Squamous cell carcinoma arising from a keratocystic odontogenic tumor: a case report | Journal of Medical Case Reports


The pathogenesis of PIOSCC arising from KCOT had been debatable and several mechanisms were suggested by different pathologists [1]. Browne and Gouch highlighted a few important landmarks in the pathogenic sequence such as epithelial hyperplasia, epithelial dysplasia of cyst epithelia, and finally keratin metaplasia [5]. In consideration of the above landmarks in pathogenesis, van der Wal et al. mentioned that the presence of keratinization in the cyst lining results in the development of SCC in odontogenic keratocysts [6]. Gardner et al. and Yu et al. viewed this from a different perspective and suggested that the formation of reactive oxygen metabolites in long-standing inflammation resulting in damage to the dioxy ribonucleic acid (DNA), protein, and cell membranes eventually leads to compensatory proliferative responses of neoplastic cells against the normal apoptotic mechanism [7, 8].


et al.



] in 2011 reviewed the literature on PIOSCC in odontogenic cysts and Table 


elaborates a comparison of their data with the present case.

Table 1

Comparison between Bodner and colleagues’ [2] analysis of 116 reported cases of squamous cell carcinoma arising in an odontogenic cyst and the present case


Mean age

60.2 years

50 years

Close to mean age

Commonest age of occurrence

6–8th decades





Male predilection










Type of cyst

Radicular cyst



KCOT malignant transformation rate is rare

Dentigerous cyst




Lateral periodontal





Carcinoma in situ


Moderately differentiated SCC


Well-differentiated SCC


Moderately differentiated SCC


Poorly differentiated SCC


Verrucous carcinoma


Spindle cell carcinoma


Signs and symptoms



Mass paresthesia present in the lower lip





Painful mass


Jaw expansion


Sensory disturbance


Treatment modality

Surgery alone


Surgery with neck dissection


Surgery with radiotherapy


Surgery, radiotherapy with chemotherapy


Surgery with chemotherapy


Radiotherapy alone


Neck dissection


Overall survival

2 years


1.5 years

Patient still under review

5 years


In their study Bodner et al. highlighted the presence of only 16 cases of PIOSCC arising from KCOT from 1938 to 2010 [2]. Further, there were two other cases reported in the literature by Lee et al. [9] in 2011 and Tamgadge et al. [1] in 2013. A review of the literature regarding PIOSCC showed the mandible (79%) as the predominant site of occurrence as compared with the maxilla (21%). Similar data were recorded in the present case as the lesion appeared in the mandible. In accordance with the literature, PIOSCC arising from a KCOT shows a wide age range, with a mean age of 57 years, and males outnumbered females with male–female ratio of 2:1. Similar findings were observed in studies conducted by Aboul-hosn Centenero et al., Mosqueda-Taylor et al., and Scheer et al.; however, our patient was a 50-year-old woman.

Even though SCC arising from KCOT is a rare phenomenon, the present case is comparable with most of the aspects cited in the literature with the exception of our case being female. Also, PIOSCC arising from KCOT is considered to be very rare whereas a radicular cyst is more common.

Some reported cases of PIOSCC demonstrated symptoms of pain, progressive swelling, and paresthesia at the initial stages of the disease. Paresthesia was associated with the current case and the pain that appeared in the latter stage was the trigger for the patient to present to the healthcare facility. Such avoidance leads to delayed presentation and poor prognosis of oral malignant lesions in the South Asian population.

Data on the prognosis of PIOSCC arising from KCOT is scarce as there are only a few reported cases. However, in the reported cases, the 2-year survival rate of patients has been 53%. The prognosis was reported to be poor when there was evidence of nodal metastasis of the neck [10]. Surgical management was recommended in most reports with adjuvant radiotherapy [11]. Radiotherapy was not given to this current case because she was tumor free and adequate resection margins were confirmed in the excision biopsy. Furthermore, no neck nodal metastases were observed histopathologically. Neck metastasis is considered to be an important prognostic indicator and according to Bodner et al. only six cases of PIOSCC arising from odontogenic cysts have been reported [2].

Debates exist about the features of the grading of a tumor among different pathologists regarding grading of tumors because it is subjective. Therefore, the data to consider prognosis for each category of grading is unreliable.

According to the current case, we emphasize the importance of careful investigation of swellings present in the mandible. Clinicians as well as patients should be aware and detect these changes to avoid being clinically negligent. Careful histopathological examination of an odontogenic cyst is also recommended as, even though it is rare, there is a possibility for its epithelial lining to become malignant.


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