not everything in the maxillary sinus

17/5/2015 NotEverythingintheMaxillarySinusIsSinusitis:ACaseofaDentigerousCyst

http://pediatrics.aappublications.org/content/121/1/e203.long 1/5

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Pediatricspediatrics.aappublications.org

Pediatrics Vol. 121 No. 1 January 1, 2008 pp. e203 -e207 (doi: 10.1542/peds.2006-1940)

EXPERIENCE&REASON

NotEverythingintheMaxillarySinusIsSinusitis:ACaseofaDentigerousCystRichard Haber, MA, MD, FRCPS

Author Affiliations

ABSTRACT

A 4-year-old girl presented to a medical clinic with a painless right facialswelling. The treating physician ordered a radiograph of the sinuses andreceived a report of maxillary sinusitis. After appropriate antibiotic treatment,the facial swelling increased, and the mother took the child to her communitypediatrician. After a period of observation and additional imaging, thediagnosis of dentigerous cyst was made. After appropriate surgicalintervention, the cyst was removed, and over the ensuing 6 weeks the facialswelling gradually diminished. Dentigerous cysts, although uncommon, need tobe considered in the differential diagnosis of children with painless facialswelling.

Key Words: ambulatory care community pediatrics diagnostic errorssinus disease

This case report is one of the very few in the pediatric medical literature thatremind us that not everything in the maxillary sinus is acute sinusitis. Becausedentigerous cysts are rarely reported in the pediatric medical literature, thepurpose of this report is to alert pediatricians to their possibility in childrenwith facial swelling.

CASEREPORT

A 4-year-old girl presented to a medical clinic because of a slight right facialswelling. Although there was no fever or sinus tenderness, the attendingphysician ordered sinus films and the radiologist reported acute bilateralmaxillary and right ethmoid sinusitis the facial bones are radiologicallynormal. A 10-day course of cefprozil (250 mg twice daily) was prescribed. Twomonths later, the child presented to her pediatrician because of a sore rightear, and a diagnosis of otitis media was made; the child was treated with a 3-day course of azithromycin. At the visit, the mother expressed concern thatthere was still some facial swelling overlying the right maxillary sinus. Physicalexamination revealed a slight right facial swelling, which was nontender andfirm, with no warmth, fluctuation, or discoloration of the overlying skin. Thechild was afebrile, and the rest of her physical examination was within normallimits. The physician decided to observe her. Six weeks later, the mother calledbecause she was alarmed that the facial swelling was increasing. Onexamination, her physician noted that the right facial swelling was indeedincreasing, and there was some watery discharge from the right eye with noconjunctival injection or redness (Fig 1). Again, the swelling was found to bepainless, with no warmth, fluctuation, or discoloration overlying it. The swellingfelt firm and bony. The child underwent additional imaging (Figs 24). Theradiologic diagnosis was 2 large dentigerous cysts each associated with dentalstructures. A surgical procedure was performed (Caldwell-Luc excision of cystand right functional endoscopic sinus surgery, as well as maxillary antrostomy).A cyst-like mass measuring 6.0 6.0 0.5 cm was excised, along with 2aberrant tooth-like structures. Multiple sections revealed a cyst lined by anonkeratinized layer of stratified squamous epithelium (Fig 5). Also presentwere small tooth-like structures consisting of enamel, dentin, and dentalfollicle (Fig 6). Therefore, the final diagnosis was a dentigerous cyst arisingfrom an odontoma. The child did well postoperatively, and 6 weeks after theprocedure, the facial swelling had decreased considerably. A repeat computed



tomography scan has shown no recurrence of the dentigerous cyst. Eighteenmonths later, there was no recurrence, and there has been complete resolutionof the facial swelling.

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FIGURE 1

Swellingoverlyingthe rightmaxillarysinus.


FIGURE 2

Two large expansile cystic lesions areseen within the right facial regionoccluding the ostiomeatal complex,obliterating the maxillary antrum,elevating the orbital floor, anddepressing the hard palate (whitearrows). Note the thin bony platerepresenting floor of maxillary sinusbetween the cystic expansile lesionand the maxillary antrum of 1 of thecysts (black arrow).


FIGURE 3

Computed tomography scan showingabnormal tooth (black arrow) withattenuated bony septum and normaltooth on the left (white arrow).




FIGURE 4

MRI showing teeth indentigerous cysts(white arrows).


FIGURE 5

Medium-power view of cyst liningconsisting of nonkeratinizedstratified squamous epithelium(hematoxylin and eosin stain; originalmagnification: 200).


FIGURE 6

Low-power view of a tooth-likestructure consisting of enamel anddentin (hematoxylin and eosin stain;original magnification: 50).

DISCUSSION

Dentigerous cysts are not usually aggressive. In this child, however, the lesionwas quite aggressive, elevating the orbital floor and depressing the hard palate(Fig 2). This raises the issue of more serious diagnoses, includingameloblastoma and odontogenic keratocyst.

Ameloblastoma is rare in the pediatric age range, and in a classic review of1036 cases of ameloblastoma, the average age was 38.9 years. Ameloblastomaoccurs rarely in the maxilla (7% of pediatric cases), with most occurring in themandible either at the angle of the mandible or the symphysis. The majority ofthese cases present radiologically as a dentigerous cyst, and it is necessary toexamine the lining of the cyst to make the correct diagnosis. This is important,because the treatment is different, if controversial, in children, with some oralsurgeons recommending radical resection to prevent recurrence.1 A unicysticameloblastoma may present, like in our subject, but the histologic features didnot support the diagnosis. Ameloblasts present were associated with enamelformation (Fig 5).

The odontogenic keratocyst is a result of a cystic change within the enamelorgan before calcification, and expansion of this lesion results from rapidproliferation of the squamous epithelial lining and not from the accumulation



of fluid as in a dentigerous cyst. This is an aggressive lesion, and recurrence iscommon.

In this patient, histologic examination confirmed the diagnosis of an odontoma,which is a radiolucent lesion consisting of multiple tiny, malformed, tooth-likestructures in various stages of development. Usually this is a slow-growinglesion, but in our patient, it was associated with a follicular or dentigerous cyst,which arose from 1 of these tooth-like structures in the odontoma (Fig 6).

Dentigerous cysts in a child are extremely uncommon. A dentigerous cyst is abenign expansive lesion derived from hydrostatic expansion of a dental follicleand surrounds the crown of an unerupted tooth.2 In our patient, the toothremained close to the alveolar bone with the cyst extending upward. Oneusually expects to see the tooth at the superior part of the cyst as the cystdevelops between the crown and the epithelium pushing the tooth upward (Fig3). The typical dentigerous cyst arises from the enamel organ afteramelogenesis is finished and is a result of the accumulation of fluid betweenthe crown and the enamel organ pushing the crown away from the alveolarbone. Dentigerous cysts are associated with unerupted teeth and are usuallyfound by the dentist during routine dental radiology.

Dentigerous cysts accounted for 14% to 20% of all jaw cysts referred to oralsurgeons in 1 series; of the 40 referred, 14 cysts were in children 12 years ofage. Most are found in the mandible.3 Another series from Brazil reviewing2356 oral biopsies of patients 14 years of age over a 15-year period revealedthat 6.5% had dentigerous cysts.4 Dentigerous cysts, especially bilateral ormultiple, have been reported in patients with basal cell nevus syndrome,mucopolysaccharidosis, and cleidocranial dysplasia; they have also beenassociated with cyclosporine use and calcium channel blockers.3 Treatment isalways surgical, requiring excision and pathologic examination to rule outother more aggressive lesions.5,6

CONCLUSIONS

Our patient presented with a painless right facial swelling initially felt to be anacute sinusitis. Over a period of several weeks, the expanding lesion in theright maxillary sinus, with additional imaging and histologic examination,revealed itself to be a dentigerous or follicular cyst associated with anodontoma. Although dentigerous cysts are more often found in the mandible,they may occur, as in our patient, in the maxilla. Histologic examination of thecyst lining is essential to differentiate this relatively benign lesion from a moreaggressive lesion, such as an ameloblastoma or an odontogenic keratocyst,both of which require aggressive resection to prevent recurrence. Most reportsof dentigerous cysts are found in the surgical literature (dental, oral/facial, orotolaryngology), with no reports in the general pediatric literature. Our patientis a reminder to general pediatricians to include the dentigerous cyst in thedifferential diagnosis of painless facial swelling.

ACKNOWLEDGMENTS

I acknowledge the assistance of Dr Peter Chauvin, associate professor anddirector of the Division of Oral Diagnostic Sciences, Faculty of Dentistry, McGillUniversity, for providing the histology slides and their descriptive text.

FOOTNOTES

Accepted June 24, 2007.

The author has indicated he has no financial relationships relevant to thisarticle to disclose.

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Copyright 2008 by the American Academy of Pediatrics

not everything in the maxillary sinus

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