OBJECTIVE: Many patients, especially women, suffer from severe gastrointestinal pain and dysmotility for several years without being diagnosed. Depletion of gonadotropin-releasing hormone (GnRH) in the enteric nervous system (ENS) has been described in some patients. The aim of this study was to examine the expression of GnRH in ENS and antibodies against GnRH in serum, in a dysmotility patient cohort of southern Sweden. MATERIALS AND METHODS: All consecutive patients (n = 35) referred for laparoscopic full-thickness biopsy because of symptoms or signs of severe dysmotility between 1998 and 2009, or patients with a severe dysmotility disorder having had a bowel resection within the time frame, were considered for inclusion. In 22 cases, representative biopsy material containing ganglia was available, and these patients were included. Medical records were scrutinized. The expression of GnRH was determined by immunohistochemistry in bowel biopsies from these patients and in patients with carcinoma or diverticulosis without ENS histopathology. Antibodies against GnRH in serum were determined by ELISA in patients and controls. RESULTS: 14 patients were diagnosed with enteric dysmotility (ED) and 8 with chronic intestinal pseudo-obstruction due to varying etiology. Immunostained biopsies showed expression of GnRH in the ENS. A reduced expression of GnRH-containing neurons was found in 5 patients, as well as antibodies against GnRH in serum. 3 of these patients had a history of in vitro fertilization (IVF) using GnRH analogs. CONCLUSIONS: A subgroup of patients with severe dysmotility had a reduced expression of GnRH-containing neurons in the ENS and expressed antibodies against GnRH in serum.
OBJECTIVE: Many patients, especially women, suffer from severe gastrointestinal pain and dysmotility for several years without being diagnosed. Depletion of gonadotropin-releasing hormone (GnRH) in the enteric nervous system (ENS) has been described in some patients. The aim of this study was to examine the expression of GnRH in ENS and antibodies against GnRH in serum, in a dysmotilitypatient cohort of southern Sweden. MATERIALS AND METHODS: All consecutive patients (n = 35) referred for laparoscopic full-thickness biopsy because of symptoms or signs of severe dysmotility between 1998 and 2009, or patients with a severe dysmotility disorder having had a bowel resection within the time frame, were considered for inclusion. In 22 cases, representative biopsy material containing ganglia was available, and these patients were included. Medical records were scrutinized. The expression of GnRH was determined by immunohistochemistry in bowel biopsies from these patients and in patients with carcinoma or diverticulosis without ENS histopathology. Antibodies against GnRH in serum were determined by ELISA in patients and controls. RESULTS: 14 patients were diagnosed with enteric dysmotility (ED) and 8 with chronic intestinal pseudo-obstruction due to varying etiology. Immunostained biopsies showed expression of GnRH in the ENS. A reduced expression of GnRH-containing neurons was found in 5 patients, as well as antibodies against GnRH in serum. 3 of these patients had a history of in vitro fertilization (IVF) using GnRH analogs. CONCLUSIONS: A subgroup of patients with severe dysmotility had a reduced expression of GnRH-containing neurons in the ENS and expressed antibodies against GnRH in serum.
Authors: Nurit Assia Batzir; Pranjali Kishor Bhagwat; Austin Larson; Zeynep Coban Akdemir; Maciej Bagłaj; Leon Bofferding; Katherine B Bosanko; Skander Bouassida; Bert Callewaert; Ashley Cannon; Yazmin Enchautegui Colon; Adolfo D Garnica; Margaret H Harr; Sandra Heck; Anna C E Hurst; Shalini N Jhangiani; Bertrand Isidor; Rebecca O Littlejohn; Pengfei Liu; Pilar Magoulas; Helen Mar Fan; Ronit Marom; Scott McLean; Marjan M Nezarati; Kimberly M Nugent; Michael B Petersen; Maria L Rocha; Elizabeth Roeder; Robert Smigiel; Ian Tully; James Weisfeld-Adams; Katerina O Wells; Jennifer E Posey; James R Lupski; Arthur L Beaudet; Michael F Wangler Journal: Hum Mutat Date: 2019-12-19 Impact factor: 4.878