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Short communication

Foregut duplication cyst of the oor of mouth in a neonate:

case report
Jason Schrotenboer a , Amer Heider b,1 , Ethan Smith c,2 , Sharon Aronovich a,
a
b
c

Oral and Maxillofacial Surgery, University of Michigan, 1515 E Hospital Dr., SPC 5222 Towsley G1109, Ann Arbor, MI, 48109
Anatomic Pathology, University of Michigan, C.S Mott Childrens Hospital, Floor 11 1540 E Hospital Dr SPC 4300 Ann Arbor, MI
Pediatric Radiology, University of Michigan, C.S. Mott Childrens Hospital Floor 3 Recp A Room 3660A, 1540 E Hospital Dr SPC 4252 Ann Arbor, MI

Accepted 1 January 2016

Abstract
Congenital oral masses may interfere with vital functions such as respiration and deglutition in neonates. We report a congenital heterotopic
oral gastrointestinal cyst that caused failure to thrive. A working knowledge of the differential diagnosis and pathophysiology of this can
guide the clinician in its evaluation and management.
2016 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.

Keywords: Foregut duplication cyst; Gastrointestinal cyst; Floor of mouth lesion; Enterocystoma; Enteric duplication cyst; Choristoma

Case report
In August 2014, the University of Michigan Department of
Pediatric Oral and Maxillofacial Surgery was asked to evaluate a healthy 2-day-old baby boy with a swelling in the floor
of the mouth, difficulty in feeding, and weight loss.
On examination there was no evidence of obstruction
of the upper airway, and our chief concern was hindrance to oral feeding and failure to thrive. The lesion was
approximately 1 x 1 cm, tissue-coloured, soft, mobile, and
partly-compressible, with no pulsation (Fig. 1). The differential diagnosis included dermoid cyst, imperforate duct
orifice, accessory Whartons duct, foregut duplication cyst,
and cystic hygroma. Other neonatal lesions that may arise in
the floor of the mouth include a venolymphatic malforma-

tion, epidermoid cyst, teratoma, congenital haemangioma,

and thyroglossal duct cyst.14
We inserted a nasogastric tube for enteral feeding. The next
day a magnetic resonance scan (MRI) was done with intravenous gadolinium contrast under general anaesthesia, which
showed a well-circumscribed, bilobed lesion in the floor of
the mouth to the right of the midline. The lesion was homogeneously hyperintense on the T2-weighted images and there
was no appreciable enhancement contrast after the infusion
of gadolinium (Fig. 2). These findings were consistent with a
benign cystic lesion. There was no evidence of sialectasia as
would be expected with an imperforate Whartons duct. The
baby was kept anaesthetised, and was transferred to theatre
for excision of the lesion.
Surgical technique

Corresponding author. Tel.: +734 936 8289; fax: +734 232 6973.
E-mail address: saronovi@med.umich.edu (S. Aronovich).
1 Tel.: 734-232-8774; fax: +734 615 2964.
2 Tel.: 734 936 4500; fax: +734 647 5452.

An incision 1 cm long was made over the lesions highest curvature, and dissection with scissors extended to the lesion. A
white capsule was evident with notable fluctuance on palpation. During dissection around the capsule, white, cloudy,

http://dx.doi.org/10.1016/j.bjoms.2016.01.002
0266-4356/ 2016 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.

Please cite this article in press as: Schrotenboer J, et al. Foregut duplication cyst of the floor of mouth in a neonate: case report. Br J Oral
Maxillofac Surg (2015), http://dx.doi.org/10.1016/j.bjoms.2016.01.002

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Fig. 1. A partly-compressible, soft, mobile lesion in the right floor of mouth

about 1 cm x 1 cm seen on examination.

keratinaceous fluid was aspirated and cytological examination showed no malignant cells. Histological examination
of a frozen section of a biopsy specimen showed part of a
cyst lined with squamous epithelium. Sharp and blunt dissection was then continued until the lesion had been completely
removed. There were no complications and he was extubated
uneventfully. He progressed to oral feeding shortly after operation. Nine months later there was no evidence of recurrence.
Histological sections showed a cystic structure lined by
squamous and respiratory epithelium. In addition, there were
foci of gastric and intestinal mucosa (Fig. 3). A diagnosis of
heterotopic gastrointestinal oral cyst was made.

Fig. 2. Magnetic resonance image (MRI) axial T2-weighted fast spin echo
images showing a bilobed, homogeneously T2-weighted hyperintense lesion
in the floor of the mouth to the right of the midline(arrow). Note the second,
smaller lobule of the lesion more medially (arrowhead).

Fig. 3. Foci of the lining of the cyst is composed of intestinal-type mucosa

with goblet cells adjacent to squamous epithelium (H&E stain, original
magnification x 10).

Discussion
Heterotopic gastrointestinal oral cyst (enterocystoma or
enteric duplication cyst) is a choristoma that occurs almost
invariably in infancy. Its origin is uncertain, but is thought to
be a developmental cyst derived either from embryonic rests
from gastric anlage, a retention cyst of the salivary glands,
entrapped primitive endoderm, or a duplication cyst. It occurs
mainly in the anterior floor of the mouth and tongue. Additional sites in the head and neck include the submandibular
gland, anterior neck, and epiglottis.5
Imaging of lesions of the floor of the mouth relies
on cross-sectional imaging techniques, including computed
tomography (CT) and MRI. The use of CT in the oral cavity
may not be particularly helpful, because contrast resolution
is relatively low, and there is limited characterisation of tissue and associated ionising radiation. Although the long-term
risks of malignancy may be small and difficult to quantify,
this may be a reasonable concern in children because of the
radiosensitivity of their tissues and their long life expectancy.
6,7 MRI is ideally suited to imaging lesions in the head and
neck, and multiphase or dynamic post-contrast imaging can
aid characterisation of vascular lesion and other masses.
Eaton et al8 reviewed six infants with masses in the anterior tongue/floor of mouth noted after birth, which were
later confirmed histologically to be foregut duplication cysts.
While no patients had respiratory concerns, three had difficulty in feeding.8 Using prenatal imaging, Kong et al9
identified a lesion of the floor of mouth with possible compromise of the airway. An ex utero intrapartum treatment
(EXIT) procedure was accompanied by aspiration and biopsy,
which showed this to be a foregut duplication cyst. It recurred
promptly and was excised completely. 9
In the management of children with sublingual abnormalities, imaging followed by operation may be done under the
same anaesthetic. If necessary, an EXIT procedure may be
planned to secure the airway with intubation, tracheostomy,

Please cite this article in press as: Schrotenboer J, et al. Foregut duplication cyst of the floor of mouth in a neonate: case report. Br J Oral
Maxillofac Surg (2015), http://dx.doi.org/10.1016/j.bjoms.2016.01.002

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or extracorporeal membrane oxygenation.10 For fluid-filled

lesions needle-decompression may be a useful temporary
measure. Finally, complete excision with preservation of vital
structures should be the definitive treatment of a foregut
duplication cyst.
Conict of Interest
We have no conflict of interest.
Ethics statement/conrmation of patients permission
There is no identifiable information in the manuscript or
figures.
References

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Please cite this article in press as: Schrotenboer J, et al. Foregut duplication cyst of the floor of mouth in a neonate: case report. Br J Oral
Maxillofac Surg (2015), http://dx.doi.org/10.1016/j.bjoms.2016.01.002