Accepted Manuscript Rare mandibular surgical ciliated cysts: report of two new cases Chia-Cheng Li, D.D.S., M.S. David M. Feinerman, D.M.D., M.D., P.A. Killian D. Maccarthy, D.M.D., M.D. Sook-Bin Woo, D.M.D., M.M.Sc. PII:
S0278-2391(14)00437-6
DOI:
10.1016/j.joms.2014.04.010
Reference:
YJOMS 56292
To appear in:
Journal of Oral and Maxillofacial Surgery
Received Date: 4 March 2014 Revised Date:
10 April 2014
Accepted Date: 12 April 2014
Please cite this article as: Li C-C, Feinerman DM, Maccarthy KD, Woo S-B, Rare mandibular surgical ciliated cysts: report of two new cases, Journal of Oral and Maxillofacial Surgery (2014), doi: 10.1016/ j.joms.2014.04.010. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
ACCEPTED MANUSCRIPT
Title
RI PT
Rare mandibular surgical ciliated cysts: report of two new cases
Authors Chia-Cheng Li, D.D.S., M.S. a, *
SC
David M. Feinerman, D.M.D., M.D., P.A. b,c Killian D Maccarthy, D.M.D., M.D. d
a
M AN U
Sook-Bin Woo, D.M.D., M.M.Sc. a, e, f
Department of Oral Medicine, Infection, and Immunity, Harvard School of Dental Medicine,
Boston, MA 02115, USA b
Private practice, Boynton Beach, FL 33436, USA
c
e
Private practice, South Portland, ME 04106, USA
EP
d
TE D
Clinical Instructor, Department of Oral and Maxillofacial Surgery, Nova Southeastern University College of Dental Medicine.
Division of Oral Medicine and Dentistry, Brigham and Women’s Hospital, Boston, MA 02115,
f
AC C
USA
Center for Oral Pathology, StrataDx, Lexington, MA 02421, USA
The authors have no financial or commercial obligations to disclose. *
Corresponding author (C.-C. Li)
Address: 188 Longwood Avenue, Boston, MA 02115, USA; Phone number: 1-617-651-0973; Fax number: 1-617-525-4751; Email: [email protected]
ACCEPTED MANUSCRIPT
Title Rare mandibular surgical ciliated cysts: report of two new cases
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Abstract
The surgical ciliated cyst, also known as postoperative maxillary cyst or implantation cyst, occurs as a result of iatrogenic implantation of respiratory epithelium into a non-contiguous
SC
surgical site following sinus surgery. It typically presents as a well-defined radiolucency in the maxilla in young adults. Histopathologically, the cyst is lined by ciliated columnar, cuboidal or
M AN U
pseudostratified squamous epithelium with mucous cells. Here, we report two rare cases of surgical ciliated cyst located in the mandible.
Introduction
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The term ”surgical ciliated cyst” is often used interchangeably with the terms “postoperative maxillary cyst” and “implantation cyst”, and this condition has been well-described in the Japanese literature since the 1980s.1, 2 However, it is uncommonly reported in Western
EP
populations.3 Typically, the surgical ciliated cyst occurs in the posterior maxilla, presenting as an expansile lesion. It is associated with a history of previous maxillary sinus surgery and is
AC C
believed to arise from the iatrogenic implantation of respiratory epithelium.2 The conventional treatment for surgical ciliated cyst is enucleation or marsupialization if the lesion is large; recurrence may occur if cyst lining is incompletely removed.4, 5 Here we report two cases of surgical ciliated cyst that developed in the mandible.
Case Reports
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Case 1 A 72 year-old male presented with a painless swelling in the anterior mandible. The panoramic radiograph revealed a 3.0 cm, well-defined radiolucency extending from the left to
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the right mandibular canine (Fig. 1A). His medical history was significant hypertension and hypercholesterolemia and he was taking atorvastatin, amlodipine and ramipril. Fifty-six years prior at the age of 16, the patient had undergone simultaneous rhinoplasty and genioplasty for
SC
cosmetic purpose.
The lesion was removed by curettage (Fig. 1B). The defect was filled with bone graft with
M AN U
recombinant bone morphogenetic protien-2 (INFUSE® Bone Graft, Memphis, TN), and the one year follow-up showed no pathology with normal healing. Histopathologically, the cyst wall was densely fibrous with scattered lymphocytes (Fig. 1C). It was generally lined by 2 layers of low cuboidal-to-columnar epithelium with luminal ciliated cells, as well as foci of nonkeratinized
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stratified squamous epithelium; mucous cells were not present (Fig. 1D). There was hyalinization of the fibrous tissue beneath the epithelium. Based on the history, radiographic and histopathologic findings, the diagnosis was surgical ciliated cyst. No recurrence was detected at a
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Case 2
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14-month follow up (Fig. 1E).
A 42 year-old male presented a well-defined radiolucency, measuring 2.7 × 0.9 cm in the right ascending ramus (Fig. 2A). In addition, he had developed numbness of his lower lip and chin of the right side three days prior. His medical history was essentially non-contributory and he had been placed on gabapentin, amoxicillin with clavulanic acid, and hydrocondone with acetaminophen by his primary care physician for a putative diagnosis of trigeminal neuralgia and
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sialadenitis. Eighteen years ago at age 24, the patient had undergone in one operative session, segmented four-piece Le Fort I with wire osteosynthesis, bilateral inferior turbinectomies, bilateral mandibular sagittal split osteotomy with interpositional bone grafts and rigid internal
RI PT
fixation and genioplasty to correct his vertical maxillary hyperplasia with anterior open bite and mandbibular retrognathism. An excision was performed and diagnosed as a “glandular
odontogenic cyst” in another institution. However, consultation with our service revealed a
SC
surgical ciliated cyst. His right mental nerve paresthesia resolved completely after the removal of this cystic lesion.
M AN U
Two years later when he presented for removal of surgical screws, an asymptomatic, approximately 1.0 × 2.5 cm, well-defined radiolucency had developed at the same location (Fig. 2B). The lesion was curetted and the screws were removed. Histopathologically, the cyst was lined by 2 layers of low cuboidal-to-columnar cells with luminal ciliated columnar cells and a
TE D
mild lymphocytic infiltrate within the fibrous cyst wall with hyalinization of the basement membrane zone (Fig. 2C-D). Based on the history, radiographic and histopathologic findings, the diagnosis was surgical ciliated cyst, either residual or recurrent. After the initial follow-up 11
EP
days after the procedure, the patient failed multiple subsequent follow-up appointments, but did
AC C
report that he had no numbness.
Discussion
Surgical ciliated cyst usually occurs in the maxilla and is a complication associated with sinus surgery.1, 2 Several theories on etiopathogenesis exist. The widely-accepted theory is that iatrogenically implanted respiratory epithelium from sinus surgery (such as the Caldwell-Luc
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procedure or Le Fort I osteotomy) contaminates the maxillary wound, proliferates and undergoes cystic change and enlargement.2, 5, 6 Surgical ciliated cyst has been well described in the Japanese literature in patients in the 4th
RI PT
and 5th decades (mean 41 years) without gender predilection; up to 20% of the patients who had undergone surgery of the maxillary sinus developed surgical ciliated cyst within 10-29 years (mean 19.6 years); no mandibular surgical cysts were noted.1, 2 Most of the surgical ciliated cysts
SC
occur in the posterior maxilla, typically presenting as an expansile swelling in the maxillary sulcus. Pain and purulent discharge were noted if secondarily infected.1, 2 Antral pseudocyst is
M AN U
often the clinical impression.7 Radiographs show a well-demarcated unilocular radiolucency with a sclerotic border. Histopathologically, the cyst is lined by ciliated columnar or cuboidal epithelium with occasional goblet cells; areas lined by nonkeratinized stratified squamous epithelium may be present.5 The underlying connective tissue is sometimes hyalinized.8
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Treatment is curettage or enucleation. Marsupialization is recommended if the lesion is extensive involving cortical bone. The prognosis is good, although residual disease may progress if not completely removed.4
EP
Surgical ciliated cysts are rarely reported in the non-Japanese population 7, and mandibular surgical ciliated cysts are even rarer. Only eight cases in the mandible have been reported in the
AC C
English literature (Table 1). The clinical symptoms, radiographic and histopathologic characteristics of mandibular surgical ciliated cysts are identical to the maxillary counterparts. The putative etiopathogenesis is that respiratory epithelium from a surgical procedure involving the maxillary sinus may have contaminated the blade or autograft material which is then inadvertently implanted into the mandible during the second subsequent surgical procedure.9 As such, the demographic features would be expected to conform to those of the conventional
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maxillary surgical ciliated cyst and a review of these 10 cases showed this to be true; namely, patients are in the 5th and 6th decades (mean 43 years, range 24-72 ) without significant gender predominance (6 males and 4 females). Development of the lesions occurs at a mean of 24.7
RI PT
years after the simultaneous surgery of the maxillary sinus and mandible. Because orthognathic surgery often involves genioplasty or mandular advancement/repositioning, the anterior mandible and chin areas are often involved (8 of 10 cases) (Table 1).
SC
Many odontogenic and non-odontogenic cysts, and odontogenic and non-odontogenic tumors may present as well-demarcated radiolucencies in the anterior mandible and the definitive
M AN U
diagnosis is made on histopathology. The most important differential diagnoses for a mandibular surgical ciliated cyst include developmental odontogenic cysts such as glandular odontogenic cyst and dentigerous cyst with mucous cell prosoplasia. Glandular odontogenic cyst is lined by stratified squamous epithelium that varies in thickness, exhibiting a complex architecture with
TE D
multiple compartments, often with epithelial plaques. The luminal cells are cuboidal with eosinophilic cytoplasm often exhibiting a hobnail appearance, apocrine snouting or ciliation.10, 11 Microcystic structures, mucous cells and clear cells may be seen. These lesions are locally
EP
invasive and the recurrence rate is up to 50%.10 The surgical ciliated cyst may show the presence of mucous cells, but is otherwise a simple cyst without duct formation and does not usually recur.
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Dentigerous cyst with mucous cell prosoplasia may look similar and in some cases ciliated cells may be noted, but it is always associated with an impacted tooth, while the surgical ciliated cyst is not.12 A primordial cyst from a supernumerary tooth (such as a mandibular premolar) is usually lined by simple stratified squamous epithelium and it is conceivable that mucous or ciliated cells may be present. Lastly, what used to be termed “median mandibular cyst” (thought to be a developmental non-odontogenic or “fissural” cyst) may demonstrate similar
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histopathologic features of a surgical ciliated cyst; however, this term is no longer used since these cysts have been re-interpreted as being of odontogenic origin.19 The history of previous sinus surgery is invaluable in definitively establishing the diagnosis.
RI PT
This is a report of two surgical ciliated cysts arising within the mandible. Both clinicians and pathologists should be aware of this entity as a potential complication in patients who had
undergone surgery in a single session involving both the maxillary sinus and the mandible. One
AC C
EP
TE D
M AN U
cyst represents recurrence or progression of residual disease.
SC
of the cases (case 2) was originally curetted and it is unclear whether the re-appearance of the
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Table 1 Clinical features of previous and current cases with mandibular surgical ciliated cyst Signs and symptoms Gradual swelling for 18 months, with ulceration and episodic pain for 3 years
Anastassov and Lee14
53/M
Anterior mandible
Gradually expanding swelling for 6 months
Kelly et al.15
56/F
Anterior mandible
Tense swelling with tenderness and pain for several weeks
Imholte et al.16
59/M
Anterior mandible
Acute, fluctuant swelling with purulent discharge
Koutlas et al.9
34/F
Left mandibular ramus
Mild discomfort and swelling with intraoral fistula
Bourgeois and Nelson3
27/F
Mandibular canine and first premolar area
Asymptomatic but with milkywhite semi-purulent aspirate, and perforation of cortical plate were noted
Lazar et al.17
24/M
Submental region
Discomfort and swelling with abscess
Ragsdale et al.18
30/M
Anterior mandible
Acute pain swelling with purulent discharge
Li et al.
72/M
Anterior mandible
Painless swelling
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Right mandibular ramus
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42/M
Past surgical history Simultaneous reduction rhinoplasty and chin augmentation 15 years prior Simultaneous septorhinoplasty and chin augmentation 39 years prior Simultaneous rhinoplasty and genioplasty 40 years prior Simultaneous septorhinoplasty and chin augmentation 40 years prior Simultaneous orthognathic surgery of maxilla and mandible 13 years prior Le Fort I vertical zygomaticomaxillary and sliding genioplasty osteotomies 4 years prior Simultaneous rhinoplasty and mentoplasty 6 years prior
Mental nerve paresthesia
Treatment Enucleation
Enucleation
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Site Anterior mandible
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Age/Gender 33/F
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Authors Nastri and Hookey13
Le Fort I vertical zygomaticomaxillary and sliding genioplasty osteotomies 16 years prior Simultaneous septorhinoplasty and genioplasty 56 years prior Simultaneous orthognathic surgery of maxilla and mandible 18 years prior
Intraoral drainage for 3 days and enucleation Enucleation
Intraoral drainage and then enucleation Enucleation
Excision with a long term intraoral drainage Enucleation and peripheral ostectomy Enucleation
Curettage
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References 1. Kaneshiro S, Nakajima T, Yoshikawa Y et al.: The postoperative maxillary cyst: report of 71 cases. J Oral Surg 39:191, 1981
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2. Yamamoto H, Takagi M: Clinicopathologic study of the postoperative maxillary cyst. Oral Surg Oral Med Oral Pathol 62:544, 1986
3. Bourgeois SL, Jr., Nelson BL: Surgical ciliated cyst of the mandible secondary to
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simultaneous Le Fort I osteotomy and genioplasty: report of case and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 100:36, 2005
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4. Yoshikawa Y, Nakajima T, Kaneshiro S, Sakaguchi M: Effective treatment of the postoperative maxillary cyst by marsupialization. J Oral Maxillofac Surg 40:487, 1982 5. Leung YY, Wong WY, Cheung LK: Surgical ciliated cysts may mimic radicular cysts or residual cysts of maxilla: report of 3 cases. J Oral Maxillofac Surg 70:e264, 2012
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6. Amin M, Witherow H, Lee R, Blenkinsopp P: Surgical ciliated cyst after maxillary orthognathic surgery: report of a case. J Oral Maxillofac Surg 61:138, 2003 7. Basu MK, Rout PG, Rippin JW, Smith AJ: The post-operative maxillary cyst. Experience
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with 23 cases. Int J Oral Maxillofac Surg 17:282, 1988 8. Cano J, Campo J, Alobera MA, Baca R: Surgical ciliated cyst of the maxilla. Clinical case.
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Med Oral Patol Oral Cir Bucal 14:E361, 2009 9. Koutlas IG, Gillum RB, Harris MW, Brown BA: Surgical (implantation) cyst of the mandible with ciliated respiratory epithelial lining: a case report. J Oral Maxillofac Surg 60:324, 2002 10. Fowler CB, Brannon RB, Kessler HP et al.: Glandular odontogenic cyst: analysis of 46 cases with special emphasis on microscopic criteria for diagnosis. Head Neck Pathol 5:364, 2011
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11. Kaplan I, Anavi Y, Hirshberg A: Glandular odontogenic cyst: a challenge in diagnosis and treatment. Oral Dis 14:575, 2008 12. Zhang LL, Yang R, Zhang L et al.: Dentigerous cyst: a retrospective clinicopathological
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analysis of 2082 dentigerous cysts in British Columbia, Canada. Int J Oral Maxillofac Surg 39:878, 2010
autogenous bone. Int J Oral Maxillofac Surg 23:372, 1994
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13. Nastri AL, Hookey SR: Respiratory epithelium in a mandibular cyst after grafting of
14. Anastassov GE, Lee H: Respiratory mucocele formation after augmentation genioplasty with
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nasal osteocartilaginous graft. Int J Oral Maxillofac Surg 57:1263, 1999
15. Kelly JP, Malik S, Stucki-McCormick SU: Tender swelling of the chin 40 years after genioplasty. J Oral Maxillofac Surg 58:203, 2000
16. Imholte M, Schwartz HC: Respiratory implantation cyst of the mandible after chin
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augmentation: report of case. Otolaryngol Head Neck Surg 124:586, 2001 17. Lazar F, zur Hausen A, Mischkowski R, Zoller JE: Atypical cyst formation following chin augmentation using a nasal osteocartilaginous graft. J Craniomaxillofac Surg 34:107, 2006
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18. Ragsdale BD, Laurent JL, Janette AJ, Epker BN: Respiratory implantation cyst of the mandible following orthognathic surgery. J Oral Maxillofac Pathol 13:30, 2009
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19. Gardner DG: An evaluation of reported cases of median mandibular cysts. Oral Surg Oral Med Oral Pathol 65:208, 1988
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Figure Legends Fig. 1A A well-circumscribed unilocular radiolucency extended from the apices of mandibular right to left canines.
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Fig. 1B The cyst bulges from the facial aspect of the anterior mandible.
Fig. 1C The cyst was lined by thin epithelium with mural fibrosis and mild chronic inflammation. (H&E, х 100)
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Fig. 1D The lining epithelium was composed of 2 layers of low cuboidal-to-columnar cells with ciliated luminal cells; there is subepithelial hyalinization. (H&E, х 600)
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Fig. 1E Panoramic radiograph at 14 months post-operative, showing good bone fill. Fig. 2A A well-demarcated unilocular radiolucency with a sclerotic border in the right mandible associated with fixation screws.
Fig. 2B A well-defined radiolucency recurred at the right ramus area, measuring 1 x 2.5 cm that
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developed two years later.
Fig. 2C The cyst was lined by thin epithelium with mural fibrosis and mild chronic inflammation. Note osteoclasts at the base of the tissue. (H&E, х 200)
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Fig. 2D The cyst was lined by 2 cell layers of low cuboidal-to-columnar cells with luminal
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ciliated cells. (H&E, х 600)
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S0278-2391(14)00437-6
DOI:
10.1016/j.joms.2014.04.010
Reference:
YJOMS 56292
To appear in:
Journal of Oral and Maxillofacial Surgery
Received Date: 4 March 2014 Revised Date:
10 April 2014
Accepted Date: 12 April 2014
Please cite this article as: Li C-C, Feinerman DM, Maccarthy KD, Woo S-B, Rare mandibular surgical ciliated cysts: report of two new cases, Journal of Oral and Maxillofacial Surgery (2014), doi: 10.1016/ j.joms.2014.04.010. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
ACCEPTED MANUSCRIPT
Title
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Rare mandibular surgical ciliated cysts: report of two new cases
Authors Chia-Cheng Li, D.D.S., M.S. a, *
SC
David M. Feinerman, D.M.D., M.D., P.A. b,c Killian D Maccarthy, D.M.D., M.D. d
a
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Sook-Bin Woo, D.M.D., M.M.Sc. a, e, f
Department of Oral Medicine, Infection, and Immunity, Harvard School of Dental Medicine,
Boston, MA 02115, USA b
Private practice, Boynton Beach, FL 33436, USA
c
e
Private practice, South Portland, ME 04106, USA
EP
d
TE D
Clinical Instructor, Department of Oral and Maxillofacial Surgery, Nova Southeastern University College of Dental Medicine.
Division of Oral Medicine and Dentistry, Brigham and Women’s Hospital, Boston, MA 02115,
f
AC C
USA
Center for Oral Pathology, StrataDx, Lexington, MA 02421, USA
The authors have no financial or commercial obligations to disclose. *
Corresponding author (C.-C. Li)
Address: 188 Longwood Avenue, Boston, MA 02115, USA; Phone number: 1-617-651-0973; Fax number: 1-617-525-4751; Email: [email protected]
ACCEPTED MANUSCRIPT
Title Rare mandibular surgical ciliated cysts: report of two new cases
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Abstract
The surgical ciliated cyst, also known as postoperative maxillary cyst or implantation cyst, occurs as a result of iatrogenic implantation of respiratory epithelium into a non-contiguous
SC
surgical site following sinus surgery. It typically presents as a well-defined radiolucency in the maxilla in young adults. Histopathologically, the cyst is lined by ciliated columnar, cuboidal or
M AN U
pseudostratified squamous epithelium with mucous cells. Here, we report two rare cases of surgical ciliated cyst located in the mandible.
Introduction
TE D
The term ”surgical ciliated cyst” is often used interchangeably with the terms “postoperative maxillary cyst” and “implantation cyst”, and this condition has been well-described in the Japanese literature since the 1980s.1, 2 However, it is uncommonly reported in Western
EP
populations.3 Typically, the surgical ciliated cyst occurs in the posterior maxilla, presenting as an expansile lesion. It is associated with a history of previous maxillary sinus surgery and is
AC C
believed to arise from the iatrogenic implantation of respiratory epithelium.2 The conventional treatment for surgical ciliated cyst is enucleation or marsupialization if the lesion is large; recurrence may occur if cyst lining is incompletely removed.4, 5 Here we report two cases of surgical ciliated cyst that developed in the mandible.
Case Reports
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Case 1 A 72 year-old male presented with a painless swelling in the anterior mandible. The panoramic radiograph revealed a 3.0 cm, well-defined radiolucency extending from the left to
RI PT
the right mandibular canine (Fig. 1A). His medical history was significant hypertension and hypercholesterolemia and he was taking atorvastatin, amlodipine and ramipril. Fifty-six years prior at the age of 16, the patient had undergone simultaneous rhinoplasty and genioplasty for
SC
cosmetic purpose.
The lesion was removed by curettage (Fig. 1B). The defect was filled with bone graft with
M AN U
recombinant bone morphogenetic protien-2 (INFUSE® Bone Graft, Memphis, TN), and the one year follow-up showed no pathology with normal healing. Histopathologically, the cyst wall was densely fibrous with scattered lymphocytes (Fig. 1C). It was generally lined by 2 layers of low cuboidal-to-columnar epithelium with luminal ciliated cells, as well as foci of nonkeratinized
TE D
stratified squamous epithelium; mucous cells were not present (Fig. 1D). There was hyalinization of the fibrous tissue beneath the epithelium. Based on the history, radiographic and histopathologic findings, the diagnosis was surgical ciliated cyst. No recurrence was detected at a
AC C
Case 2
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14-month follow up (Fig. 1E).
A 42 year-old male presented a well-defined radiolucency, measuring 2.7 × 0.9 cm in the right ascending ramus (Fig. 2A). In addition, he had developed numbness of his lower lip and chin of the right side three days prior. His medical history was essentially non-contributory and he had been placed on gabapentin, amoxicillin with clavulanic acid, and hydrocondone with acetaminophen by his primary care physician for a putative diagnosis of trigeminal neuralgia and
ACCEPTED MANUSCRIPT
sialadenitis. Eighteen years ago at age 24, the patient had undergone in one operative session, segmented four-piece Le Fort I with wire osteosynthesis, bilateral inferior turbinectomies, bilateral mandibular sagittal split osteotomy with interpositional bone grafts and rigid internal
RI PT
fixation and genioplasty to correct his vertical maxillary hyperplasia with anterior open bite and mandbibular retrognathism. An excision was performed and diagnosed as a “glandular
odontogenic cyst” in another institution. However, consultation with our service revealed a
SC
surgical ciliated cyst. His right mental nerve paresthesia resolved completely after the removal of this cystic lesion.
M AN U
Two years later when he presented for removal of surgical screws, an asymptomatic, approximately 1.0 × 2.5 cm, well-defined radiolucency had developed at the same location (Fig. 2B). The lesion was curetted and the screws were removed. Histopathologically, the cyst was lined by 2 layers of low cuboidal-to-columnar cells with luminal ciliated columnar cells and a
TE D
mild lymphocytic infiltrate within the fibrous cyst wall with hyalinization of the basement membrane zone (Fig. 2C-D). Based on the history, radiographic and histopathologic findings, the diagnosis was surgical ciliated cyst, either residual or recurrent. After the initial follow-up 11
EP
days after the procedure, the patient failed multiple subsequent follow-up appointments, but did
AC C
report that he had no numbness.
Discussion
Surgical ciliated cyst usually occurs in the maxilla and is a complication associated with sinus surgery.1, 2 Several theories on etiopathogenesis exist. The widely-accepted theory is that iatrogenically implanted respiratory epithelium from sinus surgery (such as the Caldwell-Luc
ACCEPTED MANUSCRIPT
procedure or Le Fort I osteotomy) contaminates the maxillary wound, proliferates and undergoes cystic change and enlargement.2, 5, 6 Surgical ciliated cyst has been well described in the Japanese literature in patients in the 4th
RI PT
and 5th decades (mean 41 years) without gender predilection; up to 20% of the patients who had undergone surgery of the maxillary sinus developed surgical ciliated cyst within 10-29 years (mean 19.6 years); no mandibular surgical cysts were noted.1, 2 Most of the surgical ciliated cysts
SC
occur in the posterior maxilla, typically presenting as an expansile swelling in the maxillary sulcus. Pain and purulent discharge were noted if secondarily infected.1, 2 Antral pseudocyst is
M AN U
often the clinical impression.7 Radiographs show a well-demarcated unilocular radiolucency with a sclerotic border. Histopathologically, the cyst is lined by ciliated columnar or cuboidal epithelium with occasional goblet cells; areas lined by nonkeratinized stratified squamous epithelium may be present.5 The underlying connective tissue is sometimes hyalinized.8
TE D
Treatment is curettage or enucleation. Marsupialization is recommended if the lesion is extensive involving cortical bone. The prognosis is good, although residual disease may progress if not completely removed.4
EP
Surgical ciliated cysts are rarely reported in the non-Japanese population 7, and mandibular surgical ciliated cysts are even rarer. Only eight cases in the mandible have been reported in the
AC C
English literature (Table 1). The clinical symptoms, radiographic and histopathologic characteristics of mandibular surgical ciliated cysts are identical to the maxillary counterparts. The putative etiopathogenesis is that respiratory epithelium from a surgical procedure involving the maxillary sinus may have contaminated the blade or autograft material which is then inadvertently implanted into the mandible during the second subsequent surgical procedure.9 As such, the demographic features would be expected to conform to those of the conventional
ACCEPTED MANUSCRIPT
maxillary surgical ciliated cyst and a review of these 10 cases showed this to be true; namely, patients are in the 5th and 6th decades (mean 43 years, range 24-72 ) without significant gender predominance (6 males and 4 females). Development of the lesions occurs at a mean of 24.7
RI PT
years after the simultaneous surgery of the maxillary sinus and mandible. Because orthognathic surgery often involves genioplasty or mandular advancement/repositioning, the anterior mandible and chin areas are often involved (8 of 10 cases) (Table 1).
SC
Many odontogenic and non-odontogenic cysts, and odontogenic and non-odontogenic tumors may present as well-demarcated radiolucencies in the anterior mandible and the definitive
M AN U
diagnosis is made on histopathology. The most important differential diagnoses for a mandibular surgical ciliated cyst include developmental odontogenic cysts such as glandular odontogenic cyst and dentigerous cyst with mucous cell prosoplasia. Glandular odontogenic cyst is lined by stratified squamous epithelium that varies in thickness, exhibiting a complex architecture with
TE D
multiple compartments, often with epithelial plaques. The luminal cells are cuboidal with eosinophilic cytoplasm often exhibiting a hobnail appearance, apocrine snouting or ciliation.10, 11 Microcystic structures, mucous cells and clear cells may be seen. These lesions are locally
EP
invasive and the recurrence rate is up to 50%.10 The surgical ciliated cyst may show the presence of mucous cells, but is otherwise a simple cyst without duct formation and does not usually recur.
AC C
Dentigerous cyst with mucous cell prosoplasia may look similar and in some cases ciliated cells may be noted, but it is always associated with an impacted tooth, while the surgical ciliated cyst is not.12 A primordial cyst from a supernumerary tooth (such as a mandibular premolar) is usually lined by simple stratified squamous epithelium and it is conceivable that mucous or ciliated cells may be present. Lastly, what used to be termed “median mandibular cyst” (thought to be a developmental non-odontogenic or “fissural” cyst) may demonstrate similar
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histopathologic features of a surgical ciliated cyst; however, this term is no longer used since these cysts have been re-interpreted as being of odontogenic origin.19 The history of previous sinus surgery is invaluable in definitively establishing the diagnosis.
RI PT
This is a report of two surgical ciliated cysts arising within the mandible. Both clinicians and pathologists should be aware of this entity as a potential complication in patients who had
undergone surgery in a single session involving both the maxillary sinus and the mandible. One
AC C
EP
TE D
M AN U
cyst represents recurrence or progression of residual disease.
SC
of the cases (case 2) was originally curetted and it is unclear whether the re-appearance of the
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Table 1 Clinical features of previous and current cases with mandibular surgical ciliated cyst Signs and symptoms Gradual swelling for 18 months, with ulceration and episodic pain for 3 years
Anastassov and Lee14
53/M
Anterior mandible
Gradually expanding swelling for 6 months
Kelly et al.15
56/F
Anterior mandible
Tense swelling with tenderness and pain for several weeks
Imholte et al.16
59/M
Anterior mandible
Acute, fluctuant swelling with purulent discharge
Koutlas et al.9
34/F
Left mandibular ramus
Mild discomfort and swelling with intraoral fistula
Bourgeois and Nelson3
27/F
Mandibular canine and first premolar area
Asymptomatic but with milkywhite semi-purulent aspirate, and perforation of cortical plate were noted
Lazar et al.17
24/M
Submental region
Discomfort and swelling with abscess
Ragsdale et al.18
30/M
Anterior mandible
Acute pain swelling with purulent discharge
Li et al.
72/M
Anterior mandible
Painless swelling
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Right mandibular ramus
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42/M
Past surgical history Simultaneous reduction rhinoplasty and chin augmentation 15 years prior Simultaneous septorhinoplasty and chin augmentation 39 years prior Simultaneous rhinoplasty and genioplasty 40 years prior Simultaneous septorhinoplasty and chin augmentation 40 years prior Simultaneous orthognathic surgery of maxilla and mandible 13 years prior Le Fort I vertical zygomaticomaxillary and sliding genioplasty osteotomies 4 years prior Simultaneous rhinoplasty and mentoplasty 6 years prior
Mental nerve paresthesia
Treatment Enucleation
Enucleation
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Site Anterior mandible
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Age/Gender 33/F
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Authors Nastri and Hookey13
Le Fort I vertical zygomaticomaxillary and sliding genioplasty osteotomies 16 years prior Simultaneous septorhinoplasty and genioplasty 56 years prior Simultaneous orthognathic surgery of maxilla and mandible 18 years prior
Intraoral drainage for 3 days and enucleation Enucleation
Intraoral drainage and then enucleation Enucleation
Excision with a long term intraoral drainage Enucleation and peripheral ostectomy Enucleation
Curettage
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References 1. Kaneshiro S, Nakajima T, Yoshikawa Y et al.: The postoperative maxillary cyst: report of 71 cases. J Oral Surg 39:191, 1981
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2. Yamamoto H, Takagi M: Clinicopathologic study of the postoperative maxillary cyst. Oral Surg Oral Med Oral Pathol 62:544, 1986
3. Bourgeois SL, Jr., Nelson BL: Surgical ciliated cyst of the mandible secondary to
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simultaneous Le Fort I osteotomy and genioplasty: report of case and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 100:36, 2005
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4. Yoshikawa Y, Nakajima T, Kaneshiro S, Sakaguchi M: Effective treatment of the postoperative maxillary cyst by marsupialization. J Oral Maxillofac Surg 40:487, 1982 5. Leung YY, Wong WY, Cheung LK: Surgical ciliated cysts may mimic radicular cysts or residual cysts of maxilla: report of 3 cases. J Oral Maxillofac Surg 70:e264, 2012
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6. Amin M, Witherow H, Lee R, Blenkinsopp P: Surgical ciliated cyst after maxillary orthognathic surgery: report of a case. J Oral Maxillofac Surg 61:138, 2003 7. Basu MK, Rout PG, Rippin JW, Smith AJ: The post-operative maxillary cyst. Experience
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with 23 cases. Int J Oral Maxillofac Surg 17:282, 1988 8. Cano J, Campo J, Alobera MA, Baca R: Surgical ciliated cyst of the maxilla. Clinical case.
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Med Oral Patol Oral Cir Bucal 14:E361, 2009 9. Koutlas IG, Gillum RB, Harris MW, Brown BA: Surgical (implantation) cyst of the mandible with ciliated respiratory epithelial lining: a case report. J Oral Maxillofac Surg 60:324, 2002 10. Fowler CB, Brannon RB, Kessler HP et al.: Glandular odontogenic cyst: analysis of 46 cases with special emphasis on microscopic criteria for diagnosis. Head Neck Pathol 5:364, 2011
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11. Kaplan I, Anavi Y, Hirshberg A: Glandular odontogenic cyst: a challenge in diagnosis and treatment. Oral Dis 14:575, 2008 12. Zhang LL, Yang R, Zhang L et al.: Dentigerous cyst: a retrospective clinicopathological
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analysis of 2082 dentigerous cysts in British Columbia, Canada. Int J Oral Maxillofac Surg 39:878, 2010
autogenous bone. Int J Oral Maxillofac Surg 23:372, 1994
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13. Nastri AL, Hookey SR: Respiratory epithelium in a mandibular cyst after grafting of
14. Anastassov GE, Lee H: Respiratory mucocele formation after augmentation genioplasty with
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nasal osteocartilaginous graft. Int J Oral Maxillofac Surg 57:1263, 1999
15. Kelly JP, Malik S, Stucki-McCormick SU: Tender swelling of the chin 40 years after genioplasty. J Oral Maxillofac Surg 58:203, 2000
16. Imholte M, Schwartz HC: Respiratory implantation cyst of the mandible after chin
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augmentation: report of case. Otolaryngol Head Neck Surg 124:586, 2001 17. Lazar F, zur Hausen A, Mischkowski R, Zoller JE: Atypical cyst formation following chin augmentation using a nasal osteocartilaginous graft. J Craniomaxillofac Surg 34:107, 2006
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18. Ragsdale BD, Laurent JL, Janette AJ, Epker BN: Respiratory implantation cyst of the mandible following orthognathic surgery. J Oral Maxillofac Pathol 13:30, 2009
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19. Gardner DG: An evaluation of reported cases of median mandibular cysts. Oral Surg Oral Med Oral Pathol 65:208, 1988
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Figure Legends Fig. 1A A well-circumscribed unilocular radiolucency extended from the apices of mandibular right to left canines.
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Fig. 1B The cyst bulges from the facial aspect of the anterior mandible.
Fig. 1C The cyst was lined by thin epithelium with mural fibrosis and mild chronic inflammation. (H&E, х 100)
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Fig. 1D The lining epithelium was composed of 2 layers of low cuboidal-to-columnar cells with ciliated luminal cells; there is subepithelial hyalinization. (H&E, х 600)
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Fig. 1E Panoramic radiograph at 14 months post-operative, showing good bone fill. Fig. 2A A well-demarcated unilocular radiolucency with a sclerotic border in the right mandible associated with fixation screws.
Fig. 2B A well-defined radiolucency recurred at the right ramus area, measuring 1 x 2.5 cm that
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developed two years later.
Fig. 2C The cyst was lined by thin epithelium with mural fibrosis and mild chronic inflammation. Note osteoclasts at the base of the tissue. (H&E, х 200)
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Fig. 2D The cyst was lined by 2 cell layers of low cuboidal-to-columnar cells with luminal
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ciliated cells. (H&E, х 600)
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