|Year : 2021 | Volume
| Issue : 1 | Page : 48-51
Gastric teratoma in infant: A case report
K. M. Didarul Islam, Shoheli Alam, Silwal Shirish, Noor Mahammad, Umme Habiba Dilshad Munmun
Paediatric Surgery Department, Bangabandhu Sheikh Mujib Medical University, Dhaka, Bangladesh
|Date of Submission||14-Sep-2021|
|Date of Acceptance||23-Sep-2021|
|Date of Web Publication||29-Dec-2021|
Mr. K. M. Didarul Islam
Paediatric Surgery Department, Bangabandhu Sheikh Mujib Medical University, Dhaka, 1000
Source of Support: None, Conflict of Interest: None
Gastric teratomas (GTs) are very rare tumors in children. They usually present with a palpable mass in the upper abdomen. We report a case of GT in a 4-month-old male infant who was presented with a palpable central abdominal mass. Ultrasound and computed tomography (CT) scan revealed a huge mass with heterogenous consistency and scattered calcification within the mass, extending from epigastric to hypogastric region. His biochemical marker of alpha-fetoprotein (AFP) was raised (1277 ng/mL). The preoperative clinical diagnosis was suspected teratoma but not specifically gastric one. On laparotomy, the mass was found to be attached with the greater curvature of the stomach and it was excised completely with all layers of the attached part of the greater curvature of the stomach wall. Histopathology confirmed it was a grade-I immature GT. The rarity of the origin of teratoma in addition to its immature variety prompted us to report the case.
Keywords: Gastric, immature teratoma, infant
|How to cite this article:|
Islam KM, Alam S, Shirish S, Mahammad N, Munmun UH. Gastric teratoma in infant: A case report. Paediatr Nephrol J Bangladesh 2021;6:48-51
| Introduction|| |
Teratomas are the most common germ cell tumor and are composed of elements from one or more of the embryonic germ cell layers and contain tissue foreign to the anatomic site of origin. Nearly, 25% of germ cell tumors that occur during the first year of life are teratomas, whereas teratomas at older age account for only 2%–5%. Gastric teratoma (GT) is a rare variety that comprises less than 1% of all teratomas in children., Immature teratomas are pathologically distinct from benign and malignant teratomas with their embryonic-appearing neuroglial and neuroepithelial elements. Alpha-fetoprotein (AFP) and beta-human chorionic gonadotropin (beta-HCG) may not increase if there are no malignant germ cell components., We report a 4-month-old male infant with immature GT who had a complete surgical resection.
| Case Report|| |
A 4-month-old infant presented with supraumbilical swelling, noticed by the parents, for a month which is gradually increasing in size. There was no abnormal finding in general examination. The abdomen was distended with a palpable mass around 15 × 15 cm occupying mainly umbilical region, partly epigastric and hypogastric regions. All the laboratory parameters were within normal limits except AFP, which was 1277 ng/ml. Ultrasound abdomen showed a huge heterogeneous mass lesion having internal cystic changes and tiny calcifications extending from epigastric to hypogastric region. CT scan of the abdomen revealed a well-defined septated cystic area with heterogeneous densities following intravenous contrast and areas of scattered calcification occupying the whole abdomen filling such as the contour of the abdomen. The preoperative diagnosis was intra-abdominal teratoma [Figure 1].
|Figure 1: Contrast CT scan: a well-defined septated cystic area with heterogeneous densities and area of scattered calcification occupying the whole abdomen|
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The patient was explored through a supra-umbilical transverse incision. A huge mass arising from the greater curvature of stomach, extending up to the hypogastric region was found [Figure 2]. Complete excision was done along with some portion of all layers of the stomach wall. The gastric wall was repaired in two layers. Specimens were sent for histopathology [Figure 3]. The report shows a malignant neoplasm composed of mature and immature tissues. Immature components are neuroepithelial tissue, glandular structure, cartilage, and mesenchymal tissue. Immature neural component occupying less than one per low power field. Mature components are bone and adnexal structures. Areas of necrosis are seen. One section shows unremarkable gastric tissue. Diagnosis made as immature GT, Grade-I. Postoperative course was uneventful. At 1 month of follow-up, the AFP was decreased to 158.64 ng/mL. After completion of two cycles of chemotherapy in combination of cisplatin 2.5 mg/kg/day for 7 days and doxorubicin 0.25 mg/kg/day for 3 days in each cycle, AFP was within the normal range (4.3 ng/mL). During adjuvant chemotherapy, the patient developed mild gastrointestinal symptoms such as nausea, vomiting, and was managed with medical treatment.
|Figure 2: After laparotomy, the mass was found attached with the greater curvature of the stomach|
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| Discussion|| |
Teratomas are embryonal neoplasms that contain tissue from all three germ layers, namely, ectoderm, endoderm, and mesoderm and are derived from totipotent cells. The site of origin in one of the largest series of benign and malignant teratomas in children was sacrococcygeal in 65%, mediastinal in 10%, gonadal in 11%, and presacralin 4% of cases.
GT being a rare variety was first reported by Eusterman and Sentry in 1922. GT presents with more varied characteristics than other teratomas as it predominately occur in male 90%, whereas other teratomas usually occur in female. Most GT patients are infants and have predominantly exhibited benign behavior. Only a few cases of GTs with malignant components have been reported.,,
The site of origin of GT is variable and most of them arise from the greater curvature and posterior wall of the stomach. However, other sites such as lesser curvature have also been documented. Patients presented according to the site of origin, size, and according to the position of the mass, whether intra-gastric or exogastric. Exogastric growth is usually found in about (58–70%) and intragastric growth is around 30%. Clinical presentation depends on these factors with nausea, vomiting, abdominal distention, and mass. Patients with intragastric components present with hematemesis, pain abdomen, melena, vomiting, and rarely with gastric perforation.,
An abdominal radiograph can detect calcifications and bone densities, which are hallmarks of intra-abdominal teratomas which are mainly detected by the CT scan. When combined with intravenous and oral contrasts, they can detect the origin of the tumor, its relation with gastrointestinal tract and major blood vessels, presence of bones and calcifications, tumor extent and indicates the presence of metastasis.,,
Regular monitoring with AFP and beta-HCG reflect the treatment response after excision and may be of significant value where chemotherapy is recommended in immature variety. AFP synthesis occurs in the fetal liver, yolk sac, and gastrointestinal tract, whereas beta-HCG synthesis occurs most commonly in yolk sac and less in gastrointestinal tract. For simplification of our follow-up procedure, we relied on the monitoring of the value of AFP as it is the most reliable tumor marker for gastrointestinal teratoma. In our case, the preoperative AFP level was abruptly reduced by 10 times after a month of excision. And after completion of chemotherapy, AFP level was reduced to the normal range (4.3 ng/mL).
According to the classification of Gonzales Crussi, teratomas are classified into benign and malignant forms. Benign teratomas can be subdivided into mature (well-differentiated grade-0) and immature (potentially malignant) categories. Immature teratomas have occasional microscopic foci of incompletely differentiated tissue, mainly neural tissue. Depending on the ratio of incompletely differentiated components of the sampled surface, different grades of immaturity can be attributed as follows: grade-1 up to 10%, grade-2 10–50%, and grade-3 greater than 50%.
The recommended treatment of any GT is complete surgical excision (with tumor-free margins) and close surveillance. Many authors depicted no recurrence after complete excision of the immature GT even in grades 2 and 3, and there had not been any use of postoperative chemotherapy or radiotherapy; however, in a case where the AFP start rising after few months of complete excision of teratoma, chemotherapy is added.,,,, GT is very rare and immature variety is rarest. Because of its predominant benign behavior, its prognosis is excellent. Because of the presence of immature neuroepithelial elements, GT is labeled as malignant. Advanced surgical techniques and regular follow-up with AFP show a better result.
| Conclusion|| |
Most of the GTs are benign in nature. Total excision of the tumor along with partial or total gastrectomy seems to be enough treatment. Adjuvant therapy is recommended if histopathology shows abundance of immature cells in tumors.
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
Conflicts of interest
There are no conflicts of interest.
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[Figure 1], [Figure 2], [Figure 3]