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 ::  Introduction
 ::  Case report
 ::  Discussion
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CASE REPORT
Year : 1997  |  Volume : 43  |  Issue : 2  |  Page : 43-5

Duplication cyst of the pylorus--a rare cause of gastric outlet obstruction in the newborn.


Bai Jerbal Wadia Hospital for Children, A.D. Marg, Parel, Mumbai, India., India

Correspondence Address:
M P Patel
Bai Jerbal Wadia Hospital for Children, A.D. Marg, Parel, Mumbai, India.
India
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Source of Support: None, Conflict of Interest: None


PMID: 0010740718

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 :: Abstract 

Duplication cysts of the pylorus are the rarest of alimentary tract duplications with very few reported cases. We present such a cyst in a neonate presenting with gastric outlet obstruction. We have also reviewed the literature and outlined the theories of origin, modes of presentation, diagnosis and the surgical procedures. Even though several modes of surgery have been described, it is best to individualize the surgical option in each case. The results depend on the nature and site of duplication, complications and the associated anomalies.


Keywords: Case Report, Cysts, congenital,radiography,surgery,Diagnosis, Differential, Gastric Outlet Obstruction, congenital,radiography,surgery,Human, Infant, Newborn, Male, Pylorus, abnormalities,


How to cite this article:
Patel M P, Meisheri I V, Waingankar V S, Ramesh S, Naregal A M, Muthaal P B. Duplication cyst of the pylorus--a rare cause of gastric outlet obstruction in the newborn. J Postgrad Med 1997;43:43

How to cite this URL:
Patel M P, Meisheri I V, Waingankar V S, Ramesh S, Naregal A M, Muthaal P B. Duplication cyst of the pylorus--a rare cause of gastric outlet obstruction in the newborn. J Postgrad Med [serial online] 1997 [cited 2019 Nov 15];43:43. Available from: http://www.jpgmonline.com/text.asp?1997/43/2/43/410





  ::   Introduction Top


Gastroduodenal duplications are the rarest of alimentary tract duplications, pyloric duplications are still rarer. We came across a rare case of pyloric duplication presenting as gastric outlet obstruction in a newborn child. We have also discussed the pathology, clinical presentation and the surgical options for these anomalies.


  ::   Case report Top


A 3 day old full term male infant was brought with a history of non-bilious vomiting following each feed. The parents also noticed an upper abdominal lump. The child was active, pink and crying well. There was mild dehydration and an incteric tinge with an epigastric lump, which was firm, non-tender freely mobile and separate from the liver. There was no other positive finding on systemic examination. The plain X-ray showed a distended stomach [Figure - 1]. The sonography revealed a cystic mass in the upper abdomen below the liver suggestive of ? choledochal cyst, ? duplication cyst.

After correcting the dehydration, the child was taken for surgery. On exploration, there was a cystic mass measuring 8x5 cm at the pyloric region almost encircling the pylorus and compressing the pylorus and the proximal duodenum [Figure - 2]. The cyst was excised along with the adjacent pylorus in what amounted to a limited pyloroantrectomy. The duodenum was anastomosed to the stomach in a Billroth I fashion.

The child had a good post-operative period and tolerated oral feeds after the peristalsis had resumed. The child was alright till the tenth post-operative day. Unfortunately, he developed pseudomonas meningitis in the next few days and succumbed after 5 more days despite vigorous antibiotic therapy.

On histopathological examination, the cyst wall was found to have smooth muscle coat, sub-mucosa and intact gastric epithelium without any evidence of peptic ulceration.


  ::   Discussion Top


Alimentary tract duplications are rare anomalies. Gastric duplication are even rarer with only a handful of cases being reported. All the case reports and reviews have collected cases over several years and even decades[1],[2],[3],[4],[5],[6],[7],[8],[9].

There have been several theories of embryologic origin like incomplete twinning, phylogenetic reversal, persistent embryonic diverticula, dysvacuolation and tracheo-bronchial foregut duplication theory and have been well reviewed by Lister[2]. The `Split notochord theory’[10],[11] is widely accepted and it explains the frequently associated vertebral anomalies[2].

The recommended criteria for diagnosis of duplication cyst include[12]:

1. Proximity to the alimentary tract.

2. Common blood supply

3. Outer smooth muscle coat which is usually common with the gastric muscle coat.

4. Alimentary epithelial lining (not necessarily of the adjacent gut).

The usual location of gastric duplication is along the greater curvature. More often than not, there is no communication with the stomach. The incidence is twice as common in females as in males[5],[12]. Associated anomalies can occur in upto 50% of cases and can include other alimentary tract duplications and vertebral defects[12].

Symptoms vary according to the location of the cyst. The usual presentation is with abdominal mass, vomiting, weight loss and failure to thrive[5],[8]. Cysts near the pylorus, as in our case, is known to present in the neonatal period with persistent non-bilious vomiting simulating hypertrophic pyloric stenosis[7],[13],[15]. In older children, it may present with abdominal pain, gastrointestinal bleeding and fever.[5] These complications are attributed to inadequate drainage of the pent up acid pepsin secretions leading to ulceration, haemorrhage and perforation[8]. The fever is said to be because of infection.

Although the usual age of presentation is early infancy, most are manifest by one year of age[5]. Rare cases have been reported in a 41 year old male and a 64 year old woman who had a malignancy in the duplicated portion[5].

Gastric duplication may in some way force a malunion of the pancreatic anlage, as is evident from the case reports of association with aberrant pancreas[17], communication with a pancreatic duct[18], erosion into a pseudocyst[19] and as a cause of relapsing acute pancreatitis[20].

Kremer[8] suggests that a gastric duplication cyst should strongly be suspected in a female child (especially less than one year of age) presenting with an abdominal mass pain, vomiting and anaemia and if associated with X-ray evidence of indentation along the greater curvature and depression of transverse colon.

Surgery is the undisputed line of management but the surgical option vary. It is important that the surgery be individualised to each case. The surgeries performed have varied from simple excision with or without a margin of the adjacent stomach to even gastrectomy.

The stomach has blood supply on both sides unlike the mesenteric blood supply of the intestines[22]. Hence instead of resection of the involved segment of the bowel, it is sufficient to excise the cyst alone with the stripping of the resiaul mucosal lining; the raw surface being patched with transverse colon (for large defects)[22] or omentum[14], or even leaving a sero-muscular gap, like following a Rammstedt’s pyloromyotomy[2]. There are reports of excision procedures like a limited pyloroantrectomy[16], like in our case, to even gastrectomies[8].

In all surgical procedures, it is essential to remove all of the cyst lining[23]. Drainage procedures are only rarely advisable like when extensive resection would otherwise be necessary because of huge size or close proximity to the common duct etc. Procedures like cystgastrostomy, cystenterostomy Roux en Y and a `window’ procedure, have all been done in various situations. Marsupialization has to be avoided[12].

Asymptomatic cysts should possibly be removed because of possible late complication[8].

The results depend on the type of duplication, the presence of complications and especially on the associated malformations[2].

However, in this modern era, with the sophisticated specialised care available, it should be possible to salvage most of these children.

 
 :: References Top

1. Abrami G, Dennison WM. Duplication of stomach. Surgery 1961; 49:794-801.  Back to cited text no. 1    
2.Lister J. Duplications of the alimentary tract. Lister J, Irwing M (eds): Neonatal Surgery. 3rd ed. Chap 33, England: Butterworths; 1990, pp 474-484.  Back to cited text no. 2    
3.Gross RE. The surgery of infancy and childhood, Philadelphia: W.B. Saunders; 1953.  Back to cited text no. 3    
4.Ladd WE, Gross RE. Surgical treatment of duplication of the alimentary tract. Surg Gynaec Obstet 1940; 70:295-307.  Back to cited text no. 4    
5.Pruksopon C, Donovan RT, Pinit A. Gastric duplication. J Pediatr Surg 1979; 14:83-85.  Back to cited text no. 5    
6.Hockling M, Young G. Duplications of the alimentary tract. Br J Surg 1981; 68:92-96.  Back to cited text no. 6    
7.Parker BC, Guthrie J, France NE, Attwell JD. Gastric duplications in infancy. J Pediatr Surg 1972; 7:294-298.  Back to cited text no. 7    
8.Kremer RM, Lepott RB, Izant RJ Jr. Duplication of the stomach. J Pediatr Surg 1970; 5:360-364.  Back to cited text no. 8    
9.Milsom J, Unger S, Alford BA, Bradley MR. Triplication of the oesophagus with a gastric duplication. Surgery 1985; 98:121-125.  Back to cited text no. 9    
10.Fallon M, Gorden ARG, Lendrum AC. Mediastinal cysts of foregut origin associated with vertebral abnormalities. Br J Surg 1954; 41:520-533.  Back to cited text no. 10    
11.Bentely JFR, Smith JR. Developmental posterior enteric remnants and spital malformation. Arch Dis Child 1960; 35:76-86.  Back to cited text no. 11    
12.Dudgeon DL. Lesions of the stomach in Ashcraft, Holder (Eds) Pediatric Surgery, 2nd ed. Chap. 1993; 24:295-297.  Back to cited text no. 12    
13.Anas P, Miller RS. Pyloric duplication masquerading as hypertropic pyloric stenosis. J Pediatr Surg 1971; 6:664.  Back to cited text no. 13    
14.Grosfield J, Boles T, Roiner C. Duplication of pylorus in the newborn : a rare cause of gastric outlet obtsruction. J Pediatr Surg 1970; 5:365-369.  Back to cited text no. 14    
15.Kammerer G. Duplication of the stomach resembling hypertrophic pyloric stenosis. J Am Med Ass 1969; 207:2101-2102.  Back to cited text no. 15    
16.Torma MJ. Of double stomachs. Arch Surg 1974; 109:555-557.  Back to cited text no. 16    
17.Ales Sendini P. Gastric duplication communicating with an aberrant pancreas. J Pediatr Surg 1991; 309-311.  Back to cited text no. 17    
18.Bearzi I. Gastric duplication connecting with a pancreatic duct. Ital J. Gastroenterol 1990; 22:78-81.  Back to cited text no. 18    
19.Chawla A. Erosion through the posterior gastric wall by a pancreatic pseudocyst secondary to gastric duplication. J Pediatr Gastroenterol Nutr 1990; 11:115-118.  Back to cited text no. 19    
20.Lavine JE, Harrison M, Heyman MB. Gastrointestinal duplication causing relapsing acute pancreatitis in children. Gastroenterol 1989; 97:1556-1558.  Back to cited text no. 20    
21.Schocat SJ, Strand RD, Fellows KE, Folkman J. Perforated gastric duplication with a pulmonary communication - a case report. Surgery 1971; 70:370-374.  Back to cited text no. 21    
22.White JJ, Morsan WW. Improved operative technique for gastric duplication. Surgery 1970; 67:522-526.  Back to cited text no. 22    
23.Ravitch M. Duplications of the gastrointestinal tract in Welch KJ et al. Pediatr Surg, 4th ed.. 1986; pp 914-915.  Back to cited text no. 23    
24.Spence RK, Schnaufer L, Manboubi S. Co-existent gastric duplication and accessory pancreas - clinical manifestations, embryogenesis and treatment. J Pediatr Surg 1986; 21:68-70.   Back to cited text no. 24    


    Figures

[Figure - 1], [Figure - 2]



 

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