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Year : 2010  |  Volume : 53  |  Issue : 4  |  Page : 868-870
Immature gastric teratoma in an infant: Report of a case and review of the literature


Department of Pathology, All India Institute of Medical Sciences, New Delhi, India

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Date of Web Publication27-Oct-2010
 

How to cite this article:
Sharma A, Arora R, Gupta R, Dinda AK. Immature gastric teratoma in an infant: Report of a case and review of the literature. Indian J Pathol Microbiol 2010;53:868-70

How to cite this URL:
Sharma A, Arora R, Gupta R, Dinda AK. Immature gastric teratoma in an infant: Report of a case and review of the literature. Indian J Pathol Microbiol [serial online] 2010 [cited 2020 Nov 24];53:868-70. Available from: https://www.ijpmonline.org/text.asp?2010/53/4/868/72018


Sir,

Extra-gonadal germ cell tumors, including teratomas, have been reported involving sacrococcygeal region, mediastinum, presacral region, intracranial region, and retroperitoneum. Gastric teratomas are extremely rare. Till date only about 100 cases of gastric teratomas are recorded, of which less than 15 cases of immature variant have been described. [1],[2]

We describe the case of a 5-month-old male infant who presented with gradually enlarging left upper abdominal swelling and passage of black-colored stools for 15 days. Abdominal examination revealed a 16 × 15 cm, firm-to-hard, freely mobile mass in the left hypochondrium and left lumbar regions. The mass had a smooth surface with irregular rounded margins. Laboratory investigations revealed anemia (hemoglobin 7.9 mg/dl). Renal function and liver function tests were within normal limits. Ultrasonography (USG) of the abdomen showed a 13 × 9 cm mass attached to the greater wall of stomach. The mass showed mixed echogenecity along with few necrotic and calcified areas [Figure 1]. Hepatic size and echotexture were normal. No free fluid in the peritoneal cavity was evident. A clinicoradiologic diagnosis of an intra-abdominal tumor was kept and the infant was taken up for surgery. Laparotomy revealed a large friable mass measuring 15 × 15 cm and arising from the posterior wall of the stomach with adhesions to surrounding structures. The tumor was mobilized, separated from the transverse colon and greater curvature of the stomach, and excised. Gastroplasty and reparative gastrostomy were performed.
Figure 1: Ultrasound images showing a large solid mass (arrows) with mixed echogenecity and focal cystic areas

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Pathologic Findings

We received a cystic tumor measuring 12 × 10 × 7 cm. On serial sectioning, cystic and solid areas with few glistening white cartilaginous foci were identified. Multiple sections from the tumor showed variable histologic patterns. Tissues derived from each of the three germ cell layers (ectoderm, endoderm, and mesoderm) were seen in the form of mature adipose tissue, cartilage, various epithelia (respiratory, gastrointestinal, squamous), mature glial tissue [Figure 2]a, and smooth muscle. In addition, papillary structures resembling choroid plexus [Figure 2]b, pigmented retinal epithelium, clusters of ganglion cells, and lymphoid tissue were also seen. Although majority of the tissues were well differentiated and mature in appearance, foci of immature neuroepithelial elements, at places forming rosette-like structures [Figure 2]c, d along with foci of immature cartilage were also noted. This immature neural tissue was seen in more than three high-power fields (×40 magnification), corresponding to a grade 3 immature teratoma according to the Norris grading system. Extensive sampling did not reveal any focus of malignant transformation.
Figure 2: Photomicrographs showing a mixture of cartilage (c), columnar epithelium (e) and glial (g) tissue (a, H and E, ×400). Focal papillary structures resembling choroid plexus of the central nervous system are noted (b, H and E, ×100). Immature neuroepithelium forming rosettes (c, H and E, ×100) and demonstrating high mitotic activity (d, H and E, ×200) is seen

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Teratomas are neoplasms composed of elements representing all three germ layers and are subdivided into mature or immature subtypes depending upon constituent elements. The most common sites of occurrence of teratomas in infancy and childhood are sacrococcygeal (60-65% of cases), gonadal (10-20%), mediastinal (5-10%), and presacral (5%) regions. Other locations like kidney, liver, thyroid, and orbit are rarely involved. [2]

Gastric teratomas are extremely rare neoplasms, accounting for less than 1% of all teratomas occurring in infancy and childhood. Since the first report of gastric teratoma by Eusterman and Sentry [1] in 1922, about 100 cases have been recorded in the available literature. Majority of the gastric teratomas have occurred in infants or neonates with a striking male preponderance. [1] Gastric teratomas present as a palpable mass and/or abdominal distension. Tumors with intramural extension into the stomach cause gastrointestinal bleeding and rarely, gastric perforation. [1] Our patient presented with a palpable abdominal lump and evidence of gastrointestinal bleeding in the form of malena. Radiologic evaluation, especially USG and computed tomography (CT) scan, shows the heterogeneous nature of mass along with the presence of fat and calcification, suggesting a teratoma. [1] On clinicoradiologic features, various differential diagnoses to be considered include other pediatric abdominal masses, namely, neuroblastoma, Wilms' tumor, pancreatic cyst, hepatoblastoma, rhabdomyosarcoma, liposarcoma, and retroperitoneal teratoma. [3]

Of the reported gastric teratomas, most have been of mature histologic subtype. [4] Benign gastric teratomas are composed of mature elements like tooth, bone, cartilage, adipose tissue and well-differentiated epithelial lining. Immature teratoma, on the other hand, contains immature components from one of the germ cell layers, most commonly neuroepithelial elements. Immature teratomas have been subdivided into three grades based on the proportion of the immature components. [5] To the best of our knowledge, only about a dozen cases of gastric teratomas with immature elements or malignant features have been described in the literature, as summarized in [Table 1]. [1],[2] In our patient, histologic examination showed features of immature teratoma, grade 3, using this grading system. Although immature teratomas are often labeled as malignant and treated as such, little data are available to ascertain the true biologic behavior of these neoplasms. Complete surgical excision and regular close follow-up are mandatory in these patients. [4] Teratomas occurring in infants and children should always be examined with an extravigilant watch for the presence of neuroepithelium and other immature elements, which have an important bearing on the prognosis.
Table 1: Summary of previously reported cases of immature gastric teratoma

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In conclusion, immature gastric teratoma is an extremely rare tumor. An accurate diagnosis of this tumor and differential diagnosis from other commoner intra-abdominal neoplasms is essential in spite of its rarity. The detection of immature component carries a prognostic implication and hence, extensive sampling is essential.

 
   References Top

1.Bhat NA, Chishti AS, Moghazy K, Saeed S, Bisher H. Gastric immature teratoma. Saudi Med J 2007;28:959-60.   Back to cited text no. 1
[PUBMED]    
2.Utsch B, Fleischhack G, Knöpfle G, Hasan C, Bode U. Immature gastric teratomas of the lesser curvature in a male infant. J Pediatr Gastroenterol Nutr 2001;32:204-6.  Back to cited text no. 2
    
3.Dunlap JP, James CA, Maxson RT, Bell JM, Wagner CW. Gastric teratoma with intramural extension. Pediatr Radiol 1995;25:383-4.  Back to cited text no. 3
[PUBMED]    
4.Gupta DK, Srinivas M, Dave S, Agarwala S, Bajpai M, Mitra DK. Gastric teratoma in children. Pediatr Surg Int 2000;16:329-32.  Back to cited text no. 4
[PUBMED]  [FULLTEXT]  
5.Norris HJ, Zirkin HJ, Benson WL. Immature (malignant) teratoma of ovary: A clinical and pathologic study of 58 cases. Cancer 1976;37:2359-72.  Back to cited text no. 5
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Correspondence Address:
Amit Kumar Dinda
Department of Pathology, All India Institute of Medical Science, Ansari Nagar, New Delhi - 110029
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0377-4929.72018

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    Figures

  [Figure 1], [Figure 2]
 
 
    Tables

  [Table 1]

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