Glandular odontogenic cyst

Glandular odontogenic cyst
Other names	Sialo-Odontogenic cyst
Relative incidence of odontogenic cysts. Glandular odontogenic cyst is labeled at bottom.
Symptoms	Expansion of the jaw, swelling, impairment to tooth, root and cortical plate
Causes	Cellular mutation, cyst maturation at glandular, BCL-2 protein
Differential diagnosis	Central mucoepidermoid carcinoma, odontogenic keratocyst
Treatment	Enucleation, curettage, marginal or partial resection, marsupialization
Frequency	0.12 to 0.13% of people

A glandular odontogenic cyst is a rare and benign odontogenic cyst developed at the odontogenic epithelium of the mandible or maxilla. Originally, the cyst was labeled as 'sialo-odontogenic cyst' in 1987. However, the World Health Organisation (WHO) decided to adopt the medical expression 'glandular odontogenic cyst'. Following the initial classification, only 60 medically documented cases were present in the population by 2003. It was established as its own biological growth after differentiation from other jaw cysts such as the ‘central mucoepidermoid carcinoma’, a popular type of neoplasm at the salivary glands. The cyst is usually misdiagnosed with other lesions developed at the glandular and salivary gland due to the shared clinical signs. This odontogenic cyst is commonly described to be a slow and aggressive development. The inclination of the cyst to be large and multilocular is associated with a greater chance of remission. The cyst is an infrequent manifestation with a 0.2% diagnosis in jaw lesion cases. Reported cases show that the cyst mainly impacts the mandible and male individuals. The presentation of the cyst to be at the maxilla has a very low rate of incidence. It is more common in adults in their fifth and sixth decades.

The cyst has signs and symptoms of varying sensitivities, and dysfunction. In some cases, the cyst will present no common abnormalities and remains undiagnosed until secondary complications arise. The proliferation of cyst requires insight into its unique histochemistry. The comparable characteristics of the cyst with other jaw lesions require physicians to closely examine the histology, morphology, and immunocytochemistry for a differential diagnosis. Treatment modes of the cyst follow a case-by-case approach due to its variable nature. The selected treatment must have an appropriate pre and post-operative plan.

Signs and Symptoms

The appearance of a protrusive growth will be present at their mandible or maxilla. The expansive nature of this cyst may destruct the quality of symmetry at the facial region and would be a clear physical sign of abnormality. The area of impact will likely be at the anterior region of mandible due its presence in a significant amount reported cases. A painful and swollen sensation at the jaw region caused by the cystic growth has been described. Detailing of a painless feeling or facial paraesthesia can be possible. Alongside the growth, "root resorption, cortical bone thinning and perforation, and tooth displacement may occur". Experience of swelling at the buccal and lingual zones can occur. Usually, the smaller sized cysts present no signs or symptoms to the case. The cyst can begin its proliferation at the anterior region of the teeth and eventually mediate expansion at the molars. The lesion is filled with cystic a fluid that differs in viscosity and may appear as transparent, brownish-red, or creamy in colour.

Causes

The molecular arrangement of BCL-2 protein, a potential cause to the development of the glandular odontogenic cyst. The protein can inhibit the process of apoptosis when at a high abundance.

The origin of this cyst can be understood through its biological and histopathlogical foundations. It has been suggested that the occurrence of the cyst is a result of a traumatic event. The occurrence of this cyst may be from a mutated cell from "the oral mucosa and the dental follicle" origin. Another probable cause is from pre-existing cysts or cancerous constituents. A potential biological origin is a cyst developed at a salivary gland or simple epithelium, which then matures at the tissue of the glandular. Another origin is when a primordial cyst infiltrates the glandular epithelial tissue through a highly organised cellular differentiation. Also, it can arise from a singular cystic space induced by a cancerous mass. Pathologists discovered a BCL-2 protein that is commonly present in neoplasms, to exist in the tissue layers of the cyst. The protein is capable of disrupting normal cell death function at the odontogenic region. The analysis of PTCH, a gene that specialises in neoplasm inhibition, was carried out to determine if any existing mutations played a role in the initiation of the glandular odontogenic cyst. It is confirmed that the gene has no assistance in triggering cystic advancement.

Diagnosis

Radiology

The performance of computed tomography at the area of impact is essential. Radiographic imaging of the cyst can display a defined unilocular or multilocular appearance that may be shaped "rounded or oval" upon clinical observation. The cyst is found to frequently reside at the anterior of the jaw. Scans present a distribution of the cyst commonly at the upper jaw, where there is a 71.8% prevalence in cases. The margin surrounding the cyst is usually occupied with a scalloped definition. A bilateral presentation of the cyst is possible but is not common at the maxilla and mandible sites. Analysis of scans allow physicians to differentiate the cyst and avoid misdiagnosis from other parallel lesions, i.e., ‘lateral periodontal cyst’ and ‘ameloblastoma’. Scans can display the severity of cortical plate, root, and tooth impairment, which is analysed to determine the necessary action for reconstruction. The cyst has an average size of 4.9 cm that can develop over the midline from the mandible or maxilla region.

Histology

Histological features related to the cyst differ in each scenario; however, there is a general criterion to identify this entity. The cyst can feature as a stratified squamous epithelium attached to fibrous connective tissue that is infiltrated by active immune cells. The lining of the epithelium is occupied with a very small diameter and is usually non-keratinised. Whereas, the lining of the lesion has an inconsistent length of diameter. The basal cells is associated with no origin to a carcinoma. Tissue cells may be faced with an abnormal increase in the concentration of calcium, which can cause the region to calcify. The transformation of the epithelium is associated with a focal luminal development. Eosinophilic organelles such as columnar and cuboidal cells can be observable during microscopy. Intra-epithelial crypts may be apparent in the internal framework of the epithelium or at the external space where it presents itself as papillae protrusions. Mucin is observable after the application of alcian blue dye on the tissue specimen. The histological observation of goblet cells is also featured in the odontogenic dentigerous cyst. In some circumstances, the epithelium can have variable plaque structures that appear as swirls in the tissue layers. Interestingly, histologists were able to identify hyaline bodies within the tissue framework of the cyst. It is encouraged that the histological identification of at least seven of these biological characteristics is required to accurately distinguish the cyst.

Intraepithelial Hemosiderin

Pathologists have identified hemosiderin pigments that are unique to the glandular odontogenic cyst. The discovery of this pigment can be pivotal to the differentiation of the cyst from other lesions. The staining in the epithelium is due to the haemorrhaging at the lining. The cause of the haemorrhaging can be triggered by the type of treatment, cellular degradation, or structural deformation induced by the cystic growth. Examination of the cyst tissue section indicated that red blood cells from the intraluminal space was combined with extracellular constituents. This process is carried out through transepithelial elimination. This observational aspect is beneficial to determine the benign or malignant nature of the potential cancerous cyst.

Immunocytochemistry

The examination of cytokeratin profiles is deemed useful when observing the differences between the glandular odontogenic cyst and central mucoepidermoid carcinoma. These two lesions show individualised expression for cytokeratin 18 and 19. The presence of osteodentin is supports the concept of an odontogenic pathway. Past studies observed Ki-67, p53, and PCNA expression in common jaw cysts that share similar characteristics. There is a lack of p53 expression found in radicular cysts. Similarly, Ki-67 is seen less in the central mucoepidermoid carcinoma compared to the other lesions, though this discovery is not essential to the process of differential diagnosis. Proliferating cell nuclear antigen readings are established to have no role in the differentiation process. The TGF-beta marker is present in the cyst and can explain the limited concentration of normal functioning cells.

MAML2 rearrangement

The observation of MAML2 rearrangement is described as a component useful in the differential diagnosis of the glandular odontogenic cyst and the closely related lesion, central mucoepidermoid carcinoma. A second cystic development displayed the presence of CRTC3-MAML2 fusion after the in-vitro application. The MAML2 rearrangement represents the developmental growth of central mucoepidermoid carcinoma from the glandular odontogenic cyst. The use of fusion-gene transcript may be helpful towards the differentiation of the cyst from central mucoepidermoid carcinoma of the jaw and salivary glands.

Treatment

Pre-treatment protocols

Panoramic radiography used to provide visualisations of the maxilla and mandible. X-rays will display the degree of impact caused by the cyst.

A computed tomography and panoramic x-ray must be undertaken in order to observe the severity of internal complications. From these scans, physicians will observe the size, radiolucency, cortical bone, dentition, root, and vestibular zone. In some cases, the dentition may be embedded into the cavity walls of the lesion, depending on the position of expansion at the odontogenic tissue. The diagnosis of a smaller sized cyst is related to the attachment of only two teeth. An expansive cyst develops over two teeth. Presentation of a greater sized lesion requires a biopsy to determine the differential diagnosis for a precise treatment plan.

Treatment process

The unilocular and multilocular nature is imperative to the determination of treatment style. Local anesthesia is regularly provided as the cyst is embedded within the tissue structure of the jaw and requires an invasive procedure for a safe and precise extraction. For unilocular cysts with minimal tissue deterioration, "enucleation, curettage, and marsupialization" are a suitable treatment plan. Notably, the individual performance of enucleation or curettage as the primary action is linked to an incomplete extraction of the cyst and is only recommended to the less invasive lesions. Multilocular cysts require a more invasive procedure "(i.e. peripheral ostectomy, marginal resection, or partial jaw resection)". Cysts that are associated with more severe structural damage are endorsed to undergo marsupialization as either an initial or supplementary surgery. The frequency of reappearance is likely associated with the lingering cystic tissue structures that remain after the performance of curettage. The incorporation of a "dredging method (i.e. repetition of enucleation and curettage)" is also applied for those at a high-risk, until the remnants of the lesion diminishes for certain. The treatment ensures scar tissue is removed to promote the proper reconstruction of osseous material for jaw preservation. Alongside the main treatments, bone allograft application, cryosurgery, Carnoy's solution, and an apicoectomy are available but have not been consistently recommended.The chloroform-free version of this fixative solution is recommended with the treatment type as it degenerates the remaining damaged dental lamina. The most effective type of treatment remains unknown due to the lack of data from reported cases.

Post-treatment protocols

Strict follow-up appointments are necessary after the removal of the cyst due to its high chances of remission, which is exacerbated in cases dealing with cortical plate perforation. The glandular odontogenic cyst has a significant remission rate of 21 to 55% that can potentially develop during the period of 0.5 to 7 years post-surgery. Cases occupied with a lower risk lesion are expected to follow-up with physicians for up to three years post-surgery, similar to the overlapping lesion odontogenic keratocyst. A higher-risk situation is encouraged to consistently consult with physicians during a seven-year period after treatment. Remission events need immediate attention with appropriate procedures such as enucleation or curettage. In more harmful cases of remission, tissue resection, and marsupialization are carried out.

Epidemiology

The clinical presentation of this cyst is very low in the population as noted by the 0.12 to 0.13% occurrence rate, extrapolated from a sample size of the 181 people. The cyst mainly affects older individuals in the population, especially those that are in their 40 to 60s. However, this cyst can impact younger individuals (i.e. 11), and more older individuals (i.e. 82) in the population. The age distribution starts at a much lower number for people living in Asia and Africa. Notably, those in their first 10 years of life have not been diagnosed with this cyst. The cyst does present a tendency to proliferate in more males than females. There is no definitive conclusion towards the relevance of gender and its influence on the rate of incidence.

Bibliography

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• Motooka, Naomi; Ohba, Seigo; Uehara, Masataka; Fujita, Syuichi; Asahina, Izumi (1 January 2015). "A case of glandular odontogenic cyst in the mandible treated with the dredging method". Odontology. 103 (1): 112–115. doi:10.1007/s10266-013-0143-0. PMID 24374982. S2CID 21059170.
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Further reading

• Kahn MA (2001). Basic Oral and Maxillofacial Pathology. Vol. 1.