Glandular odontogenic cyst

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Glandular odontogenic cyst
Other namesSialo-Odontogenic cyst
Relative incidence of odontogenic cysts.jpg
Relative incidence of odontogenic cysts. [1] Glandular odontogenic cyst is labeled at bottom.
Symptoms Jaw expansion, swelling, impairment to the tooth, root and cortical plate [2] [3]
CausesCellular mutation, cyst maturation at glandular, BCL-2 protein [2] [4]
Diagnostic method Biopsy, CT scans, Panoramic x-rays [5] [6]
Differential diagnosis Central mucoepidermoid carcinoma, odontogenic keratocyst [7] [6]
PreventionPost-surgery follow-ups are commonly proposed to prevent the chances of recurrence [6]
TreatmentEnucleation, curettage, marginal or partial resection, marsupialization [6]
Frequency0.12 to 0.13% of people [2]

A glandular odontogenic cyst (GOC) is a rare and usually benign odontogenic cyst developed at the odontogenic epithelium of the mandible or maxilla. [2] [8] [9] [10] Originally, the cyst was labeled as "sialo-odontogenic cyst" in 1987. [7] However, the World Health Organization (WHO) decided to adopt the medical expression "glandular odontogenic cyst". [9] Following the initial classification, only 60 medically documented cases were present in the population by 2003. [6] GOC was established as its own biological growth after differentiation from other jaw cysts such as the "central mucoepidermoid carcinoma (MEC)", a popular type of neoplasm at the salivary glands. [7] [11] GOC is usually misdiagnosed with other lesions developed at the glandular and salivary gland due to the shared clinical signs. [12] The presence of osteodentin supports the concept of an odontogenic pathway. [10] This odontogenic cyst is commonly described to be a slow and aggressive development. [13] The inclination of GOC to be large and multilocular is associated with a greater chance of remission. [10] [3] GOC is an infrequent manifestation with a 0.2% diagnosis in jaw lesion cases. [14] Reported cases show that GOC mainly impacts the mandible and male individuals. [3] The presentation of GOC at the maxilla has a very low rate of incidence. [8] The GOC development is more common in adults in their fifth and sixth decades. [1]

Contents

GOC has signs and symptoms of varying sensitivities, and dysfunction. [13] [14] In some cases, the GOC will present no classic abnormalities and remains undiagnosed until secondary complications arise. [13] The proliferation of GOC requires insight into the foundations of its unique histochemistry and biology. [7] The comparable characteristics of GOC with other jaw lesions require the close examination of its histology, morphology, and immunocytochemistry for a differential diagnosis. [10] Treatment modes of the GOC follow a case-by-case approach due to the variable nature of the cyst. [5] The selected treatment must be accompanied with an appropriate pre and post-operative plan. [5]

Signs and Symptoms

The appearance of a protrusive growth will be present at their mandible or maxilla. [2] 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. [2] [7] The area of impact may likely be at the anterior region of mandible as described in a significant number of reported cases. [8] At this region, GOC would eventually mediate expansion at the molars. [7] A painful and swollen sensation at the jaw region caused by GOC may be reported. [14] Detailing of a painless feeling or facial paraesthesia can be experienced. [7] [14] Alongside GOC, "root resorption, cortical bone thinning and perforation, and tooth displacement may occur". [3] Experience of swelling at the buccal and lingual zones can occur. [6] Usually, the smaller sized GOCs present no classical signs or symptoms to the case (i.e. "asymptomatic"). [4] GOC is filled with cystic a fluid that differs in viscosity and may appear as transparent, brownish-red, or creamy in colour. [3]

Causes

The molecular arrangement of BCL-2 protein, a potential cause to the development of the GOC. The protein can inhibit the process of apoptosis when at a high abundance. PDB 1ysw EBI.jpg
The molecular arrangement of BCL-2 protein, a potential cause to the development of the GOC. The protein can inhibit the process of apoptosis when at a high abundance.

The GOC can arise through a number of causes: [7]

The origin of the GOC can be understood through its biological and histochemistry foundations. [4] It has been suggested that GOC can be a result of a traumatic event. [12] The occurrence of GOC may be from a mutated cell from "the oral mucosa and the dental follicle" origin. [15] Another probable cause is from pre-existing cysts or cancerous constituents. [12] A potential biological origin of GOC is a cyst developed at a salivary gland or simple epithelium, which undergoes maturation at the glandular. [4] Another origin is a primordial cyst that infiltrates the glandular epithelial tissue through a highly organised cellular differentiation. [4] Pathologists discovered a BCL-2 protein, commonly present in neoplasms, to exist in the tissue layers of the GOC. [4] [15] The protein is capable of disrupting normal cell death function at the odontogenic region. [4] [15] 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 GOC. [7] It is confirmed that the gene had no assistance in triggering cystic advancement. [7]

Diagnosis

Radiology

The performance of radiographic imaging i.e. computed tomography, at the affected area is considered essential. [13] Radiographic imaging of the GOC can display a defined unilocular or multilocular appearance that may be "rounded or oval" shaped upon clinical observation. [5] [4] Scans may present a distribution of the GOC at the upper jaw as it presents a 71.8% prevalence in cases. [2] The margin surrounding the GOC is usually occupied with a scalloped definition. [2] A bilateral presentation of the GOC is possible but is not common at either the maxilla or mandible sites. [13] The GOC has an average size of 4.9 cm that can develop over the midline when positioned at the mandible or maxilla region. [3] [14] Analysis of scans allow for the differentiation of GOC from other parallel lesions, i.e. "ameloblastoma, odontogenic myxoma, or dentigerous cyst" in order to minimise the chance of a misdiagnosis. [5] These scans can display the severity of cortical plate, root, and tooth complications, which is observed to determine the necessary action for reconstruction. [5]

Histology

Histological features related to the GOC differ in each scenario; however, there is a general criterion to identify the cyst. [14] The GOC usually features a "stratified squamous epithelium" attached to connective tissue that is filled with active immune cells. [2] [7] The lining of the epithelium features a very small diameter that is usually non-keratinised. [8] [13] In contrast, the lining of the GOC has rather an inconsistent diameter. [2] The basal cells of the GOC usually has no association to a cancerous origin. [12] Tissue cells can be faced with an abnormal increase in the concentration of calcium, which can cause the region to calcify. [7] The transformation of the epithelium is associated with a focal luminal development. [2] Eosinophilic organelles such as columnar and cuboidal cells can be observed during microscopy. [11] Intra-epithelial crypts may be identified in the internal framework of the epithelium or at the external space where it presents itself as papillae protrusions. [8] [13] Mucin is observable after the application of "alcian blue dye" on the tissue specimen. [8] The histological observation of goblet cells is a common feature with the "odontogenic dentigerous cyst". [11] In some circumstances, the epithelium can have variable plaque structures that appear as swirls in the tissue layers. [8] Interestingly, histologists were able to identify hyaline bodies within the tissue framework of the GOC. [7] It is encouraged that the histological identification of at least seven of these biological characteristics is required to accurately distinguish the presence of the GOC. [11]

Intraepithelial Hemosiderin

Pathologists have identified hemosiderin pigments that are considered unique to the GOC. [12] The discovery of this pigment can be pivotal to the differentiation of the GOC from other lesions. [12] The staining at the epithelium is due to the haemorrhaging of the lining. [12] The cause of the haemorrhaging can be triggered by the type of treatment, cellular degradation, or structural deformation inflicted during GOC expansion. [12] Examination of the GOC tissue section indicated that red blood cells from the intraluminal space had combined with the extracellular constituents. [12] This process is carried out through transepithelial elimination. [12] This clinical procedure is beneficial to confirm the benign or malignant nature of the GOC. [12]

Immunocytochemistry

The examination of cytokeratin profiles is deemed useful when observing the differences between the GOC and the central MEC. [14] These two lesions show individualised expression for cytokeratin 18 and 19. [7] Past studies observed Ki-67, p53, and PCNA expression in common jaw cysts that shared similar characteristics. [7] There was a lack of p53 expression found in radicular cysts. [7] Similarly, Ki-67 was seen less in the central MEC compared to the other lesions, though this discovery is not essential to the process of differential diagnosis. [7] [14] Proliferating cell nuclear antigen readings were established to have no role in the differentiation process. [14] The TGF-beta marker is present in the GOC and can explain the limited concentration of normal functioning cells. [15]

MAML2 rearrangement

The observation of a MAML2 rearrangement is described as a procedure useful in the differential diagnosis of the GOC and its closely related lesion, the central MEC. [11] A second cystic development displayed the presence of CRTC3-MAML2 fusion after an in-vitro application. [11] The MAML2 rearrangement represents the developmental growth of the central MEC from the GOC. [11] The use of fusion-gene transcript may be helpful towards the differentiation of the GOC from the central MEC of the jaw and salivary glands. [11]

Treatment

Pre-treatment protocols

Panoramic radiography used to provide visualisations of the maxilla and mandible. X-rays will display the degree of impact on case, caused by the GOC. Panoramic Xray.jpg
Panoramic radiography used to provide visualisations of the maxilla and mandible. X-rays will display the degree of impact on case, caused by the GOC.

A computed tomography and panoramic x-ray must be undertaken in order to observe the severity of internal complications. [5] These scans allow for the observation of the GOC size, radiolucency, cortical bone, dentition, root, and vestibular zone. [5] 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. [13] The diagnosis of a smaller sized GOC is related to the attachment of only two teeth. [6] While, a greater sized GOC develops over two teeth. [6] Presentation of a greater sized lesion usually requires a biopsy for a differential diagnosis and a precise treatment plan. [6]

Treatment process

The unilocular and multilocular nature is imperative to the determination of treatment style. [6] Local anesthesia is regularly provided as the GOC is embedded within the tissue structure of the jaw and requires an invasive procedure for a safe and accurate extraction. [2] For unilocular GOCs with minimal tissue deterioration, "enucleation, curettage, and marsupialization" is a suitable treatment plan. [6] Notably, the performance of enucleation or curettage as the primary action is linked to an incomplete extraction of the GOC and is only recommended to the less invasive lesions. [6] Multilocular GOCs require a more invasive procedure such as "peripheral ostectomy, marginal resection, or partial jaw resection". [6] GOCs associated with a more severe structural damage are encouraged to undergo marsupialization as either an initial or supplementary surgery. [6] The frequency of reappearance is likely due to the lingering cystic tissue structures that remain after the performance of curettage. [13] The incorporation of a "dredging method i.e. repetition of enucleation and curettage" is also suggested until the remnants of the GOC diminishes for certain. [9] The treatment ensures scar tissue is removed to promote the successful reconstruction of osseous material for jaw preservation. [9] Alongside the main treatments, bone allograft application, cryosurgery, and apicoectomy are available but have not been consistently recommended. [9] [13] [5] Though Carnoy's solution, the chloroform-free version, is recommended with the treatment as it degenerates the majority of the damaged dental lamina. [13] The most effective type of treatment remains unknown due to the lack of detailed data from reported cases. [3]

Post-treatment protocols

Follow-up appointments are necessary after the removal of the GOC as there is a high chance of remission, which may be exacerbated in cases dealing with "cortical plate perforation". [13] [5] The GOC has a significant remission rate of 21 to 55% that can potentially develop during the period of 0.5 to 7 years post-surgery. [7] [6] Cases occupied with a lower risk lesion are expected to continue appointments with physicians for up to 3 years post-surgery. [6] A higher risk lesion is encouraged to consistently consult with physicians during a 7-year period after treatment. [13] Remission events require immediate attention and appropriate procedures such as enucleation or curettage. [6] In more damaging cases of remission, tissue resection, and marsupialization may have to be performed. [7]

Epidemiology

The clinical presentation of the GOC 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 individuals. [2] The GOC mainly affects older individuals in the population, especially those that are in their 40 to 60s. [8] However, the GOC can affect younger individuals i.e. 11, and more older individuals i.e. 82 in the population. [2] The age distribution starts at a much lower number for people living in Asia and Africa. [2] Those in their first 10 years of life have not been diagnosed with the GOC. [14] The GOC does present a tendency to proliferate in more males than females. [3] There is no definitive conclusion towards the relevance of gender and its influence on the rate of incidence. [7]

Related Research Articles

Cyst Closed sac growth on the body

A cyst is a closed sac, having a distinct envelope and division compared with the nearby tissue. Hence, it is a cluster of cells that have grouped together to form a sac ; however, the distinguishing aspect of a cyst is that the cells forming the "shell" of such a sac are distinctly abnormal when compared with all surrounding cells for that given location. A cyst may contain air, fluids, or semi-solid material. A collection of pus is called an abscess, not a cyst. Once formed, a cyst may resolve on its own. When a cyst fails to resolve, it may need to be removed surgically, but that would depend upon its type and location.

Ameloblastoma Medical condition

Ameloblastoma is a rare, benign or cancerous tumor of odontogenic epithelium much more commonly appearing in the lower jaw than the upper jaw. It was recognized in 1827 by Cusack. This type of odontogenic neoplasm was designated as an adamantinoma in 1885 by the French physician Louis-Charles Malassez. It was finally renamed to the modern name ameloblastoma in 1930 by Ivey and Churchill.

Cementoblastoma, or benign cementoblastoma, is a relatively rare benign neoplasm of the cementum of the teeth. It is derived from ectomesenchyme of odontogenic origin. Cementoblastomas represent less than 0.69–8% of all odontogemic tumors.

Mucoepidermoid carcinoma Medical condition

Mucoepidermoid carcinoma (MEC) is the most common type of minor salivary gland malignancy in adults. Mucoepidermoid carcinoma can also be found in other organs, such as bronchi, lacrimal sac, and thyroid gland.

Dentigerous cyst Medical condition

Dentigerous cyst, also known as follicular cyst is an epithelial-lined developmental cyst formed by accumulation of fluid between the reduced enamel epithelium and crown of an unerupted tooth. It is formed when there is an alteration in the reduced enamel epithelium and encloses the crown of an unerupted tooth at the cemento-enamel junction. Fluid is accumulated between reduced enamel epithelium and the crown of an unerupted tooth. Dentigerous cyst is the second most common form of benign developmental odontogenic cysts.

Central giant-cell granuloma Medical condition

Central giant-cell granuloma (CGCG) is a localised benign condition of the jaws. It is twice as common in females and is more likely to occur before age 30. Central giant-cell granulomas are more common in the anterior mandible, often crossing the midline and causing painless swellings.

Periapical cyst Medical condition

Commonly known as a dental cyst, the periapical cyst is the most common odontogenic cyst. It may develop rapidly from a periapical granuloma, as a consequence of untreated chronic periapical periodontitis.

Odontogenic keratocyst Medical condition

An odontogenic keratocyst is a rare and benign but locally aggressive developmental cyst. It most often affects the posterior mandible and most commonly presents in the third decade of life. Odontogenic keratocysts make up around 19% of jaw cysts.

“Lateral periodontal cysts (LPCs) are defined as non-keratinised and non-inflammatory developmental cysts located adjacent or lateral to the root of a vital tooth.” LPCs are a rare form of jaw cysts, with the same histopathological characteristics as gingival cysts of adults (GCA). Hence LPCs are regarded as the intraosseous form of the extraosseous GCA. They are commonly found along the lateral periodontium or within the bone between the roots of vital teeth, around mandibular canines and premolars. Standish and Shafer reported the first well-documented case of LPCs in 1958, followed by Holder and Kunkel in the same year although it was called a periodontal cyst. Since then, there has been more than 270 well-documented cases of LPCs in literature.

Calcifying odontogenic cyst Medical condition

Calcifying odotogenic cyst (COC) is a rare developmental lesion that comes from odontogenic epithelium. It is also known as a calcifying cystic odontogenic tumor, which is a proliferation of odontogenic epithelium and scattered nest of ghost cells and calcifications that may form the lining of a cyst, or present as a solid mass.

Squamous odontogenic tumors (SOTs) are very rare benign locally infiltrative odontogenic neoplasms of epithelial origin. Only some 50 cases have been documented. They occur mostly from 20-40 and are more common in males. Treatment is by simple enucleation and local curettage, and recurrence is rare.

An ameloblastic fibroma is a fibroma of the ameloblastic tissue, that is, an odontogenic tumor arising from the enamel organ or dental lamina. It may be either truly neoplastic or merely hamartomatous. In neoplastic cases, it may be labeled an ameloblastic fibrosarcoma in accord with the terminological distinction that reserves the word fibroma for benign tumors and assigns the word fibrosarcoma to malignant ones. It is more common in the first and second decades of life, when odontogenesis is ongoing, than in later decades. In 50% of cases an unerupted tooth is involved.

Odontoma Benign tumour of dental tissue

An odontoma, also known as an odontome, is a benign tumour linked to tooth development. Specifically, it is a dental hamartoma, meaning that it is composed of normal dental tissue that has grown in an irregular way. It includes both odontogenic hard and soft tissues. As with normal tooth development, odontomas stop growing once mature which makes them benign.

The odontogenic myxoma is an uncommon benign odontogenic tumor arising from embryonic connective tissue associated with tooth formation. As a myxoma, this tumor consists mainly of spindle shaped cells and scattered collagen fibers distributed through a loose, mucoid material.

The calcifying epithelial odontogenic tumor (CEOT), also known as a Pindborg tumor, is an odontogenic tumor first recognized by the Danish pathologist Jens Jørgen Pindborg in 1955. It was previously described as an adenoid adamantoblastoma, unusual ameloblastoma and a cystic odontoma. Like other odontogenic neoplasms, it is thought to arise from the epithelial element of the enamel origin. It is a typically benign and slow growing, but invasive neoplasm.

Odontogenic infection

An odontogenic infection is an infection that originates within a tooth or in the closely surrounding tissues. The term is derived from odonto- and -genic. The most common causes for odontogenic infection to be established are dental caries, deep fillings, failed root canal treatments, periodontal disease, and pericoronitis. Odontogenic infection starts as localised infection and may remain localised to the region where it started, or spread into adjacent or distant areas.

Odontogenic cyst are a group of jaw cysts that are formed from tissues involved in odontogenesis. Odontogenic cysts are closed sacs, and have a distinct membrane derived from rests of odontogenic epithelium. It may contain air, fluids, or semi-solid material. Intra-bony cysts are most common in the jaws, because the mandible and maxilla are the only bones with epithelial components. That odontogenic epithelium is critical in normal tooth development. However, epithelial rests may be the origin for the cyst lining later. Not all oral cysts are odontogenic cyst. For example, mucous cyst of the oral mucosa and nasolabial duct cyst are not of odontogenic origin.
In addition, there are several conditions with so-called (radiographic) 'pseudocystic appearance' in jaws; ranging from anatomic variants such as Stafne static bone cyst, to the aggressive aneurysmal bone cyst.

A cyst is a pathological epithelial lined cavity that fills with fluid or soft material and usually grows from internal pressure generated by fluid being drawn into the cavity from osmosis. The bones of the jaws, the mandible and maxilla, are the bones with the highest prevalence of cysts in the human body. This is due to the abundant amount of epithelial remnants that can be left in the bones of the jaws. The enamel of teeth is formed from ectoderm, and so remnants of epithelium can be left in the bone during odontogenesis. The bones of the jaws develop from embryologic processes which fuse together, and ectodermal tissue may be trapped along the lines of this fusion. This "resting" epithelium is usually dormant or undergoes atrophy, but, when stimulated, may form a cyst. The reasons why resting epithelium may proliferate and undergo cystic transformation are generally unknown, but inflammation is thought to be a major factor. The high prevalence of tooth impactions and dental infections that occur in the bones of the jaws is also significant to explain why cysts are more common at these sites.

A median mandibular cyst is a type of cyst that occurs in the midline of the mandible, thought to be created by proliferation and cystic degeneration of resting epithelial tissue that is left trapped within the substance of the bone during embryologic fusion of the two halves of the mandible, along the plane of fusion later termed the symphysis menti. A true median mandibular cyst would therefore be classified as a non-odontogenic, fissural cyst. The existence of this lesion as a unique clinical entity is controversial, and some reported cases may have represented misdiagnosed odontogenic cysts, which are by far the most common type of intrabony cyst occurring in the jaws. It has also been suggested that the mandible develops as a bilobed proliferation of mesenchyme connected with a central isthmus. Therefore, it is unlikely that epithelial tissue would become trapped as there is no ectoderm separating the lobes in the first instance.

Gingival cyst Medical condition

Gingival cyst, also known as Epstein's pearl, is a type of cysts of the jaws that originates from the dental lamina and is found in the mouth parts. It is a superficial cyst in the alveolar mucosa. It can be seen inside the mouth as small and whitish bulge. Depending on the ages in which they develop, the cysts are classified into gingival cyst of newborn and gingival cyst of adult. Structurally, the cyst is lined by thin epithelium and shows a lumen usually filled with desquamated keratin, occasionally containing inflammatory cells. The nodes are formed as a result of cystic degeneration of epithelial rests of the dental lamina.

References

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Bibliography

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