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International Journal of Medical and Dental Case Reports (2014), Article ID 010914, 3 Pages

CASE REPORT

Ciliated cyst of the maxilla following trauma: An unusual


case report
Harsha Vardhan Gowthamnath, J. S. Jesija, K. Saraswathi Gopal
Oral Medicine and Radiology, Meenakshi Ammal Dental College, Chennai, Tamil Nadu, India

Correspondence
Dr.Harsha Vardhan Gowthamnath,
OralMedicine and Radiology, Meenakshi
Ammal Dental College, Chennai, Tamil Nadu,
India. Email: bgharshavardhan@yahoo.com
Received 14.04.2014;
Accepted 01.09.2014

Abstract
Surgical ciliated cysts or post-operative maxillary cysts are benign cystic lesions
usually induced after a surgical procedure in the maxillofacial area. It is a cystic lesion
that develops, following a radical sinus surgery to treat maxillary sinusitis. The cyst
progressively enlarges due to the osmotic dierence causing destruction of the adjacent
bone and structures. The clinical scenario may be identical to a radicular cyst if a nonvital
tooth is involved. This is a case report of a healthy 17-year-old adolescent with the
complaint of swelling in the right side of the face for the past 2 years.

doi: 10.15713/ins.ijmdcr.1
Keywords: Ciliated cyst, maxilla, radiolucent, trauma
How to cite the article:
Gowthamnath HV, Jesija JS , Gopal S.
Ciliated cyst of the maxilla following trauma:
Anunusual case report. Int J Med Dent Case
Rep 2014: 1-3.

line joining the corner of right lip to the ear lobe. Anteriorly,
the swelling was found to diuse with the ala of the nose and
posteriorly it extended 3 cm short of the ear lobe. The swelling
was hard and nontender to palpation [Figure 1a]. Intra-orally,
there was obliteration of the buccal vestibule in relation to 12
and 13 with no secondary changes [Figure 1b]. Vitality test
was performed from right first premolar to left canine and 11,
12, and 13 showed no response to the test and proved to be
nonvital. Based on the history and clinical examination, a clinical
diagnosis of benign odontogenic cyst or tumor was considered.
The patient was then subjected to radiological evaluation.
Orthopantamogram revealed a single large unilocular
radiolucency measuring approximately 3 cms in diameter in
the right maxillary region extending superiorly to the level of
maxillary antrum and inferiorly toward the alveolar process
in relation to 12, 13, 14, 15, and 16 which obscured the right
maxillary sinus. There was also evidence of apical displacement
of the tooth 13 [Figure 2].
Computed tomography (CT) maxilla revealed evidence of
a well-defined, moderate-sized expansive cystic lesion with thin
bony rim and hypo dense soft tissue component arising from
the alveolar margins of right maxilla projecting into the adjacent
right maxillary sinus almost completely occluding its lumen,
predominantly expanding in the labial aspect, suggestive of
odontogenic cyst [Figure 3].
Following the above mentioned investigations, fine-needle
aspiration cytology was performed by creating a surgical

Introduction
The surgical ciliated cyst has been well-known since 1927, and it
is documented in the Japanese literature.[1] The surgical ciliated
cyst is also known as post-operative maxillary cyst, post-operative
paranasal cyst, and surgical ciliated cyst of the maxilla.[2,3] Most
of these cysts have occurred as delayed complication of radical
maxillary sinus surgeries to treat various sinus pathologies.[3,4] This
is a case report of a ciliated cyst of the maxilla that had developed
in a young adolescent boy following minor traumatic injury.
Case Report
A 17-year-old male presented to the Department of Oral
Medicine and Radiology with the complaint of swelling in the
right side of the face for the past 2 years. The patient presented
with a history of trauma 2 years ago, when he was hit by a rod
while travelling in a bus. Following the injury, he developed a
swelling in the right side of his face. There were no secondary
changes during this 2-year period. The patient had developed
pain in the upper right anterior teeth region 2 months ago. He
had consulted a private dentist, following which a root canal
therapy was initiated, but left incomplete, with no pain thereafter.
On clinical examination, the patient presented with a
diuse swelling in the right side mid face region approximately
measuring 3 cm 3 cm, extending superiorly one cm below the
infra-orbital margins and inferiorly 0.5 cm above an imaginary
1

Ciliated cyst of the maxilla

Gowthamnath, et al.

Figure 1: (a) Extra oral photograph and (b) intra oral photograph

Figure 3: Computed tomography images (a) coronal view and


(b)axial view

Figure 2: Orthopantamogram
a

window. It yielded a straw colored aspirate which showed


numerous polygonally shaped, anucleated and nucleated
epithelial cells along with inflammatory cells, predominantly
lymphocytes, few eosinophils, plasma cells, and macrophages
in a background of red blood cells suggestive of infected cystic
material.
Subsequently, incisional biopsy was performed, and
histopathology revealed cystic lining epithelium of varying
thickness supported by fibro vascular connective tissue
showing chronic inflammatory cell infiltrate predominantly of
lymphocytes and plasma cells. The cystic lining epithelium was
not continuous and was of stratified squamous type with areas
exhibiting flattened epithelial cells to cuboidal cells intermixed
with vacuolated cells, suggestive of the infected cyst.
With the results of both aspiration and incisional biopsy being
suggestive of a cystic lesion, complete surgical excision of the cyst
was planned under general anesthesia. The excised specimen
in toto was submitted for histopathological evaluation. The
microscopic features revealed pseudostratified ciliated columnar
epithelium predominantly, and few areas exhibiting 2-4 cell
layered thick stratified cuboidal/squamous nonkeratinized
epithelium associated with fibrovascular connective tissue. The
connective tissue exhibited areas of inflammation, seromucinous
acini, extensive fibrosis, areas showing irregular bony trabeculae
with osteocytes, and extensive hemorrhage suggestive of ciliated
cyst of the maxilla [Figure 4].

Figure 4: Photomicrograph (a) 4 magnification and (b) 40


magnification

disturbances to reactive or inflammatory processes to lesions


of benign or malignant origin. Irrespective of the etiology of
radiolucent lesions, they must be investigated and actively
pursued in order to treat them appropriately.
In the present case report, the radiolucent lesion represented
a ciliated cyst of the maxilla. Clinically, it is commonly seen
among middle-aged patients.[1] The presenting complaints
are usually swelling pain or discomfort involving the maxilla.
The swelling may either present extra-orally or intra-orally
with radiographs revealing a well-defined radiolucent area
anatomically distinct from the maxillary sinus.[1]
A detailed review of the literature with very few exceptions
suggested that ciliated cysts are usually post-operative. Gregory
and Shafer (1958) reported cysts following operations for
maxillary sinusitis, particularly the Cald-well Luc approach
including nasal antrostomy. Wassmund (1939) has published
two case reports on similar cysts arising after gunshot
injuries.[2] Shuttleworth and King (1951) have reported these
cysts following malar slash maxillary fractures.[5] With regard
to the pathogenesis, Kubo (1927 and 1933) postulated that the
lesion originated from the trapped sinus mucosa in the wound
either from a surgical procedure or as a result of trauma.[6] Imai
(1933) hypothesized that mucosa derived from the nasal cavity
regenerated within the antrum.[7] Further, Mohri et al. (1977)
attributed the origin of the lesions to the closure of both the
natural ostium and the intranasal opening following a surgical
procedure.[8]

Discussion
Radiolucent lesions involving the jaws are of utmost concern
to the clinician. These lesions range from developmental
2

Gowthamnath, et al.

Ciliated cyst of the maxilla

following surgery (traumatic theory).[13] Our case stands


unique with a history of trauma 2 years ago resulting in an
identical pathology.

The conventionally termed surgical ciliated cyst is


commonly encountered among the Japanese population but
appears to be a rare entity in the most other parts of the world.[9]
It has a slight male predilection with a wide presenting age range
from 21 to 80 years, and it is thought to occur 3-61 years
following a maxillary surgical procedure.[10]
Radiographs reveal well-defined radiolucent areas in close
relation to the maxillary sinus. In a series of cases reported by
Yamamoto, Takagi most of the cases were unilocular with very
few showing a multilocular presentation.[4] The buccal cortical
bone is usually intact with surrounding bone sclerosis evident
among few cases. Kaneshero et al. reported missing margins
in the lateral and posterior walls of the lesion. Occasionally,
the cystic area encroaches into the sinus but it lacks the
communication between the two which has been demonstrated
by injecting a radiopaque material (Shafer et al. 1983). In the
early diseased stage, there is no bony destruction, but as they
expand the sinus wall, it becomes thinned out and eventually
gets perforated mimicking a malignant neoplasm.[9,11]
Histologically, these cysts are usually lined by pseudostratified ciliated columnar epithelium, with squamous
metaplasia within the chronically inflamed areas.
A combination of ciliated, cuboidal, and squamous epithelium
with varying numbers and layers of mucous cells may be
seen. The underlying connective tissue can be cellular or
fibrotic.[11] Extensive hemorrhage and foci of calcification may
also be present. Mucous retention cyst of the antrum should
be considered as one of the dierential diagnoses as they are
also lined by pseudostratified columnar epithelium. However,
they do not follow an aggressive course, nor do they tend to
present clinically as a swelling. They are usually diagnosed
as incidental findings in an asymptomatic patient. Gardner
(1984) characterizes these harmless lesions as pseudocysts.[12]
To conclude, the most widely accepted theory of pathogenesis
is thought to be due to the entrapment of the sinus mucosa

References
1. Miller R, Longo J, Houston G. Surgical ciliated cyst of the
maxilla. JOral Maxillofac Surg 1988;46:310-2.
2. Sugar AW, Walker DM, Bounds GA. Surgical ciliated
(postoperative maxillary) cysts following mid-face osteotomies.
Br J Oral Maxillofac Surg 1990;28:264-7.
3. Yoshikawa Y, Nakajima T, Kaneshiro S, Sakaguchi M.
Effective treatment of the postoperative maxillary cyst by
marsupialization. JOral Maxillofac Surg 1982;40:487-91.
4. Yamamoto H, Takagi M. Clinicopathologic study of the
postoperative maxillary cyst. Oral Surg Oral Med Oral Pathol
1986;62:544-8.
5. Shuttleworth FN, King PF. Pyocele of orbit following fracture of
the maxilla. Br J Ophthalmol 1951;35:427-8.
6. Kubo I. A buccal cyst occurred after radical operation of the
maxillary sinus. ZOtol Tokyo 1927;39:896-7.
7. Imai T. Mucocele of the maxillary sinus after radical operation
for sinusitis. ZOtol Tokyo 1933;39:723-35.
8. Mohri M, Nishio M, Mohri J, Shimazu K, Akane K. Problems
relating to postoperative maxillary cyst (authors transl). Nihon
Jibiinkoka Gakkai Kaiho 1977;80:326-33.
9. Shear M, Speight P. Cysts of Oral and Maxillofacial Region.
4thed. Singapore: Blackwell Munksgaard; 2007.
10. Nishioka M, Pittella F, Hamagaki M, Okada N, Takagi M.
Prevalence of postoperative maxillary cyst significantly higher
in Japan. Oral Med Pathol 2005;10:9-13.
11. Gardner DG, Gullane PJ. Mucoceles of the maxillary sinus. Oral
Surg Oral Med Oral Pathol 1986;62:538-43.
12. Gardner DG. Pseudocysts and retention cysts of the maxillary
sinus. Oral Surg Oral Med Oral Pathol 1984;58:561-7.
13. Kaneshiro S, Nakajima T, Yoshikawa Y, Iwasaki H, Tokiwa N.
The postoperative maxillary cyst: Report of 71cases. JOral Surg
1981;39:191-8.

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