Primary presentation of Jeune's syndrome as gastric motility disorder in an infant: A case report

We report a case of a 4-week-old female neonate with Jeune's asphyxiating thoracic dystrophy (JATD) and coexistent situs anomaly, primarily presenting as gastric motility disorder. The child presented with abdominal distension and nonbilious vomiting since birth with failure to thrive. However,...

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Autores principales: Katyan, Amit, Grover, Shabnam Bhandari, Rajani, Heena, Bagga, Deepak, Antil, Neha
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Medknow Publications & Media Pvt Ltd 2018
Materias:
Gastrointestinal Radiology and Hepatology
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5894323/
https://www.ncbi.nlm.nih.gov/pubmed/29692530
http://dx.doi.org/10.4103/ijri.IJRI_303_17
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author Katyan, Amit
Grover, Shabnam Bhandari
Rajani, Heena
Bagga, Deepak
Antil, Neha
author_facet Katyan, Amit
Grover, Shabnam Bhandari
Rajani, Heena
Bagga, Deepak
Antil, Neha
author_sort Katyan, Amit
collection PubMed
description We report a case of a 4-week-old female neonate with Jeune's asphyxiating thoracic dystrophy (JATD) and coexistent situs anomaly, primarily presenting as gastric motility disorder. The child presented with abdominal distension and nonbilious vomiting since birth with failure to thrive. However, skeletal survey revealed JATD. Upper gastrointestinal contrast study showed situs inversus with delayed gastric emptying. Pyloric biopsy and intraoperative antro-duodenal manometry confirmed association of gastric motility disorder. Awareness of the unusual possibility of primary presentation of Jeune syndrome as gastric motility disorder will improve the management approach in such infants.
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spelling pubmed-58943232018-04-24 Primary presentation of Jeune's syndrome as gastric motility disorder in an infant: A case report Katyan, Amit Grover, Shabnam Bhandari Rajani, Heena Bagga, Deepak Antil, Neha Indian J Radiol Imaging Gastrointestinal Radiology and Hepatology We report a case of a 4-week-old female neonate with Jeune's asphyxiating thoracic dystrophy (JATD) and coexistent situs anomaly, primarily presenting as gastric motility disorder. The child presented with abdominal distension and nonbilious vomiting since birth with failure to thrive. However, skeletal survey revealed JATD. Upper gastrointestinal contrast study showed situs inversus with delayed gastric emptying. Pyloric biopsy and intraoperative antro-duodenal manometry confirmed association of gastric motility disorder. Awareness of the unusual possibility of primary presentation of Jeune syndrome as gastric motility disorder will improve the management approach in such infants. Medknow Publications & Media Pvt Ltd 2018 /pmc/articles/PMC5894323/ /pubmed/29692530 http://dx.doi.org/10.4103/ijri.IJRI_303_17 Text en Copyright: © 2018 Indian Journal of Radiology and Imaging http://creativecommons.org/licenses/by-nc-sa/4.0 This is an open access journal, and articles are distributed under the terms of the Creative Commons Attribution-NonCommercial-ShareAlike 4.0 License, which allows others to remix, tweak, and build upon the work non-commercially, as long as appropriate credit is given and the new creations are licensed under the identical terms.
spellingShingle Gastrointestinal Radiology and Hepatology
Katyan, Amit
Grover, Shabnam Bhandari
Rajani, Heena
Bagga, Deepak
Antil, Neha
Primary presentation of Jeune's syndrome as gastric motility disorder in an infant: A case report
title Primary presentation of Jeune's syndrome as gastric motility disorder in an infant: A case report
title_full Primary presentation of Jeune's syndrome as gastric motility disorder in an infant: A case report
title_fullStr Primary presentation of Jeune's syndrome as gastric motility disorder in an infant: A case report
title_full_unstemmed Primary presentation of Jeune's syndrome as gastric motility disorder in an infant: A case report
title_short Primary presentation of Jeune's syndrome as gastric motility disorder in an infant: A case report
title_sort primary presentation of jeune's syndrome as gastric motility disorder in an infant: a case report
topic Gastrointestinal Radiology and Hepatology
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5894323/
https://www.ncbi.nlm.nih.gov/pubmed/29692530
http://dx.doi.org/10.4103/ijri.IJRI_303_17
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