Dentigerous cyst of mandible presenting as sepsis Dustin W. Anderson MD, David Evans MD, RDMS, RDCS, FACEP PII: DOI: Reference:

S0735-6757(14)00412-4 doi: 10.1016/j.ajem.2014.05.044 YAJEM 54327

To appear in:

American Journal of Emergency Medicine

Received date: Accepted date:

6 May 2014 20 May 2014

Please cite this article as: Anderson Dustin W., Evans David, Dentigerous cyst of mandible presenting as sepsis, American Journal of Emergency Medicine (2014), doi: 10.1016/j.ajem.2014.05.044

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Dentigerous cyst of mandible presenting as sepsis

Dustin W. Anderson, MD, David Evans, MD, RDMS, RDCS, FACEP

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From the Department of Emergency Medicine, Virginia Commonwealth University Health System, Richmond, VA

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Address for Reprints: David Evans, M.D. FACEP RDMS RDCS Department of Emergency Medicine Virginia Commonwealth School of Medicine Main Hospital 2nd floor, room 606, Suite 600 1250 East Marshal St PO BOX 980401 Richmond, VA 23298-0401 (O) 804-263-4570 (F) 804-828-4999 Key Words: Airway, Imaging, Otolaryngology

ACCEPTED MANUSCRIPT Dentigerous cyst of mandible presenting as sepsis Abstract

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Dentigerous cysts are odontogenic cysts that develop by accumulation of fluid between reduced enamel epithelium and a crown of an unerupted tooth. Dentigerous cysts typically are slow growing and incidental findings on radiographic images.1 These cysts are usually small but when they become large they will cause a pathologic fracture. Occasionally they can become painful when infected which will cause swelling and erythema.1 We present a rare case of a dentigerous cyst that presented as sepsis. Dentigerous cysts are the most common type of

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noninflammatory odontogenic cyst.2 The atypical acute presentation and extent of this patient’s soft tissue manifestations resulting in tracheal midline shift and sepsis makes this a rare case for presentation.

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Case Report

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An otherwise healthy 36-year-old African American male presented to emergency department (ED) for facial swelling that he described as sudden onset when he awoke that morning (Fig 1). The patient did endorse recent illness two days prior with fevers, chills, nausea, and vomiting. He denied any recent trauma, dental issues, or recent procedures. Notably, the patient described the swelling as painless. On exam the patient was ill appearing. We was noted to be febrile and tachycardic in triage. He had extensive swelling to the right face and neck, which was warm to touch, indurated, and not painful. The patient was noted to have severe trismus of approximately

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0.5 cm. Initial labs were notable for a WBC count of 23.2 x 103/μL.

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In the ED, there was concern due to trismus and extent of soft tissue swelling of bilateral submental region extending over midline as well as right neck for impending airway compromise. He was given 900 mg clindamycin and dexamethasone 10 mg intravenously and emergent consult to otolaryngology was made to evaluate the patient’s airway. A flexible fiberoptic scope exam was performed bedside in the ED with exam showing crowding of pharyngeal walls inferiorly on the right with mild edema of the epiglottis. Mild waterbag edema of the right arytenoid was visualized with a patent supraglottis. A bedside ultrasound was performed to evaluate for abscess formation and extent of soft tissue involvement. Bedside ultrasound confirmed extensive soft tissue swelling extending from the right temporal mandibular joint, right submandibular, right neck, and bilateral submental region crossing the midline. There were no fluid collections on ultrasound appreciated. Subsequently a head computed tomography study of head and neck was ordered (Figure 2.) Results were concerning for the amount of soft tissue swelling and fat stranding in the masticator space which was exerting mass effect on the oropharynx with leftward deviation. The trachea had midline shift without evidence of collapse. Other findings showed a hypodense, nonenhancing lesion of the right mandibular ramus (2.4 x 1.4 x 2.2 cm) consistent with a right mandibular dentigerous cyst inferior of the right third unerupted third molar. The surrounding bone and cortex represented a pathologic, non-displaced fracture. Oral maxillofacial surgery was consulted and the patient was admitted for airway monitoring. On admission patient was given intravenous clindamycin 900

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mg every 8 hours and dexamethasone 10 mg every 6 hours. Otolaryngology performed bedside flexible fiberoptic scope evaluation every four hours with noticeable decrease in edema of epiglottis and supraglottic areas after administration of dexamethasone. Patient was discharged on hospital day three of admission with transition to oral clindamycin 300 mg every 6 hours for 14 days. Patient returned two weeks later to have biopsy of the dentigerous cyst performed. Final pathology results indicated a squamous epithelium-lined cyst that was inflamed.

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Dentigerous cysts are most common in second and third decade.3 They can occur over an age range from 15 years to 65 years.4 They are two times more common in males and ten times more common to affect the mandible.5 This case supports the demographics that are seen by other authors. The study by Mourseshed found that 1.44 percent of impacted teeth can transform to a dentigerous cyst.6 This was illustrated by our case. A retrospective study performed by Smith, et al, evaluated 327 charts with an admitting diagnosis of head and neck infection and dentigerous cysts that were treated at tertiary care hospital between 1975 and 2004. Their results showed that only 7 patients had dentigerous cysts that required admission. Six of the seven patients had previous infections at the same location with the most common site of infection being the ramus of the mandible involving an unerupted third molar7.

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This case presentation illustrates that a dentigerous cyst, which is typically slow growing, can expand rapidly to cause a pathological fracture. Once this cyst has expanded outside the borders of the mandible, rapid swelling, airway compromise, and secondary infection leading to sepsis can be a complication. Pain is typically seen on presentation however, in this case it was not and our patient had a prodrome of fevers, chills, nausea with vomiting prior to acute phase of inflammation. This is the first case of sepsis as a complication of dentigerous cyst with airway compromise that we are aware of.

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Figure 1. Patient on presentation

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References

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Figure 2. Computer Tomography scan of the mandible

1. Paul R, Paul G, Prasad RK, Singh S, Agarwal N, Sinha A, Appearance can be deceptive: Dentigerous cyst crossing the midline. Natl J Maxillofac Surg. 2013; 4:100-103. [PubMed: 3800369] 2. Scholl RJ, Kellett HM, Neumann DP, Lurie AG. Cysts and cystic lesions of the mandible: Clinical and radiologic-histopathologic review. Radiographics. 1999;19:1107-24. [PubMed: 10489168] 3. Shear M, Speight P. Cysts of the Oral and Maxillofacial Regions. 4th ed. Blackwell Publishing; 2007. Dentigerous cyst; p. 59. 4. Koseoglu BG, Atalay B, Erdem MA. Odontogenic cysts: A clinical study of 90 cases. J Oral

ACCEPTED MANUSCRIPT Science. 2004; 46:253-7. [PubMed: 15901071]

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5. Singh S, Singh M, Chhabra N, Nagar Y. Dentigerous cyst: A case report. J Indian Pedod Prev Dent. 2001; 19:123-6.

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6. Mourshed F. A roentgenographic study of dentigerous cysts. II. Role of roentgenograms in detecting dentigerous cyst in the early stages. Oral Surg Oral Med Oral Pathol. 1964;18:54-61. [PubMed: 14179875] 7. Smith JL 2nd, Kellman RM. Dentigerous Cysts Presenting as Head and Neck Infections.

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Otolaryngology – Head and Neck Surgery. 2005; 133:715-717.

Dentigerous cyst of mandible presenting as sepsis.

Dentigerous cysts are odontogenic cysts that develop by accumulation of fluid between reduced enamel epithelium and a crown of an unerupted tooth. Den...
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