HNO16_Feb13.inddLicensee OA Publishing London 2013. Creative
Commons Attribution License (CC-BY)
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For citation purposes: Hu R, Jiang RS. The recurrence of a soft
palate teratoma in a neonate: a case report. Head Neck Oncol. 2013
Feb 06;5(2):16.
The recurrence of a soft palate teratoma in a neonate: a case
report R Hu1, RS Jiang1*
Abstract Teratomas are rare malformations containing cells from
ectodermal, mes- odermal and endodermal layers, seldom seen in the
soft palate. A 2-day-old girl presenting a teratoma arising from
the soft palate was surgically treated. Unfortunately, recurrence
was observed during follow-up.
Introduction Teratomas are rarely observed in the soft palate. A
teratoma, as defined by Weaver et al.1, is a tumour consisting of
multiple tissues that are not indig- enous to their site of origin.
The most important complication of oral tera- tomas is respiratory
compromise, which is the main cause of death in neonates2,3.
Surgical resection is the treatment of choice for oral terato- mas.
Although the follow-up is short in most cases, there are no reports
of recurrence of oral teratomas4,5. A case of a soft palate
teratoma in a neonate is reported herein; recurrence was observed
in 4 years follow-up.
Case report A female infant was born to a 20-year- old mother by
vaginal delivery after 37+3 weeks gestation with birth weight of
3.05 kg. A congenital tumour was found in the mouth, which
prevented oral feeding. Oral clinical examination revealed an
asympto- matic mass measuring 3.0 × 4.0 × 2.5 cm, originating from
the soft palate near the midline (Figure 1). The mother did not
undergo a three-dimensional (3D)-ultrasonography examination
during her pregnancy. The prenatal and perinatal courses were
uncompli- cated, and the family history was neg- ative. The father
was 27 years old and in good health. The infant was admit- ted to
our department on the second day after birth because of irregular
respiratory cycle. Shortness of breath and lip cyanosis was
observed, espe- cially when the infant was being fed. Due to
repeated episodes of apnea, tra- cheal intubation ventilator
support was provided and the infant was transferred to a neonatal
intensive care unit. Com- puted tomography showed an oral cav- ity
mass without intracranial extension. On the seventh day after
birth, under general anaesthesia with intubation, the mass was
excised from the soft pal- ate finding a short peduncle 1 cm in
diameter and the mucosal defect was repaired by suturing a
transferred local palatal flap (Figure 2). No cerebrospi- nal fluid
leakage occurred from the excision site. After surgery, the wound
healed well and the baby tolerated oral feeds. No respiratory
compromise occurred any more. Histopathological examination
revealed a mature tera- toma composed of mature respiratory
epithelium, glandular tissues, neuro- glial tissue, choroid plexus,
muscle and blood vessels (Figure 3). Some margins showed nerve and
muscle. Signs of recurrence were found in the second year of
follow-up. Magnetic resonance imaging (MRI) indicated maxillary
tumour with cystic and solid areas with fat density (Figure 4). The
parents wanted to “wait and watch”, while hoping for a better
prognosis, instead of opting for imme- diate reoperation.
Discussion Teratomas are rare malformations containing cells from
ectodermal,
mesodermal and endodermal layers with a variable degree of
differentia- tion6. They have an incidence of 1:4000 live births;
less than 2% of these arise from the oropharyngeal cavity1,7.
Nasopharynx is one of the most fre- quent sites for head and neck
terato- mas and has a 6:1 female predominance. In contrast, oral
teratomas do not present a clear gender predilection8. Oral
teratomas arise anywhere in the oronasal cavity and are regarded as
expanding, cavity-filling lesions, espe- cially in the neonatal
period9.
The main therapy of teratomas is complete surgical excision, which
depends on the site of the tumour. Unless the teratoma is expanding
massively into the cranial area, resection of tumour may be
attempted. Initial treatment should be directed toward airway
manage- ment and feeding problems10. When a neonate is experiencing
respira- tory difficulty, the first priority should be
stabilization of the airway11. Our case presented with an
obstructive mass causing respira- tory embarrassment and immediate
threat to life, which demanded establishment of an airway with
tracheostomy.
Histologically, teratomas may pre- sent different characteristics.
In oral teratomas, the most common tissues observed are nerves and
cartilages. Other tissues commonly seen are mus- cles, bones and
respiratory epithelia9. Our case showed mature respiratory
epithelia, glandular tissues, neuroglial tissues, choroid plexuses,
muscles and blood vessels.
Teratomas are associated with concomitant malformations in 6% of
all cases, with cleft palate being the most commonly associated
anomaly5. In the present case, congenital cardiac septal
* Corresponding author Email:
[email protected] 1 Department of
Reconstructive Plastic Surgery,
the Children’s Hospital of Zhejiang University School of Medicine,
Hangzhou, Zhejiang Province, China
Page 2 of 2
Licensee OA Publishing London 2013. Creative Commons Attribution
License (CC-BY)
Co m
pe tin
g in
te re
st s:
n on
e de
cl ar
ed . C
on fli
ct o
ed ic
al E
th ic
s (A
M E)
e th
ic al
ru le
s of
d is
cl os
ur e.
For citation purposes: Hu R, Jiang RS. The recurrence of a soft
palate teratoma in a neonate: a case report. Head Neck Oncol. 2013
Feb 06;5(2):16.
defects and patency of ductusarteriosus were detected.
Richieri-Costa et al.12 have also reported the observation of
cardiac abnormalities.
Teratomas are mostly benign in the neonatal period 5%; of the cases
present malignity criteria on histopa- thology13. Usually, benign
teratomas consist of mature tissue components, while those with
malignant potential contain immature tissues; there is a higher
incidence of malignancy in adults. Incomplete resection and
presence of primitive neural tissue entail the risk of a malignant
relapse14,15. Becker et al.5 reported the recurrence of a
congenital epignathus post-operation; this did not necessar- ily
imply malignancy, although the clinician should continue follow-up
screening.
It has been 3 years since the recur- rence of oral teratoma; the
girl is still in good condition without any drugs or surgery.
Because the risk of
malignant change is evident, long- term follow-up is
mandatory.
Acknowledgements We thank the patient and her parents for
participation. We also thank Mr Grahay Rester for critical reading
of the manuscript.
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Figure 1: Presence of a congenital tumour in the mouth
preoperatively.
Figure 4: MRI indicated maxillary tumour (recurrence).
Figure 2: Mucosal defect in the soft palate after the teratoma was
removed.