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 Table of Contents  
CASE REPORT
Year : 2020  |  Volume : 10  |  Issue : 2  |  Page : 63-65

Pyloroduodenal duplication cyst presenting like hypertrophic pyloric stenosis


1 Division of Paediatric Surgery, Department of Surgery, Ahmadu Bello University and Ahmadu Bello University Teaching Hospital, Zaria, Kaduna, Nigeria
2 Department of Surgery, Ahmadu Bello University Teaching Hospital, Zaria, Kaduna, Nigeria
3 Department of Pathology, Ahmadu Bello University and Ahmadu Bello University Teaching Hospital, Zaria, Kaduna, Nigeria

Date of Submission06-Mar-2020
Date of Acceptance16-Jun-2020
Date of Web Publication20-May-2021

Correspondence Address:
Dr. Tunde T Sholadoye
Division of Paediatric Surgery, Department of Surgery, Ahmadu Bello University and Ahmadu Bello University Teaching Hospital, Zaria, Kaduna
Nigeria
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ais.ais_11_20

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  Abstract 


Gastrointestinal duplication cyst is a rare congenital anomaly. When it involves the pylorus or the first part of the duodenum, it may present with features of gastric outlet obstruction. We present a 4-month-old baby girl, second of a set of twins who presented with features of gastric outlet obstruction. A diagnosis of gastric outlet obstruction querying hypertrophic pyloric stenosis was made and she was resuscitated. The intraoperative finding was that of a huge non-communicating duplication cyst involving the pylorus and the first part of the duodenum. Excision and mucosal stripping of the cyst was done. Histology showed ectopic gastric mucosa. Pyloroduodenal duplication cyst is a rare congenital anomaly of the gastrointestinal tract and a close differential for patients who present too early or too late with features of Hypertrophic pyloric stenosis.

Keywords: Duplication cyst, gastric outlet obstruction, pyloroduodenal cyst


How to cite this article:
Sholadoye TT, Abdulkarim SU, Aliyu HO, Ogunsua OO. Pyloroduodenal duplication cyst presenting like hypertrophic pyloric stenosis. Arch Int Surg 2020;10:63-5

How to cite this URL:
Sholadoye TT, Abdulkarim SU, Aliyu HO, Ogunsua OO. Pyloroduodenal duplication cyst presenting like hypertrophic pyloric stenosis. Arch Int Surg [serial online] 2020 [cited 2021 Jun 16];10:63-5. Available from: https://www.archintsurg.org/text.asp?2020/10/2/63/316487




  Introduction Top


Alimentary tract duplications are relatively rare congenital anomalies found anywhere from the mouth to the anus, characterized by well-developed smooth muscle layer, an epithelial lining, and attachment to the gastrointestinal tract. These duplications may present with obstruction from mass effect, evidence of bleeding from gastrointestinal tract or be discovered incidentally.[1] Most commonly, duplications are classified as cystic (65%) or tubular (35%). Small intestine is the most frequently affected part, with gastric and duodenal involvement accounting for 6–8% of all forms of alimentary tract duplication. Clinical presentation depends on the type and site of the lesion. High index of suspicion coupled with imaging studies help in making the diagnosis. Early surgical intervention is central to a better prognosis.[2] We hereby present the management of a 4-month-old girl with pyloroduodunal duplication cyst mimicking hypertrophic pyloric stenosis.


  Case Report Top


A 4-month-old baby girl, second of a set of twins, presented with projectile, non-bilous, post-prandial vomiting that started at in her third week of life. She was noticed to be growing slowly when compared to her twin sister. She was not jaundiced or did she pass blood in stool or have any other symptoms. She was a product of supervised, full-term gestation with birth weight of 2.9 kg. She was calm, pale with body weight of 3.7 kg compared to 5 kg body weight of her twin sister. Abdominal examination revealed visible peristaltic movement and an ovoid mass in the epigastrium to the right of the midline. No other masses were palpable and no other anomaly noticed.

A clinical diagnosis of gastric outlet obstruction secondary to hypertrophic pyloric stenosis to rule out duplication cyst was made. She was admitted and resuscitated then maintained on glucose containing intravenous fluid and broad-spectrum antibiotics. Admitting hematocrit of 26% was corrected by blood transfusion. Other hematological and biochemical parameters were normal. An abdominal sonography revealed a sub-hepatic huge complex cystic mass next to the left lobe of the liver, with well circumscribed thick-walled capsule (0.7 cm) measuring 6.0 × 4.2 × 6.1 cm (80.7 cm3), internal echoes and no color uptake on Doppler. A radiologic diagnosis of complex sub-hepatic cyst with compression of the gallbladder and duodenum was made. Upper GI contrast study was not done because it was not available. In view of the findings on ultrasound scan, a diagnosis of gastric outlet obstruction query duplication cyst to rule out subhepatic cyst was made and the child was explored.

Intra-operative finding was that of a cystic non-communicating pyloroduodenal duplication cyst measuring 12 × 10 × 8 cm along the greater curvature wrapped by the omentum and closely related to the transverse colon. It contained clear serous fluid and the cavity was lined by inflamed Mucosa [Figure 1]. Complete excision of the cyst wall was limited by the shared wall with the stomach and the first part of the duodenum. The free edge of the cyst was excised and the mucosa was completely denuded. No other structural anomaly was seen.
Figure 1: Intraoperative photograph of the cyst

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Histology revealed an ulcerated mucosa with foci of ectopic atrophic gastric mucosa, overlying a fibrotic submucosa, muscularis propria, and serosa [Figure 2]. There were lymphoid aggregates within the submucosa and oedematous changes in the muscularis propria. The cyst aspirate showed clusters of epithelial cells with reactive features. The features were consistent with a gastroduodenal duplication cyst with secondary chronic inflammation.
Figure 2: Histopathology: Pyloroduodenal cyst wall (H&E × 40) [L, lumen; US, ulcerated surface; G, gastric type glands; MP, Muscularis propria. Inset; epithelial cluster from cyst aspirate (Giemsa × 400)]

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She recovered promptly after surgery. Oral intake commenced on the 2nd day after surgery and she was discharged home on the 4th post-operative day. At clinic visit, she weighed 5.2 kg at 6 months and has been well after 2-year follow up.


  Discussion Top


Duplication cysts of the alimentary tract occurs most commonly in the ileum while duplication cysts occurring at the pyloric region are quite rare, and are predominantly seen in white females.[3] Pyloroduodenal duplication cyst is the rarest known so far and in most cases, it is cystic and non-communicating[4] as in the present case but in a black female.

The patient presented with projectile non-bilous post-prandial vomiting and an abdominal mass mimicking Infantile Hypertrophic Pyloric Stenosis (IHPS).[5] This situation was similar to the finding in a study, where a patient had pyloroduodenal duplication cyst and presented with symptoms of gastric outlet obstruction and an abdominal mass.[4] The distinction from an IHPS may be challenging without the aid of imaging (abdominal ultrasound and gastrointestinal contrast studies) prior to exploration.[6]

In this present case, abdominal ultrasound done suggested a cystic lesion around the left lobe of the liver, probably subhepatic cyst. Alimentary tract duplications around this region can mimic other lesions such as mesenteric cyst, pseudopancreatic cyst and choledochal cyst.[4] Therefore, higher radiological imaging such as abdominal computerized tomography scan and endoscopy may be required to make accurate diagnosis of pyloroduodenal cyst.[7] The patient had no associated anomaly; however, an earlier study reported associated hypertrophic pyloric stenosis coexisting with pyloric duplication cyst.[8]

Surgical approaches to pyloroduodenal cyst range from complete excision of the cyst with pyloroduodenal enterectomy and end to end pyloroduodenal anastomosis to partial cyst wall excision with mucosal stripping.[4],[6],[7] The later was the surgical procedure done on our patient, leaving behind the cyst wall that was adherent to the normal bowel. Some authors performed partial excision of the cyst wall and mucosal stripping[4] while others did partial cyst wall excision with cauterization of the remnant epithelial lining,[6] both methods have been associated with excellent outcomes.[4],[7] It appears that when the cyst is diagnosed later in life with associated inflamed mucosa, this technique (partial cyst wall excision with mucosal stripping) is preferred. In recent years with improved skills in laparoscopy, some patients with duplicated cyst are managed laparoscopically with comparable results.[5]

Histology findings in pyloroduodenal duplication cysts reported in the literature range from no notable lesions[9],[10] to ectopic pancreatic tissue,[7] heterotopic rests of gastric mucosa,[11] to adenocarcinoma.[12] The histology finding in our patient was of gastric mucosal rests with ulceration. Lopez-Fernandez and colleagues theorized that the gastric mucosal activity gives rise to the frequent mucosal ulceration seen in such cases as was noted in our patient, contributing to the clinical presentation.[11] As in most reports, the patient did well after surgery and remained so after 2 years of follow up.


  Conclusion Top


Pyloroduodenal duplication cyst is a rare alimentary tract duplication that may clinically mimic IHPS and in most regions with limited resources, diagnosis is made intraoperatively. However, high index of suspicion and careful radiologic imaging will give a better yield of preoperative diagnosis. Partial excision with mucosal stripping or cautery may suffice as an effective surgical treatment.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
George WH, Patrick JM, Daniel JO. Ashcraft's Paediatric Surgery: Alimentary Tract Duplications. 6th ed.. Elsevier Saunders, London; 2014. p. 539.  Back to cited text no. 1
    
2.
Taiwo OC, Faturoti OI, Taiwo AO, Adefalujo AP, Ajani MA. Gastric duplication cyst in a Nigerian infant. J Case Rep 2018;8:42-5.  Back to cited text no. 2
    
3.
Sharma D, Bharany RP, Mapshekhar RV. Duplication cyst of pyloric canal: A rare cause of pediatric gastric outlet obstruction: Rare case report. Indian J Surg 2013;75:322-5.  Back to cited text no. 3
    
4.
Mirza B. Pyloroduodenal duplication cyst: The rarest alimentary tract duplication. APSP J Case Rep 2012;3:19.  Back to cited text no. 4
    
5.
Delaplain PT, Gholizadeh M, Reyna T. Minimally invasive approach to non-communicating pyloric duplication. J Pediatr Surg Case Rep 2018;37:19-23.  Back to cited text no. 5
    
6.
Upadhyaya VD, Srivastava PK, Jaiman R, Gangopadhyay AN, Gupta DK, Sharma SP. Duplication cyst of pyloroduodenal canal: A rare cause of neonatal gastric outlet obstruction: A case report. Cases J 2009;2:42.  Back to cited text no. 6
    
7.
Annigeri VM, Hegde HV, Patil PB, Halgeri AB, Rao PR. Pyloroduodenal duplication cyst. J Indian Assoc Pediatr Surg 2012;17:80-1.  Back to cited text no. 7
  [Full text]  
8.
Mitra A, Khanna K, Krishna M, Srinivas M. Double jeopardy- hypertrophic pyloric stenosis and pyloroduodenal duplication cyst in a neonate. EC Gastroenterol Dig Syst 2016;1:125-8.  Back to cited text no. 8
    
9.
Anderson K, Rialon K, Pappas T, Perez A. Evolution of surgical resection for duodenal duplication cyst. CRSLS e2016;108. doi: 10.4293/CRSLS.2016.000108.  Back to cited text no. 9
    
10.
Sharma S, Yadav AK, Mandal AK, Zaheer S, Yadav DK, Samie A. Enteric duplication cysts in children: A clinicopathological dilemma. J Clin Diagn Res 2015;9:EC08.  Back to cited text no. 10
    
11.
Lopez-Fernandez S, Hernandez-Martin S, Ramírez M, Ortiz R, Martinez L, Tovar JA. Pyloroduodenal duplication cysts: Treatment of 11 cases. Eur J Pediatr Surg 2013;23:312-6.  Back to cited text no. 11
    
12.
Falk GL, Young CJ, Parer J. Adenocarcinoma arising in a duodenal duplication cyst a case report. Aust N Z J Surg 1991;61:551-3.  Back to cited text no. 12
    


    Figures

  [Figure 1], [Figure 2]



 

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