Enucleation of a multilocular odontogenic keratocyst using sagittal osteotomy: A case report

Abstract

Odontogenic keratocysts are significant cysts of the jaw that are characterized by aggressive behavior and a high tendency to recur. The treatments of choice may be radical or conservative, and in the literature a debate is still open about the gold standard in the treatment of OKCs.This report describes a wide multilocular OKC extended in the right mandibular ramus and illustrates atypical behavior of a cystic lobe after marsupialization, not found to our knowledge in the previous literature. Moreover, an orthognathic procedure, consisting of sagittal osteotomy was chosen for the excision of the cyst to preserve as much bone as possible and the integrity of the inferior alveolar nerve.

Highlights

  • Marsupialization of a parakeratinized keratocyst is a useful part of the treatment in order to reduce the volume.

  • Marsupialization can switch the parakeratinized keratocyst in orthokeratinized one.

  • The case reports an atypical behavior of a lobe in a marsupialized keratocyst, that starts to increase instead of diminuishing.

  • An orthognathic procedure, consisting of a sagittal osteotomy was chosen for the excision of the cyst.

  • The sagittal osteotomy consents to preserve as much bone as possible and the integrity of the inferior alveolar nerve.

Introduction

Odontogenic keratocysts are significant cysts of the jaw that are characterized by aggressive behavior and a high tendency to recur . Odontogenic keratocysts are histologically divided into two sub-types: those that are orthokeratinized (OOCs), considered the less aggressive variant, and those that are parakeratinized (OKCs), which are more aggressive and correlated with a higher incidence of recurrences (compared to OOCs) .

Diagnosis is often delayed because of silent symptomatology, so OKCs are frequently discovered as high-volume lesions involving different anatomic structures; consequently, a three-dimensional presurgical study of the lesion is mandatory .

The treatments of choice may be radical or conservative, and in the literature a debate is still open about the gold standard in the treatment of OKCs. Radical approaches include resection (marginal or en bloc). Conservative treatments include simple cyst excision or marsupialization/decompression, which can be followed by a second surgery. Although conservative approaches allow for minimally invasive surgery with a low rate of post-surgical discomfort, radical treatments are correlated with a lower rate of recurrence. Nevertheless, this kind of treatment often results in aesthetic and functional deficits [ ].

This report describes a wide multilocular OKC extended in the right mandibular ramus and illustrates atypical behavior of a cystic lobe after marsupialization, not found to our knowledge in the previous literature. In addition, the surgical approach was chosen to preserve as much bone as possible and the integrity of the inferior alveolar nerve (IAN).

Presentation of the case

A 27-year-old man was referred to our operative division by his private dentist because of an osteolytic lesion found incidentally during routine panoramic radiography (OPT). The patient was in good health and didn’t report any symptoms. The panoramic radiograph showed a wide multilocular radiolucent lesion extended in the left ramus, and the body of the mandible was associated with an impacted 4.8.

Cone beam computed tomography (CBCT) was requested, which revealed a wide multilocular lesion, composed of four lobes. Axial scans revealed the expansion and thinning of the cortical bone on the lingual side, which in some areas appeared discontinuous. In addition, this osteolytic lesion compressed and displaced the canal of the IAN ( Fig. 1 A).

Fig. 1
Radiographic images of the multilocular odontogenic keratocyst (OKC): A) CBCT scan at the radiographic diagnosis: the lesion appears as multilocular, includes part of the impacted third molar and involves the IAN; B) CBCT scan 8 months after marsupialization (first surgical approach) with signs of bone deposition and progressive reduction in lesion volume; C) Radiographic control (CBCT) 20 months after marsupialization showing as the lobe of the OKC located on the posterior border of the mandible was increasing instead of diminishing; D) CBCT scan taken 12 months from the second surgical treatment (radical excision) with a good bone healing and incorporation of the osteosynthesis plate. CBCT= Cone Beam Computed Tomography, IAN = Inferior Alveolar Nerve, OKC = Odontogenic Keratocyst.

A hypothetical diagnosis of OKC or ameloblastoma was formulated based on radiographic features, so a first surgery was planned. Hence, the patient underwent marsupialization of the lesion and extraction of the 4.8. In particular, a lateral corticotomy was performed as described by Alling and Alling . Moreover, a portion of the cystic wall was removed and sent for histological analysis. The bone portion was then replaced and fixed with an osteosynthesis plate and screws. The flap was sutured, leaving a large opening in the oral cavity. The lesion was filled with a gauze soaked with gentamycin ( Fig. 2 ). The pathological result revealed that the lesion was an OKC.

Aug 8, 2020 | Posted by in Oral and Maxillofacial Surgery | Comments Off on Enucleation of a multilocular odontogenic keratocyst using sagittal osteotomy: A case report
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