Review of Isolated Hypoganglionosis and Presentation of a New Case Complicated With Sigmoid Volvulus

Overview

Isolated hypoganglionosis is rare and constitutes 3-5% of all congenital neurological bowel diseases. Since hypoganglionosis is associated with symptoms such as chronic constipation or pseudo-obstruction, it can be confused with Hirschprung's disease and it is important to make a differential diagnosis before treatment. Our aim in this study; In addition to sharing the rarely isolated hypoganglionosis and sigmoid volvulus in a 45-year-old male patient with mental retardation, the investigators present a systematic comprehensive review of hypoganglionosis.

Full Title of Study: “Systematic Review of Isolated Hypoganglionosis and Presentation of a New Case Complicated With Sigmoid Volvulus”

Study Type

  • Study Type: Observational
  • Study Design
    • Time Perspective: Other
  • Study Primary Completion Date: April 1, 2020

Detailed Description

Isolated hypoganglionosis is rare and constitutes 3-5% of all congenital neurological bowel diseases. Since hypoganglionosis is associated with symptoms such as chronic constipation or pseudo-obstruction, it can be confused with Hirschprung's disease and it is important to make a differential diagnosis before treatment. Our aim in this study; In addition to sharing the rarely isolated hypoganglionosis and sigmoid volvulus in a 45-year-old male patient with mental retardation, the investigators present a systematic comprehensive review of hypoganglionosis. Pubmed and Scopus databases were scanned. No restrictions were used in the written language. Only isolated cases of hypoganglionosis were added to the study. In the cases; author, year of publication, country of publication, age, gender, surgical or non-surgical treatments, treatment results, and mortalities were examined. When sigmoid volvulus is seen in children and adolescents, congenital neurological bowel diseases should be considered in the differential diagnosis. Hirschprung's disease must be ruled out by rectal biopsy. The histopathological investigation should be carried out for the diagnosis and determination of surgical margins in isolated hypoganglionosis.

Interventions

  • Diagnostic Test: Pathology result
    • Pathology result of rectal biopsy

Clinical Trial Outcome Measures

Primary Measures

  • Mortality
    • Time Frame: three months
    • Mortality of sigmoid volvulus due to isolated hypoganglionosis

Participating in This Clinical Trial

Inclusion Criteria

  • isolated hypoganglionosis – our case of isolated hypoganglionosis Exclusion Criteria:

  • hypoganglionosis with Hirschsprung's disease

Gender Eligibility: All

Minimum Age: N/A

Maximum Age: N/A

Are Healthy Volunteers Accepted: No

Investigator Details

  • Lead Sponsor
    • Dr. Ersin Arslan Education and Training Hospital
  • Collaborator
    • Inonu University
  • Provider of Information About this Clinical Study
    • Principal Investigator: Ufuk Uylas, Principal Investigator – Dr. Ersin Arslan Education and Training Hospital
  • Overall Official(s)
    • ufuk uylas, Specialist, Principal Investigator, Dr. Ersin Arslan Training and Resource Hospital

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