Traumatic ciliated cyst derived from zygomaticomaxillary fracture: Report of a case

doi:10.1016/j.ajoms.2013.01.004

Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology

Volume 26, Issue 2, April 2014, Pages 175-178

https://doi.org/10.1016/j.ajoms.2013.01.004 Get rights and content

Abstract

A surgical ciliated cyst of maxilla is well known as a delayed complication of radical maxillary sinus surgery for chronic sinusitis. Recent reports have suggested that orthognatic surgery, implant-related surgery, and fracture surgery also causes a development of surgical ciliated cyst. It is documented that small segments of sinus mucosa become entrapped between the bony edges of the maxilla, which resulting in cystic degeneration of the entrapped sinus mucosa many years later. However, few papers are available that fracture individual could lead to a development of a maxillary cyst with pseudostratified columnar ciliated epithelium. Here we report a unique case of the ciliated cyst in the maxilla, supposed to be derived from the zygomaticomaxillary fracture individual conservatively treated 30 years before. The diagnostic difficulty of this lesion is also discussed.

Introduction

It is widely recognized that a surgical ciliated cyst of maxilla occasionally presents as a delayed complication of radical maxillary sinus surgery for chronic sinusitis [1]. Although few reports of the cyst have been published in English-language journals, numerously have been written in Japanese literatures where are especially called as a “postoperative maxillary cyst”. The surgical ciliated cyst clinically presents as a swelling of the maxillary alveolus and palate with pain, discharge, or fistula formation. Radiographically, it appears as a well-defined unilocular radiolucency closely associated with the maxillary sinus. The pathogenesis of the surgical ciliated cyst is generally thought to be due to the entrapment of remnants of sinus mucosa in the wound after maxillary sinus surgery, or the early closure of natural ostium before the sinus is completely filled with regenerating granulation tissue [2]. Since the findings of histopathology commonly note that a cystic lesion is lined by a pseudostratified columnar ciliated epithelium of respiratory type, these vital findings could be greatly helpful to make a proper diagnosis for cyst.

Recent reports have suggested that orthognatic surgery [3], implant-related surgery [4], and fracture surgery [5] also causes a development of surgical ciliated cyst. Each report documents that small segments of sinus mucosa become entrapped between the bony edges of the maxilla, which resulting in cystic degeneration of the entrapped sinus mucosa many years later. This means that, by each manner, somehow a previous maxillary invasion could be necessary to develop the cyst. However, few reports are available that fracture individual could lead to a development of a maxillary cyst with pseudostratified columnar ciliated epithelium. Here we report a unique case of the ciliated cyst in the maxilla, supposed to be derived from the zygomaticomaxillary fracture individual that the conservative treatment was adopted 30 years before. The difficulty of diagnosis and the new diagnostic classification of this lesion are also discussed.

Section snippets

Report of a case

The patient was a 56-year-old female. In July 2010 she developed nasal discharge in the left nose and spontaneous pain and slight swelling in the left cheek region. In the beginning of August 2010 she was referred to our outpatient clinic with the swelling on the left cheek region as a chief complaint. Since she had the partial bone defect on the buccal side of the left maxilla, the lesion aspiration obtained the brown mucinous fluid. The extraoral findings showed the tenderness on the left

Discussion

To our knowledge, this is a first report to show the ciliated cyst of maxilla derived from the zygomaticomaxillary fracture individual with no surgeries. Preoperatively, the patient presented the slight swelling on the left cheek, the findings of cystic lesion were noted on the image examinations, and the medical history was recorded, as the zygomaticomaxillary fracture conservatively treated 30 years before. However, this lesion expanded as multilocular form, in marked contrast to unilocular

Conflict of interest

All authors have no financial or personal relationships with other people or organizations that could inappropriately influence (bias) their work.

Funding

None.

Ethical approval

Not required.

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Cited by (1)

Surgical ciliated cyst developing after Le Fort I osteotomy: Case report and review of the literature
2019, Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology
Citation Excerpt :
To the best of our knowledge, this represents the first reported case of a surgical ciliated cyst that occurred after maxillary orthognathic surgery within the anterior hard palate and in association with nasal mucosa. Misch et al described a case of postoperative maxillary cyst that developed after a sinus elevation procedure in 1991 [6], and Toyoshima et al described a case of traumatic ciliated cyst from zygomaticomaxillary fracture in 2014 [7]. In early-February 2012, a 20-year-old man diagnosed as jaw deformities at orthodontic clinic visited our hospital for the purpose of preoperative examination.
A surgical ciliated cyst, also referred as a locally aggressive lesion, stems from a prior surgery several years later as a delayed complication. It is reported that the cyst develops after orthognathic surgery. A 25-year-old man who had undergone Le Fort I osteotomy and bilateral intraoral vertical osteotomy of the mandible as treatment for jaw deformities in 2013 was referred to our hospital because of a swelling on the right side of the hard palate. Radiography and cone-beam computed tomography examinations revealed a unilocular cystic lesion in the right maxillary alveolus. All the teeth in the maxillary right quadrant were vital to electric pulp testing. Aspiration with an 18-gauge needle extracted rust-colored mucoid fluid. The lesion was diagnosed to be a maxillary cyst. Under general anesthesia, we removed the osteotomy fixation plates and excised the lesion.
Histopathological examination revealed a cyst comprising cellular fibrous connective tissue, mainly lined by stratified squamous epithelium; however, the regions of focal pseudostratified columnar and ciliated epithelium were also present. Surgical ciliated cyst was diagnosed on the basis of histopathological, clinical, and radiographical findings.
Although surgical ciliated cysts have sparsely been reported after orthognathic surgery, an increased awareness of such possibility is required to avoid delays in diagnosis. This lesion should always be a part of differential diagnosis of symptomatic patients who, in the past, have undergone antral or maxillary orthognathic surgery.

^☆: AsianAOMS: Asian Association of Oral and Maxillofacial Surgeons; ASOMP: Asian Society of Oral and Maxillofacial Pathology; JSOP: Japanese Society of Oral Pathology; JSOMS: Japanese Society of Oral and Maxillofacial Surgeons; JSOM: Japanese Society of Oral Medicine; JAMI: Japanese Academy of Maxillofacial Implants.

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Case reportTraumatic ciliated cyst derived from zygomaticomaxillary fracture: Report of a case☆