Primary Intraosseous Squamous Cell Carcinoma Involving the Jaw Bones: A Systematic Review and Update

Abstract

Primary intraosseous oral squamous cell carcinoma (PIOSCC) is a rare malignant neoplasm that affects the jaws. Despite its aggressive biological behavior, there are no studies that evaluated the clinicopathological features of this tumor and parameters associated with its prognosis. The objective of the present study was to conduct a systematic review of the available data on oral and maxillofacial PIOSCC in order to determine its clinicopathological characteristics and biological behavior. We conducted a systematic review in May 2020 in multiple databases using a specific search strategy. Cases diagnosed as PIOSCC in the oral cavity and maxillofacial complex that had sufficient histopathological data, absence of ulceration in the oral mucosa, a negative result for a distant primary tumor, and radiographic evidence of an osteolytic lesion that was entirely or mostly surrounded by the jaw bones were included. A total of 109 published articles were included in our systematic review, corresponding to 257 cases. PIOSCC showed a male predilection (69.3%) and a preference for the mandible (7:1), with the posterior region being the most commonly affected site. The mean age at diagnosis was 57.3 years. Cortical expansion, pain, and lip/facial paresthesia were the most common clinical features. Regarding histopathological features, most PIOSCC were well-differentiated and the solid subtype was the most common. Statistical analysis showed that PIOSCC located in the mandible (p = 0.03) and recurrence (p < 0.01) were significantly associated with a higher mortality rate. PIOSCC has a poor prognosis, with high rates of mortality.

Primary Intraosseous Carcinoma in the Pediatric Mandible

“…In this study, local recurrence occurred in three pediatric patients, including the present case, a rate of 30% although the total cases were only 10. In one report, a four-year-old female patient suffered recurrence ve months after excision and underwent additional radical excision [3]. However, 10 months later, recurrence recurred, the lesion was removed, and the area was irradiated.…”

“…PIOC is more common in adult males [3,[5][6][7][8][9][10][11]; however, seven of the total 10 pediatric cases were female. PIOC occurs more often in the mandible than maxilla [3,[5][6][7][8][9][10][11], seen in seven of the pediatric patients. The most common symptom was swelling, followed by pain [3,5,7].…”

“…PIOC occurs more often in the mandible than maxilla [3,[5][6][7][8][9][10][11], seen in seven of the pediatric patients. The most common symptom was swelling, followed by pain [3,5,7]. The symptoms of the pediatric patients were consistent.…”

“…However, in 2017, WHO reclassi ed it as a single entity after leaving out unsubstantiated references to histogenesis in 2017 [2]. Approximately 260 cases have been reported [3,4]. PIOC is more common in males and usually occurs in people 50 years and older [3,[5][6][7][8][9][10][11].…”

“…Approximately 260 cases have been reported [3,4]. PIOC is more common in males and usually occurs in people 50 years and older [3,[5][6][7][8][9][10][11]. PIOC has occurred extremely rarely in pediatric populations: prior to this case, only nine cases had been reported in the English literature [12][13][14][15][16][17][18][19][20].…”