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Journal of Indian Association of Pediatric Surgeons
     Journal of Indian Association of Pediatric Surgeons
Official journal of the Indian Association of Pediatric Surgeons         
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Table of Contents   
CASE REPORT
Year : 2018  |  Volume : 23  |  Issue : 4  |  Page : 228-231
 

Teratoma in the hepatoduodenal ligament


Department of Pediatric Surgery and Radiodiagnosis, All India Institute of Medical Sciences, New Delhi, India

Date of Web Publication4-Oct-2018

Correspondence Address:
Dr. Anjan Kumar Dhua
Room No. 4002, Department of Pediatric Surgery, Teaching Block, All India Institute of Medical Sciences, Ansari Nagar, New Delhi - 110 029
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jiaps.JIAPS_36_18

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   Abstract 


A teratoma is not an uncommon tumor in the pediatric age group. It has a predilection for specific sites, but a teratoma arising from the hepatoduodenal ligament (HDL) is very rare. Only 15 cases of HDL teratoma have been reported so far. Due to the proximity to important structures, it is imperative that the correct anatomical localization is done preoperatively to avoid intraoperative surprises. Herein, we report a case of HDL teratoma in a toddler who underwent a successful excision. The relevant literature is also discussed briefly from a pediatric surgeon's perspective.


Keywords: Hepatoduodenal ligament, teratoma, yolk sac tumor


How to cite this article:
Gundapaneni S, Dhua AK, Jain V, Jana M, Agarwala S, Bhatnagar V. Teratoma in the hepatoduodenal ligament. J Indian Assoc Pediatr Surg 2018;23:228-31

How to cite this URL:
Gundapaneni S, Dhua AK, Jain V, Jana M, Agarwala S, Bhatnagar V. Teratoma in the hepatoduodenal ligament. J Indian Assoc Pediatr Surg [serial online] 2018 [cited 2018 Dec 11];23:228-31. Available from: http://www.jiaps.com/text.asp?2018/23/4/228/242716





   Introduction Top


Teratomas are tumors containing normal tissue of various types derived from more than one germ layer. The cell of origin is derived from the germline which migrates to the midline, and hence, most of them are located in the brain, sacrococcygeal area, mediastinum, retroperitoneum, and gonads. Embryologically, the hepatoduodenal ligament (HDL) is also an erstwhile midline structure that lies at the free edge of the lesser omentum bridging the liver and lesser curvature of the stomach. Despite this anatomical predisposition, teratomas derived from this midline structure are very rare. We herein report a case of a teratoma derived from the HDL and discuss the importance of this diagnosis from a pediatric surgical perspective in light of the available literature.[1],[2],[3],[4],[5],[6],[7],[8],[9],[10],[11],[12],[13],[14]


   Case Report Top


A 2-year-old girl presented with incidentally detected abdominal lump since one month of age. The lump had been gradually increasing in size and was not associated with any other complaints. On physical examination, there was a mobile, firm, globular lump of approximately 10 cm × 8 cm in the upper abdomen. Ultrasound scan (US) of the abdomen revealed a mobile, solid mass with cysts of variable size arising from the gastrohepatic ligament. A contrast-enhanced CT scan of the abdomen also showed that the mass was originating from the gastrohepatic ligament with features of fat attenuation and calcification along with cystic areas suggestive of a mature teratoma [Figure 1]. The serum alpha-fetoprotein (AFP) level was normal. The child underwent laparotomy, and intraoperatively, a 10 cm × 8 cm × 7 cm mass with both solid and cystic components was found to arise from the hepatoduodenal ligament [Figure 2]. There was no infiltration into any surrounding structures. The tumor was excised without any damage to the structures of the HDL. Histopathological examination (HPE) confirmed the diagnosis of mature teratoma. The postoperative course was uneventful. At 6 months of follow-up, the patient has remained asymptomatic and has been advised for further clinical follow-up along with serum AFP and US of the abdomen.
Figure 1: (a) Noncontrast computed tomography abdomen axial section reveals a mass (M) about the left lobe of the liver (L) and gallbladder (GB); and anterior to the pancreas (P). The mass had a significant fat component (asterisk), cystic component, and coarse calcification (arrow). The stomach (St) is displaced to the left and posteriorly. (b) Contrast-enhanced computed tomography abdomen coronal reformatted image reveals a mass (M) to be inferomedial to the liver. After contrast administration, there is no enhancement of the mass lesion. Arrow points toward the calcification

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Figure 2: Schematic representation of the intraoperative findings. The asterisk (*) is showing the right posterolateral surface of the mass in close relation to the hepatoduodenal ligament and arising from it. All other surfaces of the mass were free from the surrounding structures (the left lobe of liver and the stomach). The posterior surface of the mass was in close contact with the gastrohepatic ligament but was completely free

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   Discussion Top


Teratomas are usually not difficult to diagnose considering the typical sites they are located in and the unique presence of a mixture of tissues derived from all the three germ layers with its characteristic radiological finding. Typically, they present with a mass effect and on evaluation they can be found to be composed of fat, bone, or cartilage in variable proportions, which can be picked by appropriate imaging. Based on the presence of malignant cells, there might be an associated rise in tumor marker for the corresponding neoplastic lineage.

A teratoma arising from the retroperitoneum was initially thought of in our case; however, the mobility in retroperitoneal teratoma is restricted, unlike the freely mobile mass in our case. The possibility of a teratoma arising from the stomach was considered, but typically it affects males, and hence, a gastric teratoma was ruled out. The diagnosis of teratoma in our case was not difficult; however, the site of origin was contentious. Due to its rarity, an HDL teratoma was never thought of as a possibility. In our case, the radiological examinations predicted the site of origin of the teratoma as the gastrohepatic ligament; however, its exact relationship with the HDL was an intraoperative surprise. Our report is only the 16th case of teratoma originating from the HDL in the English literature [Table 1],[1],[2],[3],[4],[5],[6],[7],[8],[9],[10],[11],[12],[13],[14] and diagnostic difficulty has been reported in a few of these cases. Brown et al. in 2008 reported the two instances of HDL teratoma that were initially diagnosed as choledochal cysts, but later diagnosis of HDL teratoma was confirmed on HPE.[3]
Table 1: Characteristics of the hepatoduodenal ligament teratoma reports previously described in the literature

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The management of the abdominal teratoma is always surgical but knowing its anatomical relation with a very critical structure like the HDL is also crucial. If the involvement of the HDL is known preoperatively, it alerts the surgeon to be careful while dissecting the area and also have back up plans, should the structures in HDL get damaged inadvertently. Portal vein injury and their subsequent repair have been described in two cases earlier.[8],[11] In a recent report by Ravikumar et al.,[14] complete transection of the portal vein, hepatic artery, and common bile duct was described in a neonate. Ukiyama et al. reported the use of intraoperative USG and Doppler to prevent damage to the vascular structures, especially the hepatic artery, in their case.[8] In previously reported cases, various reconstructive techniques such as choledochojejunostomy (in two cases),[2],[7] hepaticoduodenostomy (in one case),[11] Whipple procedure (in one case)[4] have been described. Demircan et al. have described a cystojejunostomy and ductocystostomy after partial excision of a distal portion of the common hepatic duct to maintain bilio-enteric continuity.[5] In our case, we were able to complete the procedure without any collateral damage, and the rest of the anatomy was also normal, and hence the need for reconstructive procedure did not arise. Histopathologically, the majority of the reported cases (11 of 15) were mature teratomas similar to our case.[2],[4],[5],[6],[7],[8],[9],[10],[11],[13] Three cases were immature teratomas[1],[12],[14] while in another case, the histopathology finding was not reported.[3] Yolk sac elements were found in two cases, one in relation to a teratoma with immature elements,[1] and the other one associated with a mature teratoma.[4] The need for chemotherapy is dependent on the histological findings. Three of these reported cases had received chemotherapy; this was because one of them had a recurrence,[2] and in another case, the histological diagnosis had revealed an endodermal sinus tumor associated with a benign cystic teratoma.[4] The third case had received three courses of chemotherapy after an incisional biopsy had revealed immature teratoma with yolk sac elements before the definitive extirpation of the mass.[1] Our case had a normal serum AFP value, and the histopathology report was indicative of mature teratoma. Hence, no chemotherapy was required. However, close surveillance on the serial AFP values with clinical and radiological assessment for early detection of recurrences and malignancy if any has been planned.

The present case re-emphasises that an HDL teratoma has to be kept in mind as a differential diagnosis of a midline, mobile upper abdominal solid-cystic mass despite their rarity. In addition, for a pediatric surgeon, it is important to know the anatomical relationship of the tumor preoperatively, as the surgical procedure that may be required could range from a simple excision to a complicated reconstructive procedure.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
De Roo AC, Siddiqui S, Mychaliska GB. Hepatoduodenal ligament teratoma with immature elements. Pediatr Surg Int 2017;33:1023-6.  Back to cited text no. 1
    
2.
Frexes M, Neblett WW 3rd, Holcomb GW Jr. Spectrum of biliary disease in childhood. South Med J 1986;79:1342-9.  Back to cited text no. 2
    
3.
Brown B, Khalil B, Batra G, Rampersad B, Bruce J. Mature cystic teratoma arising at the porta hepatis: A diagnostic dilemma. J Pediatr Surg 2008;43:e1-3.  Back to cited text no. 3
    
4.
Akimov OV. Hepatoduodenal ligament teratoma followed by hypertensive syndrome of the portal vein. Arkh Patol 1989;51:60-2.  Back to cited text no. 4
    
5.
Kim WS, Choi BI, Lee YS, Chi JG, Park HR, Kim I, et al. Endodermal sinus tumour associated with benign teratoma of the common bile duct. Pediatr Radiol 1993;23:59-60.  Back to cited text no. 5
    
6.
Wang H, Dong J. Teratoma in the hepatoduodenal ligament followed by portal hypertension syndrome. J Gastroenterol Hepatol 2004;19:477-9.  Back to cited text no. 6
    
7.
Demircan M, Uguralp S, Mutus M, Kutlu R, Mizrak B. Teratoma arising from anomalous common bile ducts: A case report. J Pediatr Surg 2004;39:e1-2.  Back to cited text no. 7
    
8.
Sasaki H, Ajiki T, Takase S, Fujino Y, Suzuki Y, Tominaga M, et al. Images of interest. Hepatobiliary and pancreatic: Mature cystic teratoma in the hepatoduodenal ligament. J Gastroenterol Hepatol 2005;20:317.  Back to cited text no. 8
    
9.
Souftas V, Polychronidis A, Giatromanolaki A, Perente S, Simopoulos C. Dermoid cyst in the hepatoduodenal ligament: Report of a case. Surg Today 2008;38:959-61.  Back to cited text no. 9
    
10.
Ukiyama E, Endo M, Yoshida F. Hepatoduodenal ligament teratoma with hepatic artery running inside. Pediatr Surg Int 2008;24:1239-42.  Back to cited text no. 10
    
11.
Bagga D, Jindal B, Naredi BK, Yadav DK, Acharya SK, Mahato R, et al. Portal teratoma causing obstructive jaundice in children: A rarity. J Pediatr Surg 2012;47:1449-52.  Back to cited text no. 11
    
12.
Ohno M, Tanaka H, Watanabe T, Sato K, Takahashi M, Yamada K. Giant infantile immature teratoma derived from the hepatoduodenal ligament: Report of a case. J Pediatr Surg Case Rep 2013;1:301-3.  Back to cited text no. 12
    
13.
Jeismann VB, Dumarco RB, Loreto Cd, Barbuti RC, Jukemura J. Rare cause of abdominal incidentaloma: Hepatoduodenal ligament teratoma. World J Gastrointest Surg 2014;6:80-3.  Back to cited text no. 13
    
14.
Ravikumar VR, Rajamani G, Raju V, Sundar R, Ravikumar S, Maniam R. Teratoma arising from hepato duodenal ligament in the newborn with transection of portal vein, hepatic artery and common bile duct: A surgical challenge. J Indian Assoc Pediatr Surg 2018;23:45-7.  Back to cited text no. 14
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