, and a repeated treatment group（Group C）, and compared according to（1）the ulcer cure rate and（2）intestinal wall thickness（sm-ss）.（1）The cure rate in Group B was significantly higher than that in Group A or C.（2）Thickness of the intestinal wall : In both affected and non-affected areas, the wall thickness in Group C was significantly greater than that in Group A or B. The effects of nutritional therapy were temporary as recurrence and exacerbation occurred repeatedly in Group C during the course of long-term follow-up and this was presumed to have led to thickening of the intestinal wall and surgical treatment. Ileus of this disease differs from CMUSE and is not a basic pathological feature.

　Conclusions. 1. Gross findings for the small intestine lesions, clinical features, and clinical course of CNSU are specific. 2. CNSU and CMUSE in Japan and in Western countries comprise vastly different cases and are not disease unit concepts. CNSU should be given an appropriate name after the results of causative gene analysis become available and established as an independent disease unit. More data on CNSU cases with proper descriptions of gross findings along with those of clinical features, microscopic findings, and long-term clinical courses need to be gathered.