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3.
BMJ Case Rep ; 14(6)2021 Jun 29.
Article in English | MEDLINE | ID: mdl-34187804

ABSTRACT

Idiopathic megacolon (IMC) and idiopathic megarectum (IMR) describe an abnormality of the colon or rectum, characterised by a permanent dilatation of the bowel diameter in the absence of an identifiable cause. We present a 23-year-old woman with chronic constipation and excessive straining during defecation who presented at the emergency department in partial gut obstruction with a palpable fecaloma. Manual faecal disimpaction and a sigmoid loop colostomy was initially done. A full thickness rectal biopsy was positive for ganglion cells. Further workup led to the diagnosis of chronic IMC and IMR. The patient underwent laparoscopic modified Duhamel procedure, with an uneventful postoperative course.


Subject(s)
Hirschsprung Disease , Laparoscopy , Megacolon , Adult , Biopsy , Constipation/etiology , Constipation/surgery , Female , Hirschsprung Disease/surgery , Humans , Megacolon/diagnostic imaging , Megacolon/surgery , Rectum/surgery , Young Adult
5.
Rev Esp Enferm Dig ; 112(5): 423-424, 2020 May.
Article in English | MEDLINE | ID: mdl-32338024

ABSTRACT

We present the case of a 37-year-old male with constipation refractory to medical treatment, in the context of megacolon due to Chagas disease. The entire gastrointestinal tract may be affected but the digestive form is characterized by megaesophagus and megacolon. There is altered peristalsis due to the destruction of the neurons of the enteric nervous system caused by the parasite. Although the mortality rate is low, the disease can have a considerable impact upon quality of life. Chagas disease is poorly recognized and undertreated by healthcare providers in non-endemic regions.


Subject(s)
Chagas Disease , Enteric Nervous System , Esophageal Achalasia , Megacolon , Adult , Chagas Disease/complications , Esophageal Achalasia/etiology , Humans , Male , Megacolon/diagnostic imaging , Megacolon/etiology , Quality of Life
6.
Medicine (Baltimore) ; 99(4): e18803, 2020 Jan.
Article in English | MEDLINE | ID: mdl-31977872

ABSTRACT

RATIONALE: Intestinal hypoganglionosis most commonly presents in infancy or childhood, with only a few cases reported in adults. Those are mainly diagnosed after elective surgery for long-standing constipation and megacolon. PATIENT CONCERNS: We report a case of a 48-year-old female from China who presented with symptoms of discontinuation of bowel movements for 2 months. A hard, round mass could be felt in her right lower abdomen. DIAGNOSIS: The following examination methods diagnosed acquired segmental sigmoid hypoganglionosis. An abdominal computed tomography revealed a dilatation of the colon and suspicious wall thickening of the sigmoid colon. Anorectal manometry revealed relaxation of the anal sphincter. Histological examination revealed lower numbers and the degeneration of ganglion cells. INTERVENTIONS: Sigmoidectomy and transverse colostomy. OUTCOMES: The patient recovered well from surgery. Three months after the surgery, barium enema revealed a recovery in colorectal dilatation. LESSONS: This case could help raise awareness of acquired segmental hypoganglionosis. Resection of TZ and enterostomy presents an effective remission strategy for patients at risk of anastomotic leakage due to poor intestinal conditions.


Subject(s)
Colon, Transverse/diagnostic imaging , Constipation/diagnostic imaging , Sigmoid Diseases/diagnostic imaging , Anal Canal/innervation , Colon, Transverse/surgery , Colostomy , Constipation/surgery , Female , Humans , Megacolon/diagnostic imaging , Middle Aged , Sigmoid Diseases/surgery
7.
Clin J Gastroenterol ; 13(3): 328-333, 2020 Jun.
Article in English | MEDLINE | ID: mdl-31828729

ABSTRACT

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.


Subject(s)
Hirschsprung Disease/etiology , Megacolon/etiology , Rectal Neoplasms/surgery , Adult , Colon/pathology , Colonoscopy , Hirschsprung Disease/complications , Hirschsprung Disease/diagnostic imaging , Hirschsprung Disease/pathology , Humans , Male , Megacolon/diagnostic imaging , Megacolon/pathology , Radiography , Rectal Neoplasms/complications , Tomography, X-Ray Computed
9.
BMJ Case Rep ; 12(1)2019 Jan 14.
Article in English | MEDLINE | ID: mdl-30642858

ABSTRACT

Diffuse intestinal ganglioneuromatosis is a rare condition associated with MEN2B. It is also seen in conditions like neurofibromatosis type 1 and Cowden syndrome. This is a report of a patient who underwent total colectomy with end ileostomy creation for a megacolon. He was diagnosed to have diffuse ganglioneuromatosis on histological examination of the resected segment of colon. The definitive management of diffuse ganglioneuromatosis is to resect and anastomose.


Subject(s)
Constipation/diagnosis , Megacolon/pathology , Multiple Endocrine Neoplasia Type 2b/complications , Adult , Aftercare , Constipation/etiology , Diagnosis, Differential , Digestive System Neoplasms/pathology , Ganglioneuroma/pathology , Humans , Laparotomy/methods , Male , Megacolon/diagnostic imaging , Megacolon/surgery , Multiple Endocrine Neoplasia Type 2b/diagnosis , Multiple Endocrine Neoplasia Type 2b/pathology , Rare Diseases , Sri Lanka/ethnology , Treatment Outcome
11.
BMJ Case Rep ; 20182018 Aug 08.
Article in English | MEDLINE | ID: mdl-30093496

ABSTRACT

A 31-year-old male patient with chronic constipation of unknown aetiology presented emergently with worsening nausea, vomiting and abdominal distension of one week duration. On examination, his abdomen was distended with minimal tenderness. A plain film of the abdomen demonstrated severe faecal loading. The patient was haemodynamically unstable on admission and appeared sick. An urgent CT abdomen and pelvis was conducted showing extensive rectal dilatation and associated proximal colonic stercoral perforation. The patient proceeded straight to theatre for laparotomy as his general condition was deteriorating rapidly. On transfer to the operating table, the patient suffered cardiopulmonary arrest. Resuscitation was immediately commenced. Abdominal compartment syndrome was suspected. Cardiac output was re-established following a midline laparotomy which acted relieve the abdominal pressure. The rectosigmoid faecal content was decompressed via an enterotomy. The perforated segment of transverse colon was resected and an end colostomy fashioned. A year later, the continuity of the bowel was re-established.


Subject(s)
Intestinal Perforation/diagnosis , Intra-Abdominal Hypertension/diagnosis , Megacolon/diagnosis , Adult , Constipation/etiology , Diagnosis, Differential , Humans , Intestinal Perforation/complications , Intestinal Perforation/diagnostic imaging , Intestinal Perforation/surgery , Intra-Abdominal Hypertension/complications , Intra-Abdominal Hypertension/diagnostic imaging , Intra-Abdominal Hypertension/surgery , Laparotomy , Male , Megacolon/complications , Megacolon/diagnostic imaging , Megacolon/surgery , Tomography, X-Ray Computed
12.
Am J Case Rep ; 19: 557-561, 2018 May 14.
Article in English | MEDLINE | ID: mdl-29755106

ABSTRACT

BACKGROUND Intestinal hypoganglionosis is very rare and accounts for 3% to 5% of all classified congenital intestinal innervation disorders. Isolated hypoganglionosis of the colon is a particularly rare form of the disease, and differential diagnosis includes association with Hirschsprung's disease and chronic intestinal pseudo-obstruction (CIPO) related to visceral myopathies. Most cases are diagnosed at an early age or in childhood with only a few cases reported in adults. CASE REPORT We report a case of isolated hypoganglionosis of the rectum and sigmoid presenting as an emergency with acute intestinal obstruction in a 20-year-old male patient. A history of chronic constipation was reported since childhood, but this condition had never been investigated. A preoperative CT scan showed a megasigmoid and megarectum. A Hartmann's procedure was performed. The patient made a slow recovery and was discharged on the 12th postoperative day in good condition. Histology showed features consistent with isolated hypoganglionosis, and a full thickness rectal biopsies taken 2 months later confirmed the diagnosis. CONCLUSIONS Isolated hypoganglionosis in an adult is very rare, and a high index of suspicion is warranted in young patients with a history of chronic constipation to avoid delayed presentation as an emergency.


Subject(s)
Colon, Sigmoid/innervation , Dilatation, Pathologic/diagnostic imaging , Megacolon/diagnostic imaging , Rectum/diagnostic imaging , Rectum/innervation , Chronic Disease , Colon, Sigmoid/diagnostic imaging , Constipation/complications , Humans , Intestinal Obstruction/etiology , Male , Young Adult
13.
Arq Bras Cir Dig ; 31(1): e1341, 2018 Mar 01.
Article in English, Portuguese | MEDLINE | ID: mdl-29513802

ABSTRACT

BACKGROUND: Researches on Chagas disease still use several animals and rats, due to size and susceptibility were preferred by many authors. AIM: To develop an experimental model of megacolon in rats inoculated with the strain Y of Trypanosoma cruzi. METHODS: Thirty male Wistar rats were distributed in three groups inoculated with different inoculants: Group A: 600000, Group B: 1000000 and Group C: 1500000 blood trypomastigotes of T. cruzi. Animals were sedated intramuscularly at zero inoculation time (T0) and 60 days after inoculation (T60), to perform the barium enema in order to evaluate the dilatation of the different segments of colon in a comparative study of the measurements obtained, using a digital caliper. Evidence of infection was performed by blood smear collected from the animal's tail 18 days after inoculation with observation of blood forms. RESULTS: Comparing the intestinal diameter of the inoculated animals with 60,0000 trypomastigotes in the T0 of infection with T60 days after the inoculation, significant dilatation was observed between the proximal, medial and distal segments (p<0.01), indicating the establishment of the megacolon model. In addition, comparing intestinal diameter between the different segments, with in the T0 of infection and the T60 after inoculation, significant alterations were observed (p<0.05). CONCLUSION: The proposed model was possible for in vivo studies of alterations due to infection by T. cruzi and functional alterations of the colon. In addition, the changes manifested in the colon are not directly proportional to the size of the inoculum, but to the time of infection that the animals were submitted, since the animals inoculated with 60,0000 blood forms were the ones which presented the most significant alterations.


Subject(s)
Chagas Disease/diagnostic imaging , Megacolon/diagnostic imaging , Megacolon/parasitology , Animals , Barium Enema , Disease Models, Animal , Male , Rats , Rats, Wistar , Trypanosoma cruzi
16.
ABCD (São Paulo, Impr.) ; 31(1): e1341, 2018. tab, graf
Article in English | LILACS | ID: biblio-885754

ABSTRACT

ABSTRACT Background: Researches on Chagas disease still use several animals and rats, due to size and susceptibility were preferred by many authors. Aim: To develop an experimental model of megacolon in rats inoculated with the strain Y of Trypanosoma cruzi. Methods: Thirty male Wistar rats were distributed in three groups inoculated with different inoculants: Group A: 600000, Group B: 1000000 and Group C: 1500000 blood trypomastigotes of T. cruzi. Animals were sedated intramuscularly at zero inoculation time (T0) and 60 days after inoculation (T60), to perform the barium enema in order to evaluate the dilatation of the different segments of colon in a comparative study of the measurements obtained, using a digital caliper. Evidence of infection was performed by blood smear collected from the animal's tail 18 days after inoculation with observation of blood forms. Results: Comparing the intestinal diameter of the inoculated animals with 60,0000 trypomastigotes in the T0 of infection with T60 days after the inoculation, significant dilatation was observed between the proximal, medial and distal segments (p<0.01), indicating the establishment of the megacolon model. In addition, comparing intestinal diameter between the different segments, with in the T0 of infection and the T60 after inoculation, significant alterations were observed (p<0.05). Conclusion: The proposed model was possible for in vivo studies of alterations due to infection by T. cruzi and functional alterations of the colon. In addition, the changes manifested in the colon are not directly proportional to the size of the inoculum, but to the time of infection that the animals were submitted, since the animals inoculated with 60,0000 blood forms were the ones which presented the most significant alterations.


RESUMO Racional: Pesquisas para doença de Chagas ainda utilizam diversos animais e o rato por seu tamanho e sua suscetibilidade foi o preferido por muitos pesquisadores. Objetivo: Desenvolver um modelo experimental de megacólon em ratos inoculados com a cepa Y de Trypanosoma cruzi. Métodos: Utilizou-se 30 ratos, machos, distribuídos em três grupos inoculados com diferentes inóculos: Grupo A: 600000, Grupo B: 1000000 e Grupo C: 1500000 tripomastigotas sanguíneos da cepa Y de T. cruzi. Os animais foram sedados via intramuscular no tempo zero de inoculação (T0) e aos 60 dias após a inoculação (T60) para realização de enema opaco para avaliação da dilatação dos diferentes segmentos do cólon em estudo comparativo das medidas obtidas, com o auxílio de um paquímetro digital. A comprovação da infecção foi realizada com esfregaço de sangue coletado a partir da cauda do animal 18 dias após a inoculação com observação das formas sanguíneas. Resultados: Ao comparar o diâmetro intestinal dos animais inoculados com 60.0000 formas tripomastigotas no T0 de infecção com T60 dias após a inoculação, observou-se dilatação significativa entre os segmentos proximal, medial e distal (p<0,01), indicando o estabelecimento do modelo de megacólon. Além disso, ao comparar o diâmetro intestinal entre os diferentes segmentos, dentro do T0 de infecção e do T60 após a inoculação, observou-se alterações significantes (p<0,05). Conclusões: O modelo proposto mostrou-se factível para estudos in vivo das alterações decorrentes da infecção pelo T. cruzi e alterações funcionais do cólon. Além disso, as alterações manifestadas no cólon não são diretamente proporcionais ao tamanho do inóculo, mas sim ao tempo de infecção que os animais foram submetidos, visto que os inoculados com 600000 formas sanguíneas foram as que mais apresentaram alterações significantes.


Subject(s)
Animals , Male , Rats , Chagas Disease/diagnostic imaging , Megacolon/parasitology , Megacolon/diagnostic imaging , Trypanosoma cruzi , Rats, Wistar , Disease Models, Animal , Barium Enema
17.
Clin Nucl Med ; 42(4): 312-314, 2017 Apr.
Article in English | MEDLINE | ID: mdl-28134699

ABSTRACT

A 7-year-old boy with chief complaint of chronic pelvic pain was referred to our nuclear medicine department for bone scintigraphy. The images showed a focus of radiotracer activity in the right side of pelvic cavity, which is further confirmed as urinary bladder by single-photon emission computed tomography (SPECT) and delayed images. Because of high possibility of mass effect in pelvic cavity, pelvic magnetic resonance imaging (MRI) was performed, and it revealed an unusual dilatation of rectosigmoid colon with no evidence of pelvic mass.


Subject(s)
Megacolon/diagnostic imaging , Tomography, Emission-Computed, Single-Photon , Urinary Bladder/diagnostic imaging , Child , Humans , Magnetic Resonance Imaging , Male , Megacolon/pathology , Pelvis/diagnostic imaging , Urinary Bladder/pathology
19.
J Pediatr Gastroenterol Nutr ; 62(2): 259-63, 2016 Feb.
Article in English | MEDLINE | ID: mdl-26196204

ABSTRACT

BACKGROUND: Abdominal x-ray (AXR) can identify complications in acute severe colitis (ASC) and may assist in selecting high-risk children for early aggressive treatment. We aimed to describe AXR findings in pediatric ASC and to explore radiological predictors of response to intravenous corticosteroid (IVCS) therapy. METHODS: A total of 56 children with ASC were included in a multicenter, retrospective 1-year cohort study (41% boys, mean age 12.1 ±â€Š4.2). Radiographs of responders to IVCS and those requiring second-line salvage therapy by discharge were analyzed independently by 2 blinded radiologists. RESULTS: A total of 33 responders to IVCS were compared with 23 nonresponders. The day-3 Pediatric Ulcerative Colitis Activity Index (PUCAI) score was significantly higher in nonresponders (63 ±â€Š16 vs 46 ±â€Š21, P = 0.001). The mean transverse colon luminal diameter was 30 ±â€Š16 mm in responders and 38 ±â€Š16 mm in nonresponders (P = 0.94). The upper range of transverse colonic diameter in children <12 years was ∼40 mm, whereas in older children it was 60 mm as accepted in adults. Ulcerations and megacolon seen on AXR were associated with nonresponse to IVCS (P = 0.006 and 0.064, respectively). CONCLUSIONS: The presence of mucosal ulcerations and megacolon on AXR could be considered in the risk stratification of children with ASC for early aggressive treatment, together with the previously known day-3 and day-5 Pediatric Ulcerative Colitis Activity Index scores, albumin, and C-reactive protein.


Subject(s)
Adrenal Cortex Hormones/therapeutic use , Colitis, Ulcerative/drug therapy , Colitis/drug therapy , Colon/pathology , Intestinal Mucosa/pathology , Megacolon/pathology , Adolescent , Adrenal Cortex Hormones/administration & dosage , Albumins/metabolism , C-Reactive Protein/metabolism , Child , Colitis/complications , Colitis/pathology , Colitis, Ulcerative/complications , Colitis, Ulcerative/pathology , Colon/diagnostic imaging , Colon, Transverse/diagnostic imaging , Colon, Transverse/pathology , Female , Humans , Intestinal Mucosa/diagnostic imaging , Male , Megacolon/complications , Megacolon/diagnostic imaging , Radiography, Abdominal/methods , Retrospective Studies , Severity of Illness Index , Treatment Outcome , X-Rays
20.
Ir Med J ; 108(5): 150-1, 2015 May.
Article in English | MEDLINE | ID: mdl-26062244

ABSTRACT

Chagas disease is a rare condition but with an increasing incidence. Megacolon is a known sequelae. Surgical management remains the only disease modifying treatment option; with variable long-term success. We highlight an interesting case to emphasize attention to this rare condition as a differential diagnosis in any patient presenting with massive intestinal dilatation.


Subject(s)
Chagas Disease , Colectomy/methods , Colon , Ganglia, Parasympathetic/parasitology , Megacolon , Trypanosoma cruzi/immunology , Aged , Chagas Disease/complications , Chagas Disease/diagnosis , Colon/innervation , Colon/pathology , Diagnosis, Differential , Humans , Immunohistochemistry , Male , Megacolon/diagnostic imaging , Megacolon/etiology , Megacolon/surgery , Radiography , Treatment Outcome
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