Conservative Algorithm for the Treatment of Locally Aggressive Tumors: A Retrospective Study

Abstract

Background

Aggressive benign odontogenic neoplasms are a group of lesions that have the potential to grow to an enormous size resulting in bony deformities, locally aggressive behavior and have high recurrence rate. Ameloblastomas, keratocystic odontogenic tumors, etc., are included under this group of lesions. Treatment of these lesions is challenging owing to their particular characteristics including high recurrence rate, locally aggressive behavior, etc.

Materials and Methods

A retrospective study was conducted in 40 patients who had undergone enucleation and open packing as treatment for benign aggressive neoplasms in the department of oral and maxillofacial surgery.

Results

In the study, all the cases were followed up for a minimum of 5 years clinically and radiographically. Satisfactory bone healing was noted with no evidence of recurrence in all cases.

Conclusion

Enucleation followed by open packing with iodoform is an excellent conservative modality with proven minimal rate of recurrences in 5-year period.

Introduction

Odontogenic keratocyst and ameloblastoma are categorized into a group of lesions called aggressive benign odontogenic neoplasms (ABONS) owing to their potential to grow to an enormous size resulting in bony deformities, locally aggressive behavior and their high recurrence rate [1]. They at the same time remain a mystery and challenge to the surgeons because of its peculiar clinical, radiographic and histopathological characteristics with benign nature and aggressive potential [2].

Various conservative modalities of treatment are mentioned in the literature which include marsupialization, enucleation alone, enucleation with open packing, enucleation and peripheral ostectomy with open packing, enucleation with chemical cauterization and aggressive procedures like resection [3].

The aim of the study was to evaluate the efficacy of a conservative surgical procedure (enucleation followed by iodoform gauze dressing) in the treatment of aggressive benign odontogenic neoplasms.

Materials and Methods

A retrospective study was carried out over a period of ten years from 2011 to 2021 in forty patients who reported to the Department of Oral and Maxillofacial Surgery with locally aggressive tumors (Unicystic Ameloblastoma, Keratocystic odontogenic tumor) in their mandible. All the patients were of Asian background and were treated by the same surgical team.

Patients with uncontrolled systemic conditions, pregnant women, elderly (> 60 years), physically and mentally challenged and those who are not willing for a long-term post-therapy maintenance program and follow-up were excluded from the study.

Clinical and radiographic evaluation (orthopantomogram and CBCT) were carried out in all patients (Table 1) and the diagnosis was confirmed with the help of an incisional biopsy (Figures 1, 2). All the patients were managed under local anesthesia. Crevicular incision was used to expose the tumor. Following which, the tumor was enucleated along with surgical removal of impacted teeth and open packing with iodoform was carried out. Patient was kept on oral antibiotics and analgesics for 5 days postoperatively. The pack was changed once in a week for 3 months. Every time the size of the pack was reduced. Clinical and radiographic (OPG) follow-up was done for 5 years to evaluate the amount of bone formation and to rule out any evidence of recurrence.

Table 1.

Clinicopathological and radiographic findings

Sex	Male	10
	Female	30
Mean age	45 years
Site	Maxilla
	Anterior maxilla	0
	Posterior maxilla	0
	Mandible
	Anterior mandible	0
	Posterior mandible	40
	Anterior and posterior mandible	0
Clinical presentation	Asymptomatic	26
	Swelling	14
	Swelling and pain	0
Radiographic features	Unilocular	32
	Multilocular	8
	Cortical bone
	Expanded	10
	Perforated	3
	Preserved	27
Histopathological diagnosis	Ameloblastoma	22
	Keratocystic odontogenic tumor	18

Fig. 1.

Preoperative photograph

Fig. 2.

Preoperative intraoral

Result

The aggressive benign odontogenic neoplasms (ABONS) show a female predilection with a mean age of occurrence of 45 years. The youngest patient was of age 8 years and the oldest were of age 65 years. The tumor is commonly detected in mandible in the premolar-molar region. The largest lesion was of size 2.5 × 2 cm and the smallest was of size 1.5 × 1 cm in maximum dimension. All the cases were treated with enucleation and iodoform packing which is changed every week for a period of 3 months. All the 40 patients had sufficient bone healing at the end of 3 months period. Also, there was no evidence of any recurrence clinically and radiographically over a period of 5-year follow-up. In younger age group, this could favor the normal eruption of permanent teeth which has not erupted or displaced due to the tumor. None of the patients had any signs of infections during the follow-up period (Fig. 3, 4, 5).

Fig. 3.

Pre-op OPG

Fig. 4.

Post-op OPG

Fig. 5.

Post-op OPG 5 year follow-up

Discussion

Aggressive benign odontogenic neoplasms or tumors of jaws include Ameloblastoma, keratocystic odontogenic tumor, pindborgs tumor and odontogenic myxoma [4, 5]. Johnson et al. reported the incidence of odontogenic tumors in the literature and they found that ameloblastomas were the most prevalent odontogenic tumors (37.9%), followed by KCOT (36.6%) [6].

OKC’s are common in second and third decade of live with male predilection [7]. They commonly occur in posterior mandibular region [7]. In the year 2005, World health organization (WHO) included this pathology in the group of odontogenic neoplasms with the name keratocystic odontogenic tumor [KCOT] because of its aggressive behavior, high mitotic activity and its association with mutation or inactivation of tumor suppressor gene [PTCH] [8, 9]. They show high tendency to recur (5–62%) following surgical treatment and the recurrence commonly occurs within the first 5 years of treatment [10, 11].

Ameloblastomas are common in the fourth decade of life with predilection to posterior mandible [12]. They are slow growing odontogenic tumors, locally aggressive and accounts for approximately 1% of all oral tumors [13].

As per literature, adjacent tooth displacement was found in 33.7% of OKCs and 55.8% of ameloblastomas. Root resorption was more common in ameloblastomas (66.7%) than in OKCs (7%). OKCs showed smooth border (60%) and unilocular shape (82%), while most ameloblastomas showed scalloped border (77.2%) and multilocular shape (68.3%). Association with impacted tooth was found in 47% of OKCs and 18.8% of ameloblastomas [14]. In our study, 80% of cases were unilocular and 20% were multilocular. 60% of OKC’s and 30% of ameloblastoma’s were associated with impacted teeth in our study.

There is still considerable controversy in the management of these lesions owing to its locally aggressive behavior and high recurrence rate [1]. Different modalities of treatment were described in the literature which include enucleation, marsupialization, adjunctive procedure, resection and a combination of these procedures [15]. Psychosocial reasoning, fear of disfigurement and social well-being often influences a surgeons decision to perform conservative treatment particularly on children; however, serious consequences may occur, which includes recurrence or malignant transformation of the tumor [16]. Radical resection with 1 cm tumor-free margins are advised by most of the investigators; however, this modality of treatment is associated with serious morbidities which include jaw deformity and dysfunction particularly in young age group which adversely affects their physical and mental well-being [17]. The best treatment modality is the one with lowest possible risk of recurrence, the least morbidity, while completely eradicating the lesion [18].

The study by Lau and Samman on Ameloblastoma reported that recurrence rates of 3.6% is there after resection, 30.5% following enucleation alone, 16% on enucleation with chemical cauterization, and 18% if enucleation is done after marsupialization [19]. The histopathological report forms an important part of diagnosis and treatment planning, and studies show high recurrence rate for unicystic ameloblastoma especially in intramural and plexiform variety of unicystic ameloblastoma [20]. However in our study, none of the cases had recurrence in a 5-year follow-up period.

Iodoform is a yellow crystalline, organo-iodine compound used as an antiseptic component for certain medications since 1822 [21]. They protect tissues of the wound both mechanically and by maintaining asepsis, also destroy toxic substances formed by micro-organisms, and thus help to promote faster healing [21].

In our study, enucleation of tumor is done followed by open packing with iodoform. The primary advantage of enucleation is that the complete removal of the cyst can be ensured which can be submitted for thorough histopathologic examination [22]. Iodoform reduces wound fluid by fibrinolytic activity, helps in reduction of pain by covering the denuded bone surface and on topical application acts as an antimicrobial agent [1]. The advantage of this procedure is that they are less traumatic for the patient, least medication and hospitalization expenses, and avoidance of the need for reconstruction through grafts that require general anesthesia and prolonged hospitalization [18].

Conclusion

Management of aggressive benign odontogenic neoplasms is still controversial. There is a general consensus stating that radical resection can completely eliminate the tumor with minimal or no recurrence rate. However owing to the significant deformity and the psychosocial consequences, a conservative modality of treatment is preferred. Enucleation followed by open packing is an excellent conservative modality with proven minimal rate of recurrences in 5-year period.

Funding

Not applicable.

Declarations

Conflict of interest

The authors declare that they have no conflict of interest.

Ethics Approval

Ethical approval taken.

Consent to Participate

Consent to participation obtained from all patients.