TY - JOUR
T1 - A case of severe megacolon due to acquired isolated hypoganglionosis after low anterior resection for lower rectal cancer
AU - Tominaga, Tetsuro
AU - Nagayama, Satoshi
AU - Takamatsu, Manabu
AU - Miyanari, Shun
AU - Nagasaki, Toshiya
AU - Yamaguchi, Tomohiro
AU - Akiyoshi, Takashi
AU - Konishi, Tsuyoshi
AU - Fujimoto, Yoshiya
AU - Fukunaga, Yosuke
AU - Ueno, Masashi
N1 - Publisher Copyright:
© 2019, The Author(s).
PY - 2020/6/1
Y1 - 2020/6/1
N2 - Acquired isolated hypoganglionosis is a rare intestinal neurological disease, which presents in adulthood with the clinical symptoms of chronic constipation. A 39-year-old man underwent laparoscopic low anterior resection and covering ileostomy for locally advanced-rectal cancer. A 6-month course of postoperative adjuvant chemotherapy was completed, followed by closure of the ileostoma. After the closure, he developed severe colitis which required 1-month of hospitalization. Mucosal erosions and pseudo-membrane formation were evident on colonoscopy and severe mucosal damage characterized by infiltration of inflammatory cells and crypt degeneration were pathologically confirmed. Even after the remission of the colitis, he suffered from severe constipation and distention. At 4 years after the stoma closure, he decided to undergo laparoscopic total colectomy. Histopathologically, the nerve fibers and ganglion cells became gradually scarcer from the non-dilated to dilated regions. Immunohistochemical staining examination confirmed that the ganglion cells gradually decreased and became degenerated from the normal to dilated region, thereby arriving at the final diagnosis of isolated hypoganglionosis. The patient recovered without any complications and there has been no evidence of any relapse of the symptoms. We present a case of acquired isolated hypoganglionosis-related megacolon, which required laparoscopic total colectomy, due to severe enterocolitis following stoma closure.
AB - Acquired isolated hypoganglionosis is a rare intestinal neurological disease, which presents in adulthood with the clinical symptoms of chronic constipation. A 39-year-old man underwent laparoscopic low anterior resection and covering ileostomy for locally advanced-rectal cancer. A 6-month course of postoperative adjuvant chemotherapy was completed, followed by closure of the ileostoma. After the closure, he developed severe colitis which required 1-month of hospitalization. Mucosal erosions and pseudo-membrane formation were evident on colonoscopy and severe mucosal damage characterized by infiltration of inflammatory cells and crypt degeneration were pathologically confirmed. Even after the remission of the colitis, he suffered from severe constipation and distention. At 4 years after the stoma closure, he decided to undergo laparoscopic total colectomy. Histopathologically, the nerve fibers and ganglion cells became gradually scarcer from the non-dilated to dilated regions. Immunohistochemical staining examination confirmed that the ganglion cells gradually decreased and became degenerated from the normal to dilated region, thereby arriving at the final diagnosis of isolated hypoganglionosis. The patient recovered without any complications and there has been no evidence of any relapse of the symptoms. We present a case of acquired isolated hypoganglionosis-related megacolon, which required laparoscopic total colectomy, due to severe enterocolitis following stoma closure.
KW - Acquired isolated hypoganglionosis
KW - Laparoscopic total colectomy
KW - Megacolon
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U2 - 10.1007/s12328-019-01079-2
DO - 10.1007/s12328-019-01079-2
M3 - Article
C2 - 31828729
AN - SCOPUS:85076414629
SN - 1865-7257
VL - 13
SP - 328
EP - 333
JO - Clinical Journal of Gastroenterology
JF - Clinical Journal of Gastroenterology
IS - 3
ER -