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Omphalomesenteric duct cyst in an omphalocele: a rare association.
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PMID:  24353646     Owner:  NLM     Status:  PubMed-not-MEDLINE    
Abstract/OtherAbstract:
Omphalomesenteric duct (OMD) remnants and omphalocele are not infrequently seen in paediatric patients. In most of the cases, OMD remnant in an omphalocele is a Meckel's diverticulum; however rarely there may be other lesions. A one-day old male baby underwent surgery for omphalocele. At exploration a 10 x 12 cm cyst containing gut contents was found as the content of the omphalocele, with proximal and distal ileal loops running in continuity with it. Resection of the cyst with end to end primary gut anastomosis was done. Baby also had complex associated cardiac anomalies and died few days after surgery due to sepsis.
Authors:
Yousuf Aziz Khan; Mumtaz Ahmed Qureshi; Jamshed Akhtar
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Publication Detail:
Type:  Journal Article    
Journal Detail:
Title:  Pakistan journal of medical sciences     Volume:  29     ISSN:  1682-024X     ISO Abbreviation:  Pak J Med Sci     Publication Date:  2013 May 
Date Detail:
Created Date:  2013-12-19     Completed Date:  2013-12-19     Revised Date:  2013-12-23    
Medline Journal Info:
Nlm Unique ID:  100913117     Medline TA:  Pak J Med Sci     Country:  Pakistan    
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Languages:  eng     Pagination:  866-8     Citation Subset:  -    
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Journal Information
Journal ID (nlm-ta): Pak J Med Sci
Journal ID (iso-abbrev): Pak J Med Sci
Journal ID (publisher-id): PJMS
ISSN: 1682-024X
ISSN: 1681-715X
Publisher: Professional Medical Publicaitons, Karachi, Pakistan
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Received Day: 1 Month: 3 Year: 2013
Revision Received Day: 16 Month: 5 Year: 2013
Accepted Day: 18 Month: 5 Year: 2013
Print publication date: Season: May-Jun Year: 2013
Volume: 29 Issue: 3
First Page: 866 Last Page: 868
PubMed Id: 24353646
ID: 3809310
Publisher Id: pjms-29-866

Omphalomesenteric Duct Cyst in an Omphalocele: A Rare Association
Yousuf Aziz Khan1
Mumtaz Ahmed Qureshi2
Jamshed Akhtar3
1Yousuf Aziz Khan, MBBS, FCPS (Paediatric Surgeon), Department of Paediatric Surgery, National Institute of Child Health, Rafiquee Shaheed Road, Karachi – 75510, Sind, Pakistan.
2Mumtaz Ahmed Qureshi, MBBS, FCPS (Paediatric Surgeon), Department of Paediatric Surgery, National Institute of Child Health, Rafiquee Shaheed Road, Karachi – 75510, Sind, Pakistan.
3Jamshed Akhtar, MBBS, FCPS (Paediatric. Surgeon). Department of Paediatric Surgery, National Institute of Child Health, Rafiquee Shaheed Road, Karachi – 75510, Sind, Pakistan.
Correspondence: Correspondence: Yousuf Aziz Khan, MBBS, FCPS (Paed. Surg.), Department of Paediatric Surgery, National Institute of Child Health, Rafiquee Shaheed Road, Karachi – 75510, Sind, Pakistan. E-mail: dr_yousufaziz@yahoo.com

INTRODUCTION

Omphalomesenteric duct is a normal embryological structure that connects the fetal midgut with the yolk sac. It usually regresses by 9th week of gestation. However if that does not happen, a spectrum of anomalies may result.1 Remnants of OMD may be present in 2% of the population with Meckel’s diverticulum (MD) as the commonest of remnants. OMD remnants may form part of an omphalocele however, omphalocele containing an OMD cyst is extremely rare and very few cases have been reported.2,3 Some of these cysts are serous, probably occurring due to the sequestration of the OMD remnants during intrauterine life. Meconium or faecal cysts probably develop as a consequence of partial obstruction at the neck of the umbilical defect where gut contents can flow into the OMD and forms a large cyst. Herein we report a case of a newborn with an omphalocele in whom an omphalomesenteric cyst was found.


CASE REPORT

A one-day old, full term male, weighing 2.6 kg, was referred with a swelling at umbilical region. Baby was delivered at a local hospital through spontaneous vaginal delivery. On arrival, he had normal morphological features. There was a large omphalocele about 10 x 12 cm, covered with a thin, intact, semi-transparent membrane with a narrow pedicle of around 3 cm diameter (Fig.1). Umbilical clamp was well away from the omphalocele. The small bowel loops and a thick sac like structure were visible through the thin membrane. Rest of the neonatal examination was unremarkable.

Echocardiography showed a large ventricular septal defect with overriding of aorta, hypertrophied right ventricle, infundibular pulmonary stenosis and bunching of atrial septum – correlated with Tetralogy of Fallot. After optimization of the general condition surgery was done. At surgery, a 10 x 12 cm meconium filled cyst located at the distal ileum was found as the major content of the sac, along with small intestinal loops. The proximal and distal ileal loops were communicating with the cyst. (Fig. 2 and 3) Rest of the bowel had no gross anomaly. Excision of cyst with primary end-to-end ileo-ileal anastomosis was performed.

Post operative course was stormy. During his stay at PICU the baby developed uncontrolled septicaemia and died in spite of best of efforts. Histopathology of the excised specimen revealed wall of small intestine with thin walled blood vessels surrounded by fibromyxomatous tissue. Ectopic mucosa was not identified.


DISCUSSION

Omphalomesenteric duct cyst is infrequently reported.2,4 Vane et al found only three cases of OMD cyst in 217 children with OMD remnants.5It may be found attached to the umbilicus, bowel or both.4 Males have higher incidence than females (4:1) as with other OM anomalies, and are not associated to maternal age, gravidity, race or prematurity.4,6 It has varied presentations.4,7-9In newborns, OMD remnants may present in an omphalocele as reported by Chattopadhyay A et al and Nicol JW et al.3,10 Ratan SK et al reported a case similar to ours in a newborn but had a cyst containing translucent fluid rather than meconium.2

In the present case, a large cyst was seen in the omphalocele sac, outside peritoneal cavity. It was a localized dilatation in the area of Meckel’s diverticulum and was in communication at both the ends with the small intestine and contained meconium. The possibility of a variant of cystic duplication of the small bowel was ruled out as it was located on anti-mesenteric side of the bowel with no aberrant vascular supply and sharing of the common wall. So an impression of a variant of OMD remnant and possibly omphalomesenteric cyst was made.

OMD cysts have a variable diameter and appear as a cystically dilated segment of small bowel. They are usually lined by columnar epithelium resembling that of the small bowel, colon or stomach. Ectopic pancreatic tissue may be present.6 The gross and histological characteristics of the specimen in our case were similar to that reported in the literature supporting the possibility of omphalomesenteric cyst. However, no ectopic tissue was identified in our case. In conclusion OMD cyst may be present in a large omphalocele and the prognosis depends upon the associated anomalies.


References
1. Rasool N,Hussain I,Akhtar J,Ahmed S,Aziz A. Various presentations of omphalomesenteric duct remnants in children J Coll Physicians Surg PakYear: 2002122047
2. Ratan SK,Rattan KN,Kalra R,Maheshwari J,Parihar D,Ratan J. Omphalomesenteric duct cyst as a content of omphalocele Indian J PediatrYear: 2007745000217526966
3. Chattopadhyay A,Prakash B,Nagendhar Y,Kumar V. Patent omphalomesenteric duct and exomphalos minor: a rare and interesting association Int SurgYear: 200792254618399095
4. Sawada F,Yoshimura R,Ito K,Nakamura K,Nawata H,Mizumoto K,et al. Adult case of an omphalomesenteric cyst resected by laparoscopic-assisted surgery World J GastroenterolYear: 2006128252716521206
5. Vane DW,West KW,Grosfeld JL. Vitelline duct anomalies: experience with 217 childhood cases Arch SurgYear: 198712254273495250
6. Lowry AW,Han YS. Omphalomesenteric duct cystConsultant For PediatriciansYear: 201110321 [Internet] (Available at: http://www.pediatricsconsultant360.com/content/omphalomesenteric-duct-cyst.
7. Ballester I,Betlloch I,Pérez-Crespo M,Toledo F,Cuesta L. Atypical presentation of an omphalomesenteric duct cyst Dermatol Online JYear: 20091513
8. Aydogan F,Aytac E,Durak H. A rare cause of palpable mass located at the suprapubic area: Abscess of omphalomesenteric duct cyst Turk J GastroenterolYear: 201021195620872341
9. Tamilselvan K,Mohan A,Cheslyn-Curtis S,Eisenhut M. Persistent umbilical discharge from an omphalomesenteric duct cyst containing gastric mucosa Case Rep PediatrYear: 20122012482185
10. Nicol JW,MacKinlay GA. Meckel's diverticulum in exomphalos minor J R Coll Surg EdinbYear: 199439677515434

Figures

[Figure ID: F1]
Fig.1 

Large omphalocele containing OMD cyst



[Figure ID: F2]
Fig.2 

Per operative picture showing cystically dilated segment of bowel in the region of Meckel’s diverticulum, in continuity with proximal and distal ileal loops



[Figure ID: F3]
Fig.3 

The excised specimen of the cyst containing gut contents



Article Categories:
  • Case Report

Keywords: Key Words Omphalomesenteric duct remnants, Omphalocele, Omphalomesenteric duct cyst, Newborn.

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