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 Table of Contents  
CASE REPORT
Year : 2019  |  Volume : 8  |  Issue : 4  |  Page : 250-251

Oral teratoma presenting with bleeding and respiratory difficulty: A very rare case


Department of Pediatric Surgery, SMS Medical College, Jaipur, Rajasthan, India

Date of Submission23-Oct-2018
Date of Decision19-Jun-2019
Date of Acceptance08-Sep-2019
Date of Web Publication04-Oct-2019

Correspondence Address:
Dr. Aditya Pratap Singh
Near the Mali Hostel, Main Bali Road, Falna, Dist-Pali, Rajasthan
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jcn.JCN_107_18

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  Abstract 


An oropharyngeal teratoma is a rare malformation. It is also known as epignathus. It is composed of all three germinal layer derived cells. Only few cases are reported. We present a case of large teratoma that was arising from the tongue in a newborn. Total surgical excision was performed and histological examination showed mature teratoma.

Keywords: Newborn, oropharyngeal, teratoma


How to cite this article:
Garg D, Singh AP. Oral teratoma presenting with bleeding and respiratory difficulty: A very rare case. J Clin Neonatol 2019;8:250-1

How to cite this URL:
Garg D, Singh AP. Oral teratoma presenting with bleeding and respiratory difficulty: A very rare case. J Clin Neonatol [serial online] 2019 [cited 2019 Dec 8];8:250-1. Available from: http://www.jcnonweb.com/text.asp?2019/8/4/250/268581




  Introduction Top


Teratoma was defined by Weaver et al. as a tumor consisting of multiple tissues that are not indigenous to their site of origin.[1] It is a tumor composed of multiple tissues foreign to the normal organ from which it arises. Most common sites are the sacrococcygeal, anterior mediastinum, testicle, ovary, or retroperitoneum.[1] Teratoma of the head and neck is rare, and only 10% of teratoma find in this area. Nasopharynx and cervical region are the most common sites.[2] Epignathus describes as a tumor in the oropharyngeal region, with an incidence of one in 35,000–200,000 live births.[3]


  Case Report Top


Parent of a 2.3-kg term female newborn presented to us with the complaints of respiratory difficulty and large irregular oral mass with bleeding. The baby was delivered by cesarean section to a 23-year mother who had a regular antenatal examination. In antenatal ultrasound, teratoma was detected. After delivery, the newborn had respiratory difficulty with bleeding from a large irregular mass arising from the tongue, protruded anteriorly [Figure 1]a. The patient had chest indrawing and tachypnea. The patient was intubated immediately and was prepared for the surgery. Complete resection of tumor was performed [Figure 1]b. The patient was shifted to the neonatal intensive care unit on ventilator after the surgery. The patient was in shock. Postoperative recovery was not uneventful, and the baby expired after 1 day because of hypovolemic shock.
Figure 1: (a) Photo shows large oral teratoma (b) excised specimen

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  Discussion Top


Teratoma contains all three germ cell layers (ectoderm, mesoderm, and endoderm).[3] Histologically, they may be mature, immature, or malignant.[4] They may arise from different sites in the body, the most common is sacrococcygeal (accounts for approximately 40% of total cases).[5] Less than 5% occur in the head and neck.[5] Epignathus is a teratoma that arises from the oropharynx.[4] When large, they distort the facial anatomy and cause respiratory embarrassment at birth. Our case was noted to have oral mass causing respiratory difficulty.

Teratomas are more common in female.[2] When present during early childhood, they are usually benign.[2] As in most reported cases, our patient was female, and the tumor was benign.

Teratoma may be diagnosed antenatally on ultrasound or magnetic resonance, which permits early multidisciplinary management. The diagnosis was confirmed by polyhydramnios.[6] The differential diagnosis is limited but includes hamartoma, dermoid cyst, and heterotopic gastrointestinal cyst.[6] Teeth within the mass confirm teratoma. The fetus with teratoma is also at risk of high-output cardiac failure, placentomegaly, and hydrops with subsequent fetal demise secondary to arteriovenous shunting through the tumor or chronic fetal anemia due to hemorrhage within the tumor.

Resection is the treatment of choice, as there may be a small chance of malignant transformation, the longer they are left.[7] If antenatal diagnosis is made, two treatment procedures may be used: ex utero intrapartum treatment (EXIT) procedures and operation on placental support (OOPS).

In the EXIT procedure, after a low transverse uterine incision, the head and at least one hand of the fetus are delivered. In the OOPS procedure, first, the airway is secured by endotracheal intubation or tracheostomy; later, the baby is completely delivered and the umbilical cord is clamped. In our case, the patient was delivered outside from our center; so, EXIT procedure was not possible.

Kolekar et al. reported a case of oral teratoma in a newborn with respiratory difficulty.[8] The neonate's prognosis worsens as the size of tumor increases. Mortality in oral teratoma is high in the absence of well-prepared resuscitation team or meticulous delivery planning to secure the airway. Das et al. reported a case of nasopharyngeal teratoma in a newborn causing airway obstruction.[9] In our case, tumor detected antenatal and cesarean section was planned because of large size but due to handling during delivery bleeding started. The patient was intubated immediately and operated in an emergency department within 2 h after the delivery. Hence, it is important to handle these teratomas with care during delivery. Other causes of death in oral teratoma are respiratory tract obstruction and feeding difficulties. Causes of bleeding in teratoma are disseminated intravascular coagulation and thrombocytopenia.


  Conclusion Top


This report emphasizes on antenatal detection of teratoma and delivery by the EXIT procedure. Large oral teratoma should be handled gently.

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.

Acknowledgment

The authors would like to thank Dr. Maryem Ansari, Assistant Professor, MD in Pathology, SMS Medical College Jaipur, Rajasthan, India.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Weaver RG, Meyerhoff WL, Gates GA. Teratomas of the head and neck. Surg Forum 1976;27:539-44.  Back to cited text no. 1
    
2.
Kountakis SE, Minotti AM, Maillard A, Stiernberg CM. Teratomas of the head and neck. Am J Otolaryngol 1994;15:292-6.  Back to cited text no. 2
    
3.
Vandenhaute B, Leteurtre E, Lecomte-Houcke M, Pellerin P, Nuyts JP, Cuisset JM, et al. Epignathus teratoma: Report of three cases with a review of the literature. Cleft Palate Craniofac J 2000;37:83-91.  Back to cited text no. 3
    
4.
Lionel J, Valvoda M, Al-Abdul Hadi KA. Giant epignathus: A case report. Kuwait Med J 2004;36:217-20.  Back to cited text no. 4
    
5.
Okafor CL, Okafor CO, Odike MA. Congenital teratomaof the face. J Obstet Gynaecol 2004;24:828-9.  Back to cited text no. 5
    
6.
Takeuchi K, Masuda Y, Narita F, Kiyoshi K, Mizutori M, Maruo T. Prenatal evaluation of bidirectional epignathus: Comparison of ultrasonography and magnetic resonance imaging. Fetal Diagn Ther 2003;18:26-8.  Back to cited text no. 6
    
7.
Keswani RK, Chugh TD, Dhall JC, Mehrotra GC. Epignathus – A case report. Br J Plast Surg 1968;21:355-9.  Back to cited text no. 7
    
8.
Kolekar SS, Chincholi T, Nangare N, Patankar R. Oral teratoma. Int J Appl Basic Med Res 2016;6:54-6.  Back to cited text no. 8
    
9.
Das M, Mukherjee D, Gupta SD, Mukherjee S, Chatterjee U, Datta C, et al. Congenital nasopharyngeal teratoma causing airway obstruction in the newborn. J Cancer Res Ther 2015;11:1040.  Back to cited text no. 9
    


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