Meckel憩室gastrointestinal stromal tumorの1例

鈴木 陽三; 池永 雅一; 竹山 廣志; 小田切 数基; 柳本 喜智; 山下 雅史; 清水 潤三; 今村 博司; 田村 裕美; 足立 史朗; 冨田 尚裕; 堂野 恵三

doi:10.5833/jjgs.2023.0038

Abstract

症例は75歳の男性で，夕食後突然出現した腹痛を主訴に救急搬送された．造影CTで骨盤内左側小腸に造影効果を伴う肥厚した壁から成る憩室構造と，その内部に直線状の高吸収域がみられ，Meckel憩室魚骨迷入ならびに異所性組織あるいは粘膜下腫瘍と診断した．Free airは見られず腹痛も緩解したが，穿孔リスクと腫瘍性病変の可能性を考慮し単孔式腹腔鏡下小腸部分切除術を施行した．回腸末端から60 cm口側の小腸間膜付着部対側に憩室構造がみられ，内部には魚骨が迷入し，それを囲むように17 mm大の白色調充実性腫瘍を認めた．病理組織学的検査で紡錘型細胞が束状構造を呈し，CD117・CD34陽性，desmin・S100陰性であったことからgastrointestinal stromal tumor（以下，GISTと略記）と診断した．Meckel憩室GISTはまれであるため報告する．

Translated Abstract

A 75-year-old man was taken by ambulance to the emergency department with a complaint of severe abdominal pain after dinner. An enhanced CT scan showed a fish bone in a small intestinal diverticulum with an enhanced thick wall in the left pelvis without free air. A diagnosis of fish-bone migration and submucosal atopic tissue or a tumor of Meckel’s diverticulum was made. Abdominal pain resolved spontaneously during the course, but in consideration of the risk of perforation and the possibility of a tumor in the wall, single-incisional laparoscopic partial resection of the ileum was performed. The diverticulum was diagnosed as Meckel’s diverticulum because it was located approximately 60 cm from the ileocecal valve on the anti-mesenteric side of the ileum containing a full layer of the small intestine. A fish bone was found stuck in the Meckel’s diverticulum without evidence of perforation, which led to diagnosis of fish-bone migration into Meckel’s diverticulum. Histopathologic analysis revealed a 17-mm white submucosal tumor consisting of bundled spindle-like cells without mitosis. Immunohistochemical analysis showed the tumor to be positive for CD117 and CD34, and negative for desmin and S100. These findings confirmed the diagnosis of gastrointestinal stromal tumor arising from Meckel’s diverticulum.

はじめに

Meckel憩室は卵黄管の消失不全により形成される小腸の真性憩室で，一般集団の0.3～4%にみられる消化管領域で最も多い先天異常である¹⁾．ほとんどの場合は無症状で経過するが，4%に症状が出現し，その原因は出血・閉塞・憩室炎がほとんどで異物穿孔は1.1%とまれである²⁾³⁾．

一方，gastrointestinal stromal tumor（以下，GISTと略記）は消化管の筋間神経叢周囲や輪状筋層内に存在する消化運動のペースメーカー的役割を果たすCajal細胞から発生する粘膜下腫瘍で，消化器悪性腫瘍の1～2%を占める⁴⁾．

この度，我々は魚骨迷入を契機に存在が診断され，治癒切除しえたMeckel憩室GISTの1例を経験したので，若干の文献的考察を加えて報告する．

症例

患者：75歳，男性

主訴：腹痛

既往歴：高血圧症，肺炎

現病歴：鰈の煮付けを含む夕食を摂取した後に突然腹痛が出現し当院に救急搬送された．

現症：身長165 cm，体重49 kg，血圧156/110 mmHg，脈拍83回/分，体温37.0°C．腹部全体の自発痛・圧痛を認めたが，あきらかな反跳痛，筋性防御は認めなかった．

血液生化学検査所見：WBC 6,100/μl，RBC 529×10⁴/μl，Hb 16.1 g/dl，Plt 25.9×10⁴/μl，TP 6.8 g/dl，ALB 4.1 g/dl，BUN 15 mg/dl，Cre 0.69 mg/dl，LDH 155 U/l，CK 102 U/l，CRP 0.11 mg/dlと特記すべき異常は認めなかった．

腹部造影CT所見：腹腔内に明らかなfree airは指摘しえなかったが，骨盤内左側小腸に憩室構造を認めた．その壁には造影効果を伴う肥厚がみられ，また内部には両端が壁内に留まる直線状の高吸収域がみられた（Fig. 1a～f）．

Fig. 1 Preoperative enhanced abdominal CT revealed a 2-cm diverticulum with a thick enhanced wall (arrowheads) and a fishbone stuck in the wall. a: axial section. b: coronal reconstruction. c: sagittal reconstruction. d: oblique axial reconstruction. e: oblique coronal reconstruction. f: oblique sagittal reconstruction.

以上より，Meckel憩室の魚骨迷入，ならびに異所性組織あるいは粘膜下腫瘍を疑った．受診後疼痛は徐々に軽快した．魚骨の穿孔・腹腔内膿瘍形成への移行，ならびに腫瘍性病変であった場合腫瘍が破裂や出血を来し，あるいはリンパ節転移・遠隔転移などを起こすなど進行する可能性があり診断的治療が必要と判断し，発症から12日目に手術を施行した．

手術所見：臍部に縦2.5 cmの小切開を置き，単孔式腹腔鏡操作で腹腔内を観察したところ，明らかな腹水や癒着は認めなかった．回盲部から60 cmの回腸の腸間膜付着部対側に，大網による被覆された膨隆を認めた．臍創部より体外に導出したところ，大網は容易に剥離可能で，膨隆は大きさ約20 mmの憩室であった（Fig. 2a）．魚骨の明らかな露出は認めず，腸管リンパ節の腫大も認めず，2.5 cmのmarginを確保して回腸を部分切除し手術を終了した．

Fig. 2 Intraoperative and macroscopic findings. a: Intraoperatively, a thick-walled Meckel’s diverticulum without evidence of perforation of a fish bone was found. b: The resected ileum specimen appeared to harbor a fish bone in Meckel’s diverticulum (between forceps). The insets show a magnified view of the orifice (left), and the fish bone stuck in the Meckel’s diverticulum (right). c: The resected ileum specimen appeared to be 17-mm tumor surrounding the Meckel’s diverticulum. d: Upon sectioning, the tumor (arrows) was a well-circumscribed, solid and yellowish-white mass.

切除標本肉眼所見：Meckel憩室内に魚骨を認めた（Fig. 2b）．Meckel憩室を囲むように17 mm大の白色調充実性腫瘍を認めた（Fig. 2c, d）．

病理組織学的検査所見：腫瘍は漿膜下層に存在し（Fig. 3a），HE染色では紡錘型細胞の束状構造を認め，細胞異形は軽度，腫瘍細胞分裂像数は0/50 high power fieldであった（Fig. 3b）．免疫組織化学的染色ではCD117（+）（Fig. 3c），CD34（+）（Fig. 3d），desmin（–）（Fig. 3e），S100（–）（Fig. 3f）であった．異所性胃粘膜や膵組織は認めなかった．腫瘍が回盲部から60 cm口側の回腸の腸間膜付着部対側に開口する真性憩室の粘膜下層にみられたことから，Meckel憩室に発生したGISTと診断した．

Fig. 3 Histological and immunohistochemical (IHC) studies. a: A loupe image of a HE-stained section showed that the tumor was located in the subserosal layer. b: HE-stained sections (box in loupe image) showed that the tumor consisted of spindle-shaped cells with interweaving bundle patterns (inset). In IHC staining, the tumor was positive for CD117 (c) and CD34 (d), but negative for anti-desmin (e) and S100 (f). Scale bars, 1 mm (a) and 200 μm (b–f). Original ×10 magnification (b–f).

術後経過：経過良好で術後8日目に退院し，現在定期外来観察中である．

考察

魚骨は義歯，press through pack包装シートなどとともに成人異物誤飲の最も高頻度な要因の一つであり9～45%を占めるとされているが⁵⁾，2000年1月から2022年12月の期間で，医学中央雑誌を用いて「Meckel憩室」and「魚骨」をキーワードとして（会議録を除く），PubMedを用いて「Meckel’s diverticulum」and「fish bone」をキーワードとして（抄録を参照可能なもののみ）それぞれ調べたところ，Meckel憩室にみられた魚骨症例は，和文で15例，英文で12例とまれで^6)～31)，腫瘍の併存は見られなかった．

Meckel憩室は長さ当たりの腫瘍発生頻度が他の小腸部分よりも70倍高いとされている³²⁾．また，GISTの臓器分布は，胃55.6%，小腸31.8%，大腸6.0%，食道0.7%とされており，Meckel憩室の含まれる小腸が2番目に発症の多い臓器ということになる³³⁾．一方で，Meckel憩室に発生する腫瘍の頻度は2.5%で²⁾，またその種類は神経内分泌腫瘍（neuroendocrine tumor；以下，NETと略記）が76～77%，腺癌が0～11%，間質腫瘍・平滑筋腫・平滑筋肉腫が10～20%，平滑筋肉腫が0～4%と報告されており²⁾³²⁾，GISTはまれである．

小腸粘膜下腫瘍の治療方針決定においては生検による術前診断確定も考慮されるが，本症例では魚骨迷入状態であり合併症リスクが高いため実施せず，NETの場合であっても局所病変で手術合併症リスクが少ない場合は切除が推奨されるため³⁴⁾，診断的治療として手術を行った．また，2 cm以下で細胞分裂像の見られない小腸のGISTは再発低リスクに相当するため，術後は補助化学療法を行わずに経過観察の方針とした^35)～37)．

2000年1月から2022年12月の期間で，医学中央雑誌を用いて「Meckel憩室」and「GIST」をキーワードとして（会議録を除く），PubMedを用いて「Meckel’s diverticulum」and「gatrointestinal stromal tumor」，および，「Meckel’s diverticulum」and「gastrointestinal stromal tumour」をキーワードとしてそれぞれ調べたところ，Meckel憩室に発生したGISTは，和文で3例，英文で47例報告があった^38)～87)．本症例を含めた51例について検討は以下の通りである（Table 1）．患者年齢（中央値）は59（50～68）歳，性別は男性が71%とやや多く，92%で症状を有し，症状の内訳は腹痛59%，下血25%，嘔吐16%であった．腫瘍径（中央値）は55（35～80）mm，穿孔の割合は33.3%であった．腹腔内併存疾患を8例に認め，腫瘍性病変5.9%，軸捻転3.9%，Crohn病2.0%，虫垂炎2.0%，魚骨2.0%（本症例）であった．腹痛がみられる場合には通常の急性腹症同様に穿孔の可能性を考慮し緊急手術を考慮する必要があると考えられた．本症例では激しい腹痛を契機に受診されたが，受診後短時間で自然軽快したことから，腹痛は魚骨が迷入した際に生じたものと推測された．また，腫瘍径によらず穿孔を発症しうることから，Meckel憩室GISTが疑われた症例に対しては耐術能があれば手術を検討すべきと考えられた．

Table 1 Summary of reported cases of GIST arising from Meckel’s diverticulum

Age (year)	59 (50–68)
Sex (Female : Male : ND)	14 (27.5) : 36 (70.6) : 1 (2.0)
Symptoms	47 (92.2)
Abdominal pain*	30 (58.8)
Melena*	13 (25.5)
Vomit*	8 (15.7)
Distension*	5 (9.8)
Nausea*	3 (5.9)
Fever*	2 (3.9)
Tumor size (mm)	55 (35–80)
Perforation	17 (33.3)
Concomitant abdominal disease	8 (15.7)
Volvulus	2 (3.9)
Acute appendicitis	1 (2.0)
Colorectal cancer	1 (2.0)
Crohn’s disease	1 (2.0)
Endometrial carcinoma	1 (2.0)
Fish bone	1 (2.0)
Pheochromocytoma	1 (2.0)

Data are shown as n (%) or median (interquartile range). ND: not described. *Some symptoms co-existed.

利益相反：なし

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