Cartilage in the walls of odontogenic keratocyst

VICENTE-BARRERO M, BÁEZ-MARRERO O, ALFONSO-MARTÍN JL, KNEZEVIC M, BÁEZ-ACOSTA B, CAMACHO-GARCÍA MC, MONTENEGRO-DÁMASO T. CARTILAGE IN THE WALLS OF ODONTOGENIC KERATOCYST. MED ORAL 2004;9:268-70.

SUMMARY

There are seven published cases in world literature on cartilage in the walls of odontogenic keratocysts. Herein is presented one further case with keratin inclusions in the cystic wall, which also bears a cartilaginous component. X-rays, clinical images and pathohistological images are included.

Key words: Cartilage, keratocyst, odontogenic cyst.

INTRODUCTION

Odontogenic keratocyst (OKC) is a developmental maxillary cyst with either an ortho or a parakerati-nized stratified, squamous epithelial wall. The term OKC was first coined by Philipsen in 1956 (1), who described a massive, aggressive lesion with a higher recurrence rate than other maxillary cysts. It probably stems from the dental lamina or its residues, though some authors believe it is derived from reduced enamel ephitelium.

OKC represents approximately 4% of all odontogenic cysts. Gorlin-Goltz Syndrome is associated with 5% of these cases. OKC is statistically somewhat more frequent in women, with peak incidence somewhere between the second and fourth decades of life. It is more frequent in the Anglo/white race. The most common location is in the rear sectors, especially in the third molar area, with double the frequency in the mandible (versus the maxilla).

Clinical presentation of OKC can appear to be a dentigerous (associated with the crown of an impacted tooth), a primordial cyst (in the area corresponding to a tooth that hasn't developed), a residual cyst (in the area where a tooth previously existed), a periodontal lateral cyst, or in an area that can be confused with a globulomaxillary or nasopalatine cyst. Most OKC bear no clinical symptoms. Occasionally there may be pain, tumor, localized pain or even intraoral fistulization.

Radiologic findings are similar to other odontogenic cysts. For this reason diagnosis is usually made with histopathology studies, which reveal the regular keratinized squamous epithelium, with ortho or parakeratinization in different parts of the cyst. There are few published cases where OKC presents cartilage in its lining/wall.

CLINICAL CASE

A 41-year-old woman came to our office for a painless tumor that had been growing slowly over the past 6 months around the upper maxilar, between the 14 and 15 premolars, which remained intact with their roots slightly out of place. There was no pain, trophic alterations, or sign of inflammation. The exam revealed an entire swelling in the vestibular cortex (fig. 1 & 2).

The histopathologic study returned a diagnosis of OKC with isolated corneal microinclusions in the cystic wall, with an isolated cartilaginous component. There was no ameloblastic cellular proliferation (fig. 3 & 4).

DISCUSSION

To date there are seven published cases of cartilage in the OKC wall (2-4), this being the eighth. The number of cases is too small to establish a pattern separate from typical OKC. But we should take note of some differentiating considerations:

Though OKC can occur at any age, more than half of the cases happen between 10 and 40 years of age, while 7 of the 8 cases in this series happened above the age of 40.

The most frequent location is in the rear maxillary sector, with greater incidence in the mandible. The OKC with cartilaginous inclusion in the lining/wall is more frequent (7 cases) in the front section&emdash;more precisely, in the area of premolar to molar (four cases in the upper maxillary and three in the lower).

Typical OKC is slightly more frequent in women, while in this series 4 were men and 4 were women.

Kratochvil and Brannon (3) consider various explanations for the presence of cartilage in the OKC wall: a) coexistence of a chondroma, b) persistence of displaced parts of the embryonic Meckel's cartilage, c) metaplastic change in the connective fibrous tissue in response to a chronic irritation, and d) possible induction in the cystic wall by the epithelial linning (such is the case presented here).

The fact that in most cases the chondroid tissue is in close contact with the epithelial linning in many areas suggests that the etiopathogeny would be more an induction of the lesions, rather than a fusion of two differents lesions.

REFERENCES

1. Philipsen HP. Om keratocysts (Kolesteatomer) I Kaeberne, Tandlaegebladet 1956;60:963-80        [ Links ]

2. Fornatora ML, Reich RF, Chotkowski G, Freedman PD. Odontogenic keratocyst with mural cartilaginous metaplasia: A case report and a review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2001;92:430-4        [ Links ]

3. Kratochvil FJ, Brannon RB. Cartilage in the walls of odontogenic keratocysts. J Oral Pathol Med 1993;22:282-5        [ Links ]

4. Mosqueda-Taylor A, de la Piedra-Garza JM, Troncozo-Vazquez F. Odontogenic keratocyst with condroid fibrous wall. A case report. Int J Oral Maxillofac Surg 1998;27:58-60        [ Links ]