Considering that 95% of POF cases occur in 10-19 years old pediatric patients, only few studies report of POF cases in children under 10 years [
24]; for this reason, we decided to carry out a brief review of the relevant literature about POF in pediatric patients, and the results are presented as follows. Tomáz Ferraresso [
25] described an interesting case of POF of the anterior mandible in a 6-year-old patient, affected by Ellis-van Creveld (EVC) syndrome, an autosomal recessive disease consisting in skeletal and ectodermal dysplasia, characterized by short stature, postaxial polydactyly, ectodermal dysplasia, and congenital heart disease. The patient showed a small nodular neoformation on the edentulous alveolar ridge in the mandible, without radiological investigations, treated by conventional surgical excision, hemostasis and suture, without recurrence. Botazzo Delbem [
15] described a case of the lateral maxillary region in a 5-year-old girl presenting as a non-ulcerated painless swelling, with radiological signs of osteolysis in correspondence of 1.2. The tumor was completely removed, without recurrence, by scalpel excision followed by curettage and periodontal scaling, diagnosed as “Peripheral Cemento-Ossifying Fibroma (PCOF)”. Lima et al. [
20] reported a rare case of generalized POF in a 4 years old child, presenting as extensive exophytic lesion covering several teeth and interfering with the chewing, treated by a partial debulking to improve the masticatory function, but characterized by more than 30 recurrences in 9 years. Moreover, two authors described POF in newborns of 2 and 5 months. Singh et al. [
26] presented a case report of POF manifesting as two distinct swellings localized in the mandibular edentulous ridge that did not allow a correct feeding. Interestingly, these two swellings began to grow after the exfoliations of two neonatal teeth. Both were removed by conventional surgery and showed no recurrence. Soares Tavares [
27] reported a POF in a 2-month-old infant, localized on the mandibular alveolar ridge during the eruption of 8.1, impairing the patient’s feeding. In contrast with the previous study, the authors decided to perform an excisional biopsy using laser, providing a valuable sample for the pathologists and a better post-operative course without bleeding, pain, and suture. No recurrence was shown in this case. Rallan et al. [
8] described a retro-incisive POF in a 12-year-old child. The clinical presentation was like our case but in an older patient. Only an intraoral Rx was performed, which showed no radiolucency and lamina dura integrity. The treatment consisted of a cold blade excision and bone debridement to exclude further recurrences. Choubey [
28] reported a POF localized in the mandibular canine region in a 13-year-old girl. She was treated with pre-operative scaling and root planing to reduce the local irritative factor as much as possible. Scalpel excision was carried out, and an aesthetic gingival recontouring was performed. Histopathologic examination reported “Peripheral Cementifying Fibroma (PCF)”, quite similar to POF, but basophilic concentric calcifications consisting of “cementicles” were encountered at the center of the lesion. Franco-Barrera [
14] conducted an interesting narrative review and reported a maxillary POF in an 11 years old girl, manifesting as an exophytic, painless and not ulcerated lesion of the maxillary right canine area, approximately measuring 4 × 3 × 1.5 cm, undergoing first to an incisional biopsy to exclude malignancy, then followed by scalpel excision and deep extension to the bone, with no recurrence. Yu [
11] reported a case, very similar to the one reported by Lima et al., of a diffuse POF in a 13 years old girl, localized on the maxilla, mandible, and palate, presenting with difficulties in speech, feeding, and oral hygiene; after a first debulking, the patient received a more aggressive excision in the following year that allowed complete healing without recurrence. Hasanuddin [
29] reported a maxillary POF in a 15 years old female that impeded lip competence because of its dimension of 2.4 x 2 cm, without radiolucency on the orthopantomography; the patient was subjected to an excisional biopsy with a scalpel following a bone debridement, and no recurrence was detected after 2 years. Nair and Tewari [
30,
31] described two similar cases of POF located in the anterior mandible, respectively in 3 and 2 months patients, with medical history of neonatal teeth; in both cases, lesions did not allow correct breastfeeding, so their surgical excision was necessary. All data mentioned above are summarized in
Table 1 (
Table 1).
Considering the most relevant literature on this topic, we found 12 case reports in the last 15 years, confirming a relatively rare occurrence in children or a low publication rate; 5 were between the range of 10 and 20 (42%), 3 between the 1-10 years (25%), 4 in newborn (< 1 year, 33%). Of particular interest are data about sex distribution: 7 cases (58%) occurred in females and 5 (42%) in males, confirming the female predilection of POF. Besides the age range, all cases show common histopathologic features: para-keratinized epithelium with a chronic inflammatory infiltrate, which delimitates a spindle fibroblast stroma, and calcification spots without areas of cellular atypia. Considering that POF occurrence in pediatric patients is very rare, the sample of this narrative review is too small to carry out any statistical analysis, but several considerations might be deduced. First of all, the distribution of sex and localization is consistent with previous studies, with a slight predominance in females [
14,
26]. In half of the cases, POF was localized in the mandible, then maxilla (33%) or generalized (17%), thus confirming that the epidemiological data of POF in infants are different from POF in adults, which predilects the maxillary region [
12]. It is relevant to highlight that 4/6 of the cases presented in the anterior mandibula (inferior incisor region) belongs to newborn with neonatal teeth history; however, such association is persistent but not statistically demonstrated [
30,
33]. The case we presented was in contrast with the literature we found, as the maxillary POF mainly occurred in the vestibular region of the maxilla and only one case in the upper retroincisal area; in fact, as reported by Rallan et al. [
8], the palatal occurrence further expands the differential diagnosis including reactive lesions as well neoplastic lesions both odontogenic and salivary. As often happens in such cases, in our patient the challenge was to predict the lesion’s nature without a pre-operative biopsy. In this, we were helped only by a CT scan that, although performed under general anesthesia, showed no cortical bone erosion and no tooth displacement, leading to exclude malignancy. In addition, CT was very useful in evaluating a little calcification spot in the lesion center, directing us toward an ossifying/cementifying lesion.
As found in literature, the pre-operative assessment of similar lesions, especially in children, lacks consensus. In the studies considered in this review, the most used radiogram was the periapical radiogram, followed by OPT. Both are first-level exams that must be executed if the patient is compliant, they are very useful for central lesions (lesions with exclusive bony involvement) but useless for peripheral lesions because of the overlapping of healthy hard tissues such as teeth and bone [
34]. The radiological exam suggestable for this type of lesion is the CT or CT Cone Beam [
35]. In previous studies, CT was executed only once, precisely by Franco-Barrera [
14], as a pre-operative assessment of a large POF removed under general anesthesia.