Home | About Us | Current Issue | Ahead of print | Archives | Search | Instructions | Subscription | Feedback | Editorial Board | e-Alerts | Login 
Journal of Indian Association of Pediatric Surgeons
     Journal of Indian Association of Pediatric Surgeons
Official journal of the Indian Association of Pediatric Surgeons         
 Users Online:378 
  Print this page Email this page   Small font sizeDefault font sizeIncrease font size


 
CASE REPORT
Year : 2007  |  Volume : 12  |  Issue : 4  |  Page : 228-230
 

Concurrent dextrogastria, reverse midgut rotation and intestinal atresia in a neonate


Department of Surgery, University of Ilorin Teaching Hospital, Ilorin, Kwara State, Nigeria

Correspondence Address:
L O Abdur-Rahman
P. O. Box 5291, Ilorin - 240001
Nigeria
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0971-9261.40843

Rights and Permissions

 

   Abstract 

A case of dextrogastria associated with multiple jejunal atresia and inverse rotation of the bowel in a neonate is presented. The management of this rare condition is highlighted with a review of the literature.


Keywords: Dextrogastria, intestinal atresia, malrotation


How to cite this article:
Abdur-Rahman L O, Adeniran J O, Taiwo J O. Concurrent dextrogastria, reverse midgut rotation and intestinal atresia in a neonate. J Indian Assoc Pediatr Surg 2007;12:228-30

How to cite this URL:
Abdur-Rahman L O, Adeniran J O, Taiwo J O. Concurrent dextrogastria, reverse midgut rotation and intestinal atresia in a neonate. J Indian Assoc Pediatr Surg [serial online] 2007 [cited 2017 Oct 23];12:228-30. Available from: http://www.jiaps.com/text.asp?2007/12/4/228/40843



   Introduction Top


Neonatal intestinal obstruction is a relatively common anomaly in pediatric surgical practice. The common etiologic factors in our region are anorectal malformations, Hirschsprung's disease and jejunoileal atresias. [1],[2],[3] Situs inversus occurs in approximately 1 in 8,500 people; however, the incidence of situs inversus abdominus (SIA) varies between 1 in 4,000 and 1 in 20,000 live births and may present a diagnostic and management challenge to the clinician and radiologist. [4] Situs ambiguous or heterotaxy implies a disordered organ arrangement in the chest or abdomen, whereas complete situs inversus implies that the organs of the chest and abdomen are arranged in opposite positions (the heart is on the right (dextrocardia) as is the two-lobed lung, while the liver, spleen and three-lobed lung are on the left). We report a case of neonatal intestinal obstruction from multiple intestinal atresias and the inverse rotation of midgut in a patient with dextrogastria and a review of recent literature.


   Case History Top


Y.T. presented at 22 h of life with bilious vomiting, associated with progressive abdominal distension. He was at 33 weeks gestation. Mother was 36 years of age and para 5+0 (3 alive). There was history of polyhydramnios during pregnancy. There was no family history of congenital anomalies. During presentation, he was an active neonate, pink in room air, anicteric and afebrile. There was no facial dysmorphism. The fontanelles were patent and normotensive. He was tachypneic, but the lung fields were clear. Heart sounds were normal. Abdomen was grossly distended with visible bowel loops and hyperactive bowel sounds. There was no evidence of peritonitis. The anus was normally sited and patent. Other systems were essentially normal. Parents were pauper and could not afford required investigations and prescribed drugs.

Packed cell volume was 54% and electrolytes and urea were normal. Abdominal X-ray [Figure - 1] showed centrally placed dilated bowel loops with multiple air-fluid levels. The liver shadow was displaced to the left. Gastric fundus gas was absent in the left hypochondrium and there was no gas in the rectum. The radiologist had an impression of small intestinal obstruction due to atresia; dextrogastria was not observed and reported by them.

The infant was stabilized and the clinical status was optimized. At exploratory laparotomy [Figure - 2],[Figure - 3], the stomach was located on the right and grossly dilated up to the proximal jejunum (20 cm from the duodenojejunal junction). The right lobe of liver was in the left hypochondrium. Spleen was absent. There were multiple jejunal atresia (type IV) and reverse rotation of the midgut; the transverse colon was behind the duodenum and the root of the mesentery. Post-atretic part of the bowel was stenosed but patent. The atretic parts of the bowel and about 8 cm of the distal part of bulbous jejunum were resected. End-to-side jejunoileal anastomosis was performed. Incidental appendicectomy was also performed. The immediate postoperative period was uneventful. Patient passed greenish mucoid stool on the fifth postoperative day and normal stool from the sixth day. However, he developed a high-grade fever and deep cholestatic jaundice (conjugated bilirubin, 45% of total) on the seventh day. Fever was continuous and jaundice worsened; however, there was no evidence of peritonitis. The infant still passed stool 2-3 times daily. Blood culture yielded the heavy growth of Klebsiella spp., which was resistant to the cefotaxime hitherto used, but sensitive to ceftazidime. Despite the change in antibiotics, the condition of the infant deteriorated and he died on the tenth postoperative day. Post mortem showed clean peritoneal cavity with intact anastomotic site.


   Discussion Top


Situs anomalies are rare conditions [5] and can present with diagnostic and management challenges. [4] There have been reports on the overlapping spectrum of situs anomalies isolating organs or encompassing both the thoracic and abdominal viscera. [5] From our literature, the occurrence of dextrogastria, multiple jejunal atresia and inverse rotation of the intestine with levocardia in a neonate have not been reported in Africa, although there are reports of isolated intestinal malrotation in children of Nigeria. [6],[7] Budhiraja [8] reported the fourth case of isolated levocardia associated with SIA and malrotation with volvulus. Earlier reports have shown occurrence of isolated levocardia with SIA [9],[10],[11],[12] and another report has shown that dextrogastria can also occur in isolation. [13]

Embryologically, the mechanism responsible for the normal rotation of the loops of the heart and stomach to the left is not fully understood. The rotational direction of the stomach is owing to the forces exerted on it and the mesentery by the adjacent organs. [14] Genetic and familial theories have been adduced for the etiology; [5] however, mostly, isolated and accidental events are reported. The presence of multiple intestinal atresias could be explained by a vascular insult from the associated reverse rotation of the midgut in this patient.

SIA has a spectrum of presentation, which could be overlapping, and the intestinal obstruction is considered to be common. The occurrence of accompanying intestinal atresia that is observed in this patient is very rare [9] and has led to early presentation. To the uninitiated, situs anomalies present a diagnostic challenge and can be confusing for the experienced practitioner, particularly in the face of intestinal obstruction. [4] Plain and contrast enhanced X-rays and abdominal ultrasound can assist in preoperative diagnosis. Computerized tomography and radionuclear studies may enhance the diagnosis of the spectrum of anomalies where these facilities are available. The gross intestinal obstruction did not give room for a contrast GI series in this patient and dextrogastria was not suspected in this patient on the plain film.

Malrotation of the bowel is frequently observed in heterotaxy syndrome, which could be an asplenic or polysplenic syndrome. [15] An asplenic heterotaxy syndrome leads to an immune deficiency state and the newborn surgical patients are further susceptible to overwhelming nosocomial infection, as observed in this case.

In conclusion, although the combination of dextrogastria, intestinal atresia and malrotation are rare and poses a diagnostic challenge, the careful evaluation of patients may assist in facing this. Early presentation and appropriate surgical management may offer a chance of survival. Efforts should be made for proper aseptic procedures and nursing care of these patients. The early isolation of offending organisms and proper selection of appropriate antibiotics would reduce morbidity and prevent mortality.


   Acknowledgment Top


We are grateful to Drs. T. O. Odebode and G. A. Rahman who assisted in the review of the manuscript.

 
   References Top

1.Adeniran JO, Adekanye AO. Anorectal anomalies in children in Ilorin, Nigeria: A 10-yr review. Nigerian J Surg R 2003;5:148-51.  Back to cited text no. 1    
2.Chirdan LB, Uba AF, Pam SD. Intestinal atresia: Management problems in a developing country. Pediatr Surg Int 2004;20:834-7.  Back to cited text no. 2  [PUBMED]  [FULLTEXT]
3.Ameh EA, Chirdan LB. Neonatal intestinal obstruction in Zaria, Nigeria. East Afr Med J 2000;77:510-3.  Back to cited text no. 3  [PUBMED]  
4.Applegate KE, Goske MJ, Pierce G, Murphy D. Situs revisited: Imaging of the heterotaxy syndrome. http://radiographics.rsnajnls.org/cgi/content/abstract/19/4/837 cited September 2005  Back to cited text no. 4    
5.Gray SW, Skandalakis JE. Body assymetry and spelnic anomalies. In : Gray SW, Skandalakis JE, editors. Embryolorgy for surgeons. Saunders: Philadelphia PA; 1972. p. 877-95.  Back to cited text no. 5    
6.Adejuyigbe O, Bashorun AA, Ajayi AI, Adeoba EA. Experience with intestinal malrotation in Nigeria children. Nigerian Med J 1992;23:80-4.  Back to cited text no. 6    
7.Ameh EA, Nmadu PT. Intestinal Volvulus: Aetiology, morbidity and mortality in Nigeria children. Pediatr Surg Int 2000;16:50-2.  Back to cited text no. 7  [PUBMED]  [FULLTEXT]
8.Budhiraja S, Singh G, Miglani HP, Mitra SR. Neonatal intestinal obstruction with isolated levocardia. J Pediatr Surg 2000;35:1115-6.  Back to cited text no. 8    
9.Rattan KN, Pandit SK, Budhiraja S. Duodenal obstruction with isolated levocardia. Indian J Gastroenterol 1996;15:74.  Back to cited text no. 9  [PUBMED]  [FULLTEXT]
10.Ruben GD, Templeton JM Jr, Ziegler MM. Situs inversus: The complex inducing neonatal intestinal obstruction. J Pediatr Surg 1983;18:751-6.  Back to cited text no. 10  [PUBMED]  [FULLTEXT]
11.Kisham J, Bhargara RK, Kalani BP, Elzouki AY, Mir NA. Situs inversus abdominus with intestinal atresia. Indian Pediatr 1985;22:384-7.  Back to cited text no. 11    
12.Tryfonas GI, Chaidos C, Artzoglon PP, Zioutis J, Klokaris A, Papanastasopoulos A. Partial Situs inversus: Duodenal obstruction in a neonate with isolated levocardia. J Pediatr Surg 1992;27:1584-6.  Back to cited text no. 12    
13.Ardill W. Dextrogastria. J Am Coll Surg 2002;194:676.  Back to cited text no. 13  [PUBMED]  [FULLTEXT]
14.Nabot-Cegarra J, Maraculla-Sanz E, Reina-De la Toure F. Factors Involved in the Human embroyonic stomach around its longitudinal axis: Computer assisted morphometric analysis. J Anat 1999;194:61-9.  Back to cited text no. 14    
15.Ditchfield MR, Hutson JM. Intestinal rotational abnormalities in Polysplenia and asplenia syndromes. Pediatr Radiol 1998;28:303-6.  Back to cited text no. 15  [PUBMED]  [FULLTEXT]


    Figures

  [Figure - 1], [Figure - 2], [Figure - 3]


This article has been cited by
1 Splenic Torsion in a Child with Polysplenia and Situs Inversus: A Very Rare Presentation
Manas R. Dash,Anand V. Upasani,Sudhir B. Chandna,Pradip B. Rathod,Kirti K. Prajapati,Dhiren N. Patel
Indian Journal of Surgery. 2013; 75(S1): 236
[Pubmed] | [DOI]
2 How standard transesophageal echocardiography views change with dextrocardia
Raut, M.S. and Maheshwari, A. and Shad, S. and Rachna, G.
Annals of Cardiac Anaesthesia. 2013; 16(3): 218-220
[Pubmed]
3 Dextrocardia and situs inversus totalis in a nigerian cadaver: A case report of rare anomaly [Dextrocardia y situs inversus totalis en un cadáver nigeriano: Reporte de caso de una rara anomalía]
Ofusori, D.A. and Okwuonu, C.U. and Ude, R.A. and Adesanya, O.A.
International Journal of Morphology. 2009; 27(3): 837-840
[Pubmed]



 

Top
Print this article  Email this article
Previous article Next article

    

 
  Search
 
   Next article
   Previous article 
   Table of Contents
  
 
    Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
 Related articles
    Article in PDF (47 KB)
    Citation Manager
    Access Statistics
    Reader Comments
    Email Alert *
    Add to My List *
* Registration required (free)  


    Abstract
    Introduction
    Case History
    Discussion
    Acknowledgment
    References
    Article Figures

 Article Access Statistics
    Viewed4251    
    Printed98    
    Emailed1    
    PDF Downloaded175    
    Comments [Add]    
    Cited by others 3    

Recommend this journal


Contact us | Sitemap | Advertise | What's New | Copyright and Disclaimer 

  2005 - Journal of Indian Association of Pediatric Surgeons | Published by Wolters Kluwer - Medknow 

Online since 1st May '05