妊娠を契機に発症した慢性特発性偽性腸閉塞症の1例

症例は女性で,23歳・妊娠12週時より腹痛・嘔吐が出現した.画像上小腸全体の拡張を認めたが,器質的な閉塞機転を認めず,機能的な腸管運動不全が疑われた.出産後も症状は軽快せず,24歳時に回盲部切除術を施行された.病理組織学的検査所見で腸管神経節細胞脱落を認め,慢性特発性偽性腸閉塞症(chronic idiopathic intestinal pseudo-obstruction;以下,CIIPと略記)と診断された.25歳時にさらに大腸亜全摘・回腸人工肛門術を施行されたが,症状は改善しなかった.29歳時に空腸人工肛門造設術を施行後,症状が軽快し経口摂取可能となった.CIIPの発症機序は不明だが,近...

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Published in日本消化器外科学会雑誌 Vol. 45; no. 3; pp. 326 - 332
Main Authors 田島, 陽介, 島田, 能史, 飯合, 恒夫, 伏木, 麻恵, 細井, 愛, 畠山, 勝義, 関根, 和彦, 野上, 仁, 亀山, 仁史, 梅津, 哉
Format Journal Article
LanguageJapanese
Published 一般社団法人 日本消化器外科学会 01.03.2012
Online AccessGet full text
ISSN0386-9768
1348-9372
DOI10.5833/jjgs.45.326

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Abstract 症例は女性で,23歳・妊娠12週時より腹痛・嘔吐が出現した.画像上小腸全体の拡張を認めたが,器質的な閉塞機転を認めず,機能的な腸管運動不全が疑われた.出産後も症状は軽快せず,24歳時に回盲部切除術を施行された.病理組織学的検査所見で腸管神経節細胞脱落を認め,慢性特発性偽性腸閉塞症(chronic idiopathic intestinal pseudo-obstruction;以下,CIIPと略記)と診断された.25歳時にさらに大腸亜全摘・回腸人工肛門術を施行されたが,症状は改善しなかった.29歳時に空腸人工肛門造設術を施行後,症状が軽快し経口摂取可能となった.CIIPの発症機序は不明だが,近年その発症に自己免疫学的異常が関わるとする報告が散見される.一方,種々の自己免疫疾患と妊娠との関連が示唆されている.本症例もCIIP発症に自己免疫学的機序が関与しているかもしれない.
AbstractList 症例は女性で,23歳・妊娠12週時より腹痛・嘔吐が出現した.画像上小腸全体の拡張を認めたが,器質的な閉塞機転を認めず,機能的な腸管運動不全が疑われた.出産後も症状は軽快せず,24歳時に回盲部切除術を施行された.病理組織学的検査所見で腸管神経節細胞脱落を認め,慢性特発性偽性腸閉塞症(chronic idiopathic intestinal pseudo-obstruction;以下,CIIPと略記)と診断された.25歳時にさらに大腸亜全摘・回腸人工肛門術を施行されたが,症状は改善しなかった.29歳時に空腸人工肛門造設術を施行後,症状が軽快し経口摂取可能となった.CIIPの発症機序は不明だが,近年その発症に自己免疫学的異常が関わるとする報告が散見される.一方,種々の自己免疫疾患と妊娠との関連が示唆されている.本症例もCIIP発症に自己免疫学的機序が関与しているかもしれない.
Author 野上, 仁
関根, 和彦
亀山, 仁史
田島, 陽介
伏木, 麻恵
梅津, 哉
飯合, 恒夫
細井, 愛
畠山, 勝義
島田, 能史
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References 16) Lapointe R. Chronic idiopathic intestinal pseudo-obstruction treated by near total small bowel resection: a 20-year experience. J Gastrointest Surg. 2010 Dec;14(12):1937-42.
6) Fell JM, Smith VV, Milla PJ. Infantile chronic idiopathic intestinal pseudo-obstruction: the role of small intestinal manometry as a diagnostic tool and prognostic indicator. Gut. 1996 Aug;39(2):306-11.
13) Borchers AT, Naguwa SM, Keen CL, Gershwin ME. The implications of autoimmunity and pregnancy. J Autoimmun. 2010 May;34(3):J287-99.
15) Murr MM, Sarr MG, Camilleri M. The surgeon's role in the treatment of chronic intestinal pseudoobstruction. Am J Gastroenterol. 1995 Dec;90(12):2147-51.
11) De Giorgio R, Barbara G, Stanghellini V, De Ponti F, Salvioli B, Tonini M, et al. Clinical and morphofunctional features of idiopathic myenteric ganglionitis underlying severe intestinal motor dysfunction: a study of three cases. Am J Gastroenterol. 2002 Sep;97(9):2454-9.
14) Soudah HC, Hasler WL, Owyang C. Effect of octreotide on intestinal motility and bacterial overgrowth in scleroderma. N Engl J Med. 1991 Nov 21;325(21):1461-7.
8) De Giorgio R, Sarnelli G, Corinaldesi R, Stanghellini V. Advances in our understanding of the pathology of chronic intestinal pseudo-obstruction. Gut. 2004 Nov;53(11):1549-52.
12) Smith VV, Gregson N, Foggensteiner L, Neale G, Milla PJ. Acquired intestinal aganglionosis and circulating autoantibodies without neoplasia or other neural involvement. Gastroenterology. 1997 Apr;112(4):1366-71.
18) Lindberg G, Iwarzon M, Tornblom H. Clinical features and long-term survival in chronic intestinal pseudo-obstruction and enteric dysmotility. Scand J Gastroenterol. 2009;44(6):692-9.
17) Pironi L, Spinucci G, Paganelli F, Merli C, Masetti M, Miglioli M, et al. Italian guidelines for intestinal transplantation: potential candidates among the adult patients managed by a medical referral center for chronic intestinal failure. Transplant Proc. 2004 Apr;36(3):659-61.
2) Maldonado JE, Gregg JA, Green PA, Brown AL Jr. Chronic idiopathic intestinal pseudo-obstruction. Am J Med. 1970 Aug;49(2):203-12.
1) Dudley HA, Sinclair IS, McLaren IF, McNair TJ, Newsam JE. Intestinal pseudo-obstruction. J R Coll Surg Edinb. 1958 Mar;3(3):206-17.
9) Stanghellini V, Cogliandro RF, De Giorgio R, Barbara G, Salvioli B, Corinaldesi R. Chronic intestinal pseudo-obstruction: manifestations, natural history and management. Neurogastroenterol Motil. 2007 Jun;19(6):440-52.
7) De Giorgio R, Camilleri M. Human enteric neuropathies: morphology and molecular pathology. Neurogastroenterol Motil. 2004 Oct;16(5):515-31.
3) Lloyd DA, Vega R, Bassett P, Forbes A, Gabe SM. Survival and dependence on home parenteral nutrition: experience over a 25-year period in a UK referral centre. Aliment Pharmacol Ther. 2006 Oct 15;24(8):1231-40.
4) Stanghellini V, Corinaldesi R, Barbara L. Pseudo-obstruction syndromes. Baillieres Clin Gastroenterol. 1988 Jan;2(1):225-54.
10) Törnblom H, Lang B, Clover L, Knowles CH, Vincent A, Lindberg G. Autoantibodies in patients with gut motility disorders and enteric neuropathy. Scand J Gastroenterol. 2007 Nov;42(11):1289-93.
5) 厚生労働科学研究費補助金難治性疾患克服研究事業 慢性特発性偽性腸閉塞症(CIIP)の我が国における疫学・診断・治療の実態調査研究班.平成21年度総括・分担研究報告書
19) Ueno T, Fukuzawa M. Current status of intestinal transplantation. Surg Today. 2010 Dec;40(12):1112-22.
References_xml – reference: 19) Ueno T, Fukuzawa M. Current status of intestinal transplantation. Surg Today. 2010 Dec;40(12):1112-22.
– reference: 2) Maldonado JE, Gregg JA, Green PA, Brown AL Jr. Chronic idiopathic intestinal pseudo-obstruction. Am J Med. 1970 Aug;49(2):203-12.
– reference: 5) 厚生労働科学研究費補助金難治性疾患克服研究事業 慢性特発性偽性腸閉塞症(CIIP)の我が国における疫学・診断・治療の実態調査研究班.平成21年度総括・分担研究報告書.
– reference: 11) De Giorgio R, Barbara G, Stanghellini V, De Ponti F, Salvioli B, Tonini M, et al. Clinical and morphofunctional features of idiopathic myenteric ganglionitis underlying severe intestinal motor dysfunction: a study of three cases. Am J Gastroenterol. 2002 Sep;97(9):2454-9.
– reference: 7) De Giorgio R, Camilleri M. Human enteric neuropathies: morphology and molecular pathology. Neurogastroenterol Motil. 2004 Oct;16(5):515-31.
– reference: 3) Lloyd DA, Vega R, Bassett P, Forbes A, Gabe SM. Survival and dependence on home parenteral nutrition: experience over a 25-year period in a UK referral centre. Aliment Pharmacol Ther. 2006 Oct 15;24(8):1231-40.
– reference: 12) Smith VV, Gregson N, Foggensteiner L, Neale G, Milla PJ. Acquired intestinal aganglionosis and circulating autoantibodies without neoplasia or other neural involvement. Gastroenterology. 1997 Apr;112(4):1366-71.
– reference: 14) Soudah HC, Hasler WL, Owyang C. Effect of octreotide on intestinal motility and bacterial overgrowth in scleroderma. N Engl J Med. 1991 Nov 21;325(21):1461-7.
– reference: 17) Pironi L, Spinucci G, Paganelli F, Merli C, Masetti M, Miglioli M, et al. Italian guidelines for intestinal transplantation: potential candidates among the adult patients managed by a medical referral center for chronic intestinal failure. Transplant Proc. 2004 Apr;36(3):659-61.
– reference: 1) Dudley HA, Sinclair IS, McLaren IF, McNair TJ, Newsam JE. Intestinal pseudo-obstruction. J R Coll Surg Edinb. 1958 Mar;3(3):206-17.
– reference: 8) De Giorgio R, Sarnelli G, Corinaldesi R, Stanghellini V. Advances in our understanding of the pathology of chronic intestinal pseudo-obstruction. Gut. 2004 Nov;53(11):1549-52.
– reference: 4) Stanghellini V, Corinaldesi R, Barbara L. Pseudo-obstruction syndromes. Baillieres Clin Gastroenterol. 1988 Jan;2(1):225-54.
– reference: 10) Törnblom H, Lang B, Clover L, Knowles CH, Vincent A, Lindberg G. Autoantibodies in patients with gut motility disorders and enteric neuropathy. Scand J Gastroenterol. 2007 Nov;42(11):1289-93.
– reference: 13) Borchers AT, Naguwa SM, Keen CL, Gershwin ME. The implications of autoimmunity and pregnancy. J Autoimmun. 2010 May;34(3):J287-99.
– reference: 6) Fell JM, Smith VV, Milla PJ. Infantile chronic idiopathic intestinal pseudo-obstruction: the role of small intestinal manometry as a diagnostic tool and prognostic indicator. Gut. 1996 Aug;39(2):306-11.
– reference: 9) Stanghellini V, Cogliandro RF, De Giorgio R, Barbara G, Salvioli B, Corinaldesi R. Chronic intestinal pseudo-obstruction: manifestations, natural history and management. Neurogastroenterol Motil. 2007 Jun;19(6):440-52.
– reference: 15) Murr MM, Sarr MG, Camilleri M. The surgeon's role in the treatment of chronic intestinal pseudoobstruction. Am J Gastroenterol. 1995 Dec;90(12):2147-51.
– reference: 16) Lapointe R. Chronic idiopathic intestinal pseudo-obstruction treated by near total small bowel resection: a 20-year experience. J Gastrointest Surg. 2010 Dec;14(12):1937-42.
– reference: 18) Lindberg G, Iwarzon M, Tornblom H. Clinical features and long-term survival in chronic intestinal pseudo-obstruction and enteric dysmotility. Scand J Gastroenterol. 2009;44(6):692-9.
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Snippet 症例は女性で,23歳・妊娠12週時より腹痛・嘔吐が出現した.画像上小腸全体の拡張を認めたが,器質的な閉塞機転を認めず,機能的な腸管運動不全が疑われた.出産後も症...
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Title 妊娠を契機に発症した慢性特発性偽性腸閉塞症の1例
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