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Neonatal intestinal obstruction associated with situs inversus totalis: two case reports and a review of the literature

BACKGROUND: The association of neonatal intestinal obstruction with situs inversus totalis is extremely rare with only few cases reported in the literature to date. This association poses dilemmas in management. We present two such cases (of Indian origin), and briefly discuss the pertinent literatu...

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Autores principales: Gupta, Rahul, Soni, Varsha, Valse, Prakash Devidas, Goyal, Ram Babu, Gupta, Arun Kumar, Mathur, Praveen
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2017
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5603179/
https://www.ncbi.nlm.nih.gov/pubmed/28918753
http://dx.doi.org/10.1186/s13256-017-1423-z
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author Gupta, Rahul
Soni, Varsha
Valse, Prakash Devidas
Goyal, Ram Babu
Gupta, Arun Kumar
Mathur, Praveen
author_facet Gupta, Rahul
Soni, Varsha
Valse, Prakash Devidas
Goyal, Ram Babu
Gupta, Arun Kumar
Mathur, Praveen
author_sort Gupta, Rahul
collection PubMed
description BACKGROUND: The association of neonatal intestinal obstruction with situs inversus totalis is extremely rare with only few cases reported in the literature to date. This association poses dilemmas in management. We present two such cases (of Indian origin), and briefly discuss the pertinent literature and measures to prevent unfavorable outcome. CASE PRESENTATION: Case 1: a 1-month-old preterm (1300 g) male neonate belonging to Hindu (Indian) ethnicity presented with recurrent bile-stained vomiting, non-passage of stools and epigastric fullness. A babygram and upper gastrointestinal contrast studies revealed situs inversus and suggested proximal jejunal obstruction with midgut volvulus. Exploration confirmed situs inversus totalis along with reverse rotation and midgut volvulus. There was a small gangrenous area in the proximal jejunal loop. A Ladd’s procedure, resection of the gangrenous jejunal loop, and jejunojejunal anastomosis was performed. Note was made of the unusual appearance of the intestines suggestive either of fibrous or fatty infiltration. Postoperatively, our patient developed septicemia and died. Case 2: a 4-day-old female neonate belonging to Hindu (Indian) ethnicity, small (1320 g) for gestation, presented with history of non-passage of meconium since birth, refusal to accept feeds, and episodes of recurrent bilious vomiting with abdominal distension. A plain radiogram revealed situs inversus and proximal jejunal obstruction. Ultrasonography of her abdomen revealed renal dysplastic changes in both her kidneys. Laparotomy confirmed multiple jejunoileal atresias with situs inversus totalis. Resection anastomoses was performed for multiple atresias. Our patient passed a few pellets of meconium stools postoperatively; feeds were started gradually on the sixth day. Our patient gradually developed oliguria and renal failure, followed by respiratory distress and generalized edema requiring ventilatory support. She died later due to multiorgan failure. CONCLUSIONS: Clinicians should have high index of suspicion for malrotation with midgut volvulus or intestinal atresias in neonates of situs inversus presenting with bilious vomiting. The surgical treatment should follow the same surgical principles. In situs inversus, because of transposition of viscera, midgut volvulus may occur in an anticlockwise direction, hence derotation is performed clockwise. Prognosis was poor in our series because of low birth weight, late presentation, presence of gangrenous locus in the small bowel and development of septicemia in our first case and multiorgan fibrosis/dysplasia in our second case. Early diagnosis and timely referral is paramount for favorable outcome.
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spelling pubmed-56031792017-09-21 Neonatal intestinal obstruction associated with situs inversus totalis: two case reports and a review of the literature Gupta, Rahul Soni, Varsha Valse, Prakash Devidas Goyal, Ram Babu Gupta, Arun Kumar Mathur, Praveen J Med Case Rep Case Report BACKGROUND: The association of neonatal intestinal obstruction with situs inversus totalis is extremely rare with only few cases reported in the literature to date. This association poses dilemmas in management. We present two such cases (of Indian origin), and briefly discuss the pertinent literature and measures to prevent unfavorable outcome. CASE PRESENTATION: Case 1: a 1-month-old preterm (1300 g) male neonate belonging to Hindu (Indian) ethnicity presented with recurrent bile-stained vomiting, non-passage of stools and epigastric fullness. A babygram and upper gastrointestinal contrast studies revealed situs inversus and suggested proximal jejunal obstruction with midgut volvulus. Exploration confirmed situs inversus totalis along with reverse rotation and midgut volvulus. There was a small gangrenous area in the proximal jejunal loop. A Ladd’s procedure, resection of the gangrenous jejunal loop, and jejunojejunal anastomosis was performed. Note was made of the unusual appearance of the intestines suggestive either of fibrous or fatty infiltration. Postoperatively, our patient developed septicemia and died. Case 2: a 4-day-old female neonate belonging to Hindu (Indian) ethnicity, small (1320 g) for gestation, presented with history of non-passage of meconium since birth, refusal to accept feeds, and episodes of recurrent bilious vomiting with abdominal distension. A plain radiogram revealed situs inversus and proximal jejunal obstruction. Ultrasonography of her abdomen revealed renal dysplastic changes in both her kidneys. Laparotomy confirmed multiple jejunoileal atresias with situs inversus totalis. Resection anastomoses was performed for multiple atresias. Our patient passed a few pellets of meconium stools postoperatively; feeds were started gradually on the sixth day. Our patient gradually developed oliguria and renal failure, followed by respiratory distress and generalized edema requiring ventilatory support. She died later due to multiorgan failure. CONCLUSIONS: Clinicians should have high index of suspicion for malrotation with midgut volvulus or intestinal atresias in neonates of situs inversus presenting with bilious vomiting. The surgical treatment should follow the same surgical principles. In situs inversus, because of transposition of viscera, midgut volvulus may occur in an anticlockwise direction, hence derotation is performed clockwise. Prognosis was poor in our series because of low birth weight, late presentation, presence of gangrenous locus in the small bowel and development of septicemia in our first case and multiorgan fibrosis/dysplasia in our second case. Early diagnosis and timely referral is paramount for favorable outcome. BioMed Central 2017-09-18 /pmc/articles/PMC5603179/ /pubmed/28918753 http://dx.doi.org/10.1186/s13256-017-1423-z Text en © The Author(s). 2017 Open AccessThis article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.
spellingShingle Case Report
Gupta, Rahul
Soni, Varsha
Valse, Prakash Devidas
Goyal, Ram Babu
Gupta, Arun Kumar
Mathur, Praveen
Neonatal intestinal obstruction associated with situs inversus totalis: two case reports and a review of the literature
title Neonatal intestinal obstruction associated with situs inversus totalis: two case reports and a review of the literature
title_full Neonatal intestinal obstruction associated with situs inversus totalis: two case reports and a review of the literature
title_fullStr Neonatal intestinal obstruction associated with situs inversus totalis: two case reports and a review of the literature
title_full_unstemmed Neonatal intestinal obstruction associated with situs inversus totalis: two case reports and a review of the literature
title_short Neonatal intestinal obstruction associated with situs inversus totalis: two case reports and a review of the literature
title_sort neonatal intestinal obstruction associated with situs inversus totalis: two case reports and a review of the literature
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5603179/
https://www.ncbi.nlm.nih.gov/pubmed/28918753
http://dx.doi.org/10.1186/s13256-017-1423-z
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