Indian Journal of Pathology and Microbiology
Home About us Instructions Submission Subscribe Advertise Contact e-Alerts Ahead Of Print Login 
Users Online: 1463
Print this page  Email this page Bookmark this page Small font sizeDefault font sizeIncrease font size

CASE REPORT Table of Contents  
Ahead of print publication
Mature teratoma of liver in an adult male patient


1 Associate Consultant, HBP Section, Specialized Surgery center, King Abdullah Medical City, Makkah, Saudi Arabia
2 Consultant and Head, Anatomic Pathology, HBP Section, Specialized Surgery center, King Abdullah Medical City, Makkah, Saudi Arabia
3 Associate Consultant, Hepatobiliary surgery, HBP Section, Specialized Surgery center, King Abdullah Medical City, Makkah, Saudi Arabia
4 Assistant Professor, Department of Pathology, Faculty of Applied Medical Sciences, Umm al Qura University, Makkah, Saudi Arabia

Click here for correspondence address and email

Date of Submission02-May-2021
Date of Decision28-Dec-2021
Date of Acceptance30-Dec-2021
Date of Web Publication07-Jun-2022
 

   Abstract 


Teratomas are germ cell tumors occurring usually in gonadal organs. They are neoplasms composed of one or more of the three germ layers. Extragonadal teratomas are uncommonly reported in the literature. The liver is an extremely rare site for teratoma, constituting < 1% of all teratomas. The majority of the liver teratomas are found in the pediatric population with only a dozen cases reported in adults to the best of our knowledge. We present a case of a 27-year-old male with a history of abdominal pain of 5 months duration. CT scan revealed liver mass suggesting teratoma. The patient underwent cholecystectomy and segmental liver resection. Histopathology revealed mature cystic teratoma. Complete resection remains the best treatment option. Teratomas of the liver are extremely rare, constituting < 1% of all teratomas. An extensive review of the literature yielded < 50 cases of primary hepatic teratomas with only a dozen cases in adults and only two cases in males.

Keywords: Germ cell tumor, liver, teratoma


How to cite this URL:
Saba F, Hamid T, M Alrawashdeh HY, Iqbal MS, Tabassum A. Mature teratoma of liver in an adult male patient. Indian J Pathol Microbiol [Epub ahead of print] [cited 2022 Nov 27]. Available from: https://www.ijpmonline.org/preprintarticle.asp?id=346846





   Introduction Top


The word teratoma is derived from the Greek word “teratos” which means a monster.[1] Teratomas are germ cell tumors composed of multiple cells derived from one or more of the three germ layers.[2] Teratomas occur commonly in ovaries and testis.[3] Teratomas occur in decreasing order of frequency in ovaries, testis, anterior mediastinum, retroperitoneum, sacrococcygeal region, and cranium. Mesenteric and gastrointestinal tract involvement is rare with very few cases reported in adults. Teratomas of the liver account for < 1% of all teratomas.[4] An extensive review of literature till 2019 yielded < 50 cases of primary hepatic teratomas with only a dozen of cases in adults and two cases in males.[3–5] This rarity of occurrence of teratoma in the adult population, especially males, and its relatively rare occurrence in the left lobe of the liver necessitates this case to be presented.

Case details

A 27-year-old male presented with a history of pain in the right upper quadrant of the abdomen for 5 months. The pain was dull aching and radiating to the back. There was no history of vomiting, jaundice, or gastrointestinal bleeding. On examination, the abdomen was soft with no tenderness. Laboratory investigations revealed leucocytosis. Other hematological tests were unremarkable. Liver function tests were normal. Alpha fetoprotein (AFP) levels were insignificant, and Echinococcal indirect hemagglutination assay (EIHA) was negative.

Radiological findings

A Trans axial post intravenous (IV) contrast computed tomography (CT) scan of the abdomen and pelvis was performed utilizing routine triphasic liver protocol with sagittal and coronal reformats. An encapsulated extrahepatic/sub-hepatic lesion measuring approximately 5.9 × 8.4 × 6.3 cm containing predominantly macroscopic fatty component with internal septations, heterogeneous soft tissue, and calcifications was present [Figure 1]a and [Figure 1]b. A radiological diagnosis of benign mature cystic teratoma (dermoid cyst) was made. There was no convincing aggressive infiltration of the surrounding soft tissue structures and hepatic parenchyma. Liver demonstrated homogenous parenchymal enhancement with no worrisome focal hepatic lesions. The portal and hepatic veins were patent, with the rest of the visualized major abdominal vascular structures within the normal limits. The gall bladder, pancreas, spleen, bilateral adrenals, and kidneys were unremarkable. There were no enlarged mesenteric, retroperitoneal, pelvic, or inguinal lymph nodes by CT size criteria. The visualized small and large bowel loops were grossly unremarkable, and no abdominal/pelvic free fluid was demonstrated. No aggressive osseous lesions were demonstrated within the visualized skeleton.
Figure 1: Computed tomography image showing encapsulated extrahepatic/subhepatic lesion. (arrow)

Click here to view


Pathological findings

Segmental resection of the liver was performed along with cholecystectomy. The resected lesion was a cut opened cyst measuring 7 × 5 × 5 cm in the left lobe of the liver. The external surface was grey tan. The focal liver tissue measured 4 × 2 × 1 cm. The cut section of the cystic lesion showed shafts of hair with a grey, yellow solid area [Figure 2].
Figure 2: Gross and Microscopic pictures show (a) cyst with shafts of hair with focal grey, yellow solid areas. (b) Skin adnexal structures including the sebaceous gland and smooth muscle. (H&E 10 ×) (c) Colonic mucosa. (H&E 20 × (d) Choroid Plexus tissue. (H&E 10 ×) (e) Fat and Skeletal muscle. (H&E 20 ×) (f) Fat and Skeletal muscle. (H&E 20 ×)

Click here to view


Microscopy from the gall bladder revealed features of chronic cholecystitis. The sections from the cystic lesion in the liver showed mature derivatives of germ layers. It showed skin with adnexal structure, respiratory mucosa, gastrointestinal mucosa, muscle, fat, bone, choroid plexus, and seromucinous glands. Focal foreign body giant cell reaction was also noted. [Figure 2]. No immature elements were seen. A pathological diagnosis of mature cystic teratoma was done. As primary teratoma in the liver is rare, clinical and radiological correlation was suggested to rule out other primary origin.

The chest radiograph was completely normal. Contrast-enhanced CT scan of the chest was performed. Lungs, heart, and mediastinum were unremarkable. There was no pleural effusion and no significant intrathoracic lymph nodes present. The thyroid gland was unremarkable. There were no suspicious lytic or sclerotic lesions in the chest wall and bones. There was no specific evidence of metastatic disease to the chest. An ultrasound examination of the scrotum showed both testes of normal size, site, shape, and homogenous echotexture. No scrotal lesion was noted [Figure 3].
Figure 3: (a) Chest X ray (PA) view, normal study (b and d) CT chest, unremarkable (c) USG Scrotum: Normal findings

Click here to view



   Discussion Top


Teratomas are congenital neoplasms characterized by an abnormal growth of a combination of tissues derived from ectoderm, mesoderm, and endodermal germ layers.[5] Teratomas can present at any age. They contain tissue elements that are foreign to the organ or anatomic site.[6] Embryologically, they are thought to arise from primordial germ cells that arrest along their migration path from allantois hindgut to the gonads during the first week of life. They commonly arise in the midline and paramedian locations which is also explained by Willis who proposed that teratomas arise from foci of totipotent cells in the area of primitive streak that somehow escape the influence of primary organizer during embryonic development.[7] Teratomas rarely occur in the liver. They are common in the pediatric age group but although uncommon in the liver comprising < 1% of all liver neoplasms. For reasons unexplained, their incidence is morein female patients and most common in the right hepatic lobe. There is no characteristic clinical presentation, and most cases are detected incidentally.[8] [Table 1] summarizes the clinicopathological and follow-up data of the reported cases of liver teratoma in adults in the literature.
Table 1: Clinicopathological data and follow-up status of liver teratoma cases in adults

Click here to view
[15]

The literature describes four histological variants of teratomas: Mature teratoma, immature teratoma, teratoma with malignant transformation, and monodermal teratoma.[9] The presence of immature tissue in any of their germ layer influences an unfavorable prognosis, which is based on histopathological features categorized as benign and malignant.[4] Mature teratomas are characteristically cystic and most of them are benign, although some can undergo malignant transformation.[9] Malignant transformation commonly involves squamous cell carcinoma (80%), adenocarcinoma (6.8%), sarcoma, and carcinoid tumors may also occur.[4] A previous report described hepatic cystic teratoma in a child harboring a hepatoblastoma.[10]

Mature cystic teratoma grows slowly at a rate of 1.8 mm each year, prompting some to advocate nonsurgical management of smaller less than 6 cm tumors. However, histopathological examination provides a definitive diagnosis, and surgical resection is considered the mainstay of treatment because the presence of immature tissue will affect the prognosis of the patient adversely.[9] The key differential diagnosis includes amebic abscess, Echinococcal cyst, and necrotic metastasis.[11]

Excellent prognosis has been reported after complete resection and with no additional therapy.[6] UK children's cancer study group found that even in incomplete resection of mature teratomas, 5-year event-free survival (EFS) was 92%, and overall survival (OS) rate was 99%.[6] In our case, 6 months follow-up showed no active complaints, normal tumor markers, and CT scan findings were unremarkable.


   Conclusion Top


We have reported a case of hepatic teratoma in an adult male. Only a dozen cases in adults and only two cases in males have been reported. Complete resection forms the mainstay of treatment.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Ramkumar J, Best A, Gurung A, Dufresne AM, Melich G, Vikis E, et al. Resection of ruptured hepatic teratoma in an adult. Int J Surg Case Rep 2018;53:414-9.  Back to cited text no. 1
    
2.
Aghajanzadeh M, Hemmati H, Delshad MSE, Rimaz S, Rad OM, Jafaryparvar Z, et al. Simultaneous multiple organs immature teratoma: Case report and review of literature. Oxf Med Case Rep 2018;4:134-7.  Back to cited text no. 2
    
3.
Gupta R, Bansal K, Manchanda V, Gupta R. Mature teratoma of liver. APSP J Case Rep 2013;4:13.  Back to cited text no. 3
    
4.
Jaklitsch M, Sobral M, de Figueiredo AAFP, Martins A, Marques HP. Rare giant mature cystic teratoma liver. J Surg Case Rep 2019;12:1-3.  Back to cited text no. 4
    
5.
Rahmat K, Vijayananthan A, Abdullah BJJ, Amin SM. Benign teratoma of liver: A rare case of cholangitis. Biomed Imaging Interv J 2006;2:e20.  Back to cited text no. 5
    
6.
O'Donovan EJ, Thway K, Moskovic EC. Extragonadal teratomas of adult abdomen and pelvis: A pictorial review. Br J Radiol 2014;87:20140116.  Back to cited text no. 6
    
7.
Martin LC, Papadatos D, Michaud C, Thomas J. Best cases of AFIP- liver teratoma. Radiographics 2004;24:1467-71.  Back to cited text no. 7
    
8.
Madan M, Arora R, Singh J, Kaur A. Mature teratoma of liver in adult female. Indian J Pathol Microbiol 2010;53:872-3.  Back to cited text no. 8
[PUBMED]  [Full text]  
9.
Harris B, Simone N. Primary mature cystic teratoma of liver: Report of a rare case. Am J Clin Med 2011;8:1.  Back to cited text no. 9
    
10.
Krainev AA, Mathavan VK, Klink D, Fuentes RC, Birhiray R Resection of a mature cystic teratoma of liver harboring carcinoid tumor. J Surg Case Rep 2018;10:1-4.  Back to cited text no. 10
    
11.
Karlo C, Leschka S, Dettmer M, Breitenstein S, Stolzmann P. Hepatic teratoma and peritoneal gliomatosis: A case report. Cases J 2009;2:9302.  Back to cited text no. 11
    
12.
Cöl C. Immature teratoma in both mediastinum and liver of a 21-Year-old female patient. Acta Med Austriaca 2003;30:26-8.  Back to cited text no. 12
    
13.
Silva DS, Dominguez M, Silvestre F, Calhim I, Daniel J, Teixeira M, et al. Liver teratoma in an adult. Eur Surg 2007;39:372–5.  Back to cited text no. 13
    
14.
Malek-Hosseini SA, Baezzat SR, Shamsaie A, Geramizadeh B, Salahi R, Salahi H, et al. Huge immature teratoma of the liver in an adult: A case report and review of the literature. Clin J Gastroenterol 2010;3:332-6.  Back to cited text no. 14
    
15.
Nirmala V, Chopra P, Machado NO. An unusual adult hepatic teratoma. Histopathology 2003;43:306-8.  Back to cited text no. 15
    

Top
Correspondence Address:
Mohammad Shahid Iqbal,
Department of Pathology, Faculty of Applied Medical Sciences, Umm Al Qura University, Makkah
Saudi Arabia
Login to access the Email id

Source of Support: None, Conflict of Interest: None

DOI: 10.4103/ijpm.ijpm_430_21



    Figures

  [Figure 1], [Figure 2], [Figure 3]
 
 
    Tables

  [Table 1]



 

Top
 
  Search
 
  
  
 Article in PDF
     Search Pubmed for
 
    -  Saba F
    -  Hamid T
    -  M Alrawashdeh HY
    -  Iqbal MS
    -  Tabassum A


    Abstract
   Introduction
   Discussion
   Conclusion
    References
    Article Figures
    Article Tables

 Article Access Statistics
    Viewed294    
    PDF Downloaded22    

Recommend this journal