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

The safety and scientific validity of this study is the responsibility of the study sponsor and investigators. Listing a study does not mean it has been evaluated by the U.S. Federal Government. Read our disclaimer for details.
 
ClinicalTrials.gov Identifier: NCT04363125
Recruitment Status : Completed
First Posted : April 27, 2020
Last Update Posted : April 28, 2020
Sponsor:
Collaborator:
Inonu University
Information provided by (Responsible Party):
Ufuk Uylas, Dr. Ersin Arslan Education and Training Hospital

Brief Summary:
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.

Condition or disease Intervention/treatment
Pathology Sigmoid Volvulus Hypoganglionosis Diagnostic Test: Pathology result

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.

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Study Type : Observational
Actual Enrollment : 310 participants
Observational Model: Case-Only
Time Perspective: Other
Official Title: Systematic Review of Isolated Hypoganglionosis and Presentation of a New Case Complicated With Sigmoid Volvulus
Actual Study Start Date : January 1, 2019
Actual Primary Completion Date : April 1, 2020
Actual Study Completion Date : April 20, 2020

Resource links provided by the National Library of Medicine



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


Primary Outcome Measures :
  1. Mortality [ Time Frame: three months ]
    Mortality of sigmoid volvulus due to isolated hypoganglionosis



Information from the National Library of Medicine

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Ages Eligible for Study:   Child, Adult, Older Adult
Sexes Eligible for Study:   All
Accepts Healthy Volunteers:   No
Sampling Method:   Non-Probability Sample
Study Population
our case of isolated hypoganglionosis and published isolated hypoganglionosis studies patients.
Criteria

Inclusion Criteria:

  • isolated hypoganglionosis
  • our case of isolated hypoganglionosis

Exclusion Criteria:

  • hypoganglionosis with Hirschsprung's disease

Information from the National Library of Medicine

To learn more about this study, you or your doctor may contact the study research staff using the contact information provided by the sponsor.

Please refer to this study by its ClinicalTrials.gov identifier (NCT number): NCT04363125


Locations
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Turkey
Dr. Ersin Arslan Training and Resource Hospital
Gaziantep, Turkey
Sponsors and Collaborators
Dr. Ersin Arslan Education and Training Hospital
Inonu University
Investigators
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Principal Investigator: ufuk uylas, Specialist Dr. Ersin Arslan Training and Resource Hospital
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Responsible Party: Ufuk Uylas, Principal Investigator, Dr. Ersin Arslan Education and Training Hospital
ClinicalTrials.gov Identifier: NCT04363125    
Other Study ID Numbers: 23042020
First Posted: April 27, 2020    Key Record Dates
Last Update Posted: April 28, 2020
Last Verified: April 2020

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Studies a U.S. FDA-regulated Drug Product: No
Studies a U.S. FDA-regulated Device Product: No
Keywords provided by Ufuk Uylas, Dr. Ersin Arslan Education and Training Hospital:
colon
constipation
derotation
obstruction
Hypoganglionosis
intestinal neuronal dysplasia
Additional relevant MeSH terms:
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Intestinal Volvulus
Intestinal Obstruction
Intestinal Diseases
Gastrointestinal Diseases
Digestive System Diseases
Torsion Abnormality
Pathological Conditions, Anatomical