Abstract
Background
The postoperative recurrence of mandibular odontogenic keratocyst (OKC) accompanied by secondary actinomycotic osteomyelitis is extremely rare and presents significant challenges in clinical diagnosis and treatment. This case study combines imaging, pathological data, and relevant literature to offer guidance for clinical management.
Patient information
A 21-years-old female patient underwent surgical treatment for a polycystic OKC of the mandible 4 years ago. She was hospitalized again due to sharp subgingival sensation under the labial gums in the mandibular anterior tooth area. Pathological examination revealed OKC and chronic actinomycetic osteomyelitis. Mandibular mass resection and curettage of chronic osteomyelitis were performed. Clindamycin was used continuously for 4 weeks after operation, and the prognosis was good.
Conclusion
OKC frequently recurs after surgery and can damage the oral mucosal barrier, potentially leading to actinomycotic osteomyelitis. Pathology is crucial for accurate diagnosis. Surgical resection of secondary OKC, and postoperative administration of standard antibiotics are effective treatment methods.
Keywords: Mandibular, Odontogenic keratocystic OKC, Actinomycetic osteomyelitis, Cone-beam computed tomography CBCT, Pathological
Introduction
First reported by Philipsen in 1956, OKC has attracted wide attention due to its significant differences in invasive growth pattern, clinical manifestations and postoperative high recurrence rate compared with other types of jaw cystic lesions [1].
The recurrence rate reported in previous studies was 17%~54.5% [2]. Risk factors associated with keratocyst recurrence included younger patient age, the presence of multilocular lesions, larger lesion size, and a longer anteroposterior dimension [3]– [4]. OKC is prone to secondary infection when it penetrates into soft tissue, but no secondary osteomyelitis has been reported so far.
Mandibular osteomyelitis is mostly odontogenic suppurative osteomyelitis in which Staphylococcus aureus and Staphylococcus epidermidis are the mainly pathogenic bacteria [4]. While osteomyelitis caused by actinomyces infection is very rare. The mandibular angle and mandibular rami are the most common sites [5, 6]. The main clinical manifestations are hard soft tissue mass, multiple sinuses and varying degrees of opening limitation, and the disease is prone to recurrence [7, 8].
At present, the postoperative recurrence of mandibular OKC and secondary actinomycetic osteomyelitis have not been reported. The clinical symptoms and imaging characteristics of this disease is lack. The case of recurrent mandibular OKC and secondary actinomycetic osteomyelitis treated in Jinan Stomatological Hospital were analyzed to provide help for clinical diagnosis and treatment.
Case data
The patient was a 21-year-old female with no family genetic disorders. She was first treated of soft tissue swelling in the area of mandibular anterior tooth 5 years ago (March 2018). Preoperative Cone-beam computed tomography (CBCT) image showed a multi-cystic lesions.(Figs. 1 and 2) The larger one was from the right mandibular lateral incisor to the left mandibular cuspid teeth area accompanied by labial bone plate absorption and soft tissue mass formation.The smaller one is between the left mandibular first and second premolars with the lingual bone destruction as shown in Fig. 1. Pathological examination results: “mandibular odontogenic keratosis”, as shown in Fig. 3.
Fig. 1.
CBCT horizontal position: From the right mandibular lateral incisor to the left mandibular cupisp teeth, multi-saccular low-density focus with uneven density; labial bone plate absorption; bony septum in the capsule; thickening of surrounding soft tissue were observed. A cystic lesion approximately 1 cm in diameter was observed on the lingual side, located between the left mandibular first and second premolars
Fig. 2.
CBCT sagittal position: labial bone plate absorption, thickening of labial soft tissue and air accumulation with cystic lesions in the mandibular anterior teeth
Fig. 3.
Pathological section: capsular wall of covered squamous epithelial fibrous tissue with hyperkeratosis and parakeratinised, infiltration of inflammatory cells in the interstitium. It was consistent with keratosis (HE×100)
Regular reexamination after operation showed no discomfort in the operative area. In July 2020, the one-year re-examination of the oral panoramic film showed that the dentition was generally good and low density could be seen in the operative area, which was considered to be related to postoperative bone tissue defects, as shown in Figure 4. Postoperative two-years oral panoramic film was taken in July 2021, and there was no significant change compared with the oral panoramic film in 2020, as shown in Figure 5.
Fig. 4.
Panoramic oral film: The apical area of the central and lateral incisors of the normal mandible shows small flake and low density; the alveolar bone of the upper part of the root of the first and second premolars of the left mandible shows low density, with clear boundaries and clear peridental membranes surrounding the adjacent teeth
Fig. 5.
Oral panorama: Compared with Figure 4, there was no significant change in the operative area image
In September 2023, the patient sought treatment at the hospital for a sharp and hard sensation in the labial subgingival area of the lower front teeth, along with red and swollen gums, a small amount of yellowish discharge, and limited effectiveness of oral antibiotics.
Physical examination
The patient developed normally and had mild pain in the anterior dental area. Intra-oral examination: surgical scar was observed in the labial mucosa of the mandibular anterior tooth area; the left mandibular incisor was distally inclined; the left mandibular cuspid teeth was mesially inclined; the labial gum was locally raised; and the surrounding mucosa was mildly red and swollen with a hard texture.
CBCT showed a circular low-density shadow on the left and right sides of the mandibular anterior tooth region. Partial labial bone loss was observed from the left mandibular central incisor to cuspid teeth, and several small flake “sequestrum” shadows of different sizes were shown in the corresponding parts. The soft tissue of the lip was thickened with uneven density (Figs. 6, 7 and 8). The initial diagnosis was a recurrence of odontogenic keratosis and chronic osteomyelitis of the left mandible.
Fig. 6.
CBCT horizontal position: The apical area of the right mandibular central incisor showed a low-density cystic focus with a size of about 5mm×5mm×0.6mm and a clear boundary. Partial bone labial loss was observed from the left mandibular central incisor to cuspid teeth. The corresponding parts showed several small flake "sequestrum" shadows of different sizes. The labial soft tissue thickened and had uneven density. The apical area from the left mandibular cuspid teeth to the first molar showed an irregular sac-like low-density focus; the size was about 0.7mm×0.7mm×0.8mm; the boundary was clear; and the lingual bone plate was partially absorbed
Fig. 7.
CBCT sagittal position: Partial labial bone loss in the left mandibular central incisor, lateral incisor and cuspid teeth, with several small dissociate sequestrum shadows of varying sizes
Fig. 8.
Apical film: The root canal of the right mandibular central incisor showed high-density shadow filling, and the apical area showed circular cystic lesions
Postoperative pathological diagnosis: (1) OKC (Figs. 9 and 10); (2) Chronic osteomyelitis (Fig. 11); (3) Actinomycosis (Fig. 12).
Fig. 9.
Cysts in the apical region of the right mandibular central incisor and the cysts in the first premolar region of the left mandibular cusp revealed that fibrous capsular wall tissue was lined with lamellar squamous epithelium and a little keratinides accompanied by parakeratinised were found, which were consistent with odontogenic keratocyst (HE×100)
Fig. 10.
Cysts in the apical region of the right mandibular central incisor and the cysts in the first premolar region of the left mandibular cusp revealed that fibrous capsular wall tissue was lined with lamellar squamous epithelium and a little keratinides accompanied by parakeratinised were found, which were consistent with odontogenic keratocyst (HE×100)
Fig. 11.
Pathological sections showed that there was a large number of acute and chronic inflammatory cell infiltration in fibrous connective tissue, inflammatory granulation tissue hyperplasia, necrotic tissue and chronic inflammatory cell infiltration between bone trabeculae, which was consistent with chronic osteomyelitis (HE×100)
Fig. 12.
Pathological section showed that there was a large number of inflammatory cells infiltrating the interstitium, and actinomycetes and mycelia were visible, which was consistent with actinomycosis (HE×100)
Treatment
One week before surgery, the root canal treatment of the right mandibular central incisor was taken (Figure 8), and clindamycin hydrochloride capsule was taken orally for anti-infection.
Following the management of the mandibular anterior tooth, a mandibular mass resection and curettage of chronic osteomyelitis were performed. Postoperatively, intravenous clindamycin was administered for seven days, and the incision healed well. Upon discharge, oral therapy was continued for three weeks.
Follow-up and outcomes
The prognosis was good. OKC showed no signs of recurrence, osteomyelitis was cured (see Figs. 13 and 14),The patients were satisfied with the therapeutic effect.
Fig. 13.
CBCT horizontal and sagittal position: The OKC lesions and "sequestrum" were completely removed, some bone defects were found in the corresponding parts, and the surrounding bone hyperplasia and sclerosis were observed
Fig. 14.
CBCT horizontal and sagittal position: The OKC lesions and "sequestrum" were completely removed, some bone defects were found in the corresponding parts, and the surrounding bone hyperplasia and sclerosis were observed
Discussion
The imaging features and chinical manifestations of OKC
The WHO Classification of Head and Neck Tumors, Fourth Edition, published in 2017 [9], renamed “OKC”and classified it as an odontogenic cyst based on its clinical, pathological, and biological characteristics.
The disease most commonly occurs in the posterior dental region of the lower jaw or the mandibular angle [10]. The typical imaging manifestations are cystic diseases growing along the long axis of the jaw with a significant lingual dilation [5].
In this case, the initial site of the disease was in the mandibular anterior region, which was not typically high occurrences of OKC. The lesions presented multi-cystic manifestations with buccal expansion and bone plate absorption rather than the typical manifestations of growth along the long axis of the jaw. Meanwhile, the lesions were accompanied by the formation of soft tissue masses. These features were easily lead to misdiagnosed as ameloblastoma. Therefore, Pathological diagnosis provides crucial evidence for diagnosing complex jaw cystic lesions.
In this case, OKC was the parakeratinized type, located around the roots of several mandibular anterior teeth. It has a relatively complex structure. During surgery, efforts were made to preserve as much of the alveolar bone as possible, although this poses a risk for postoperative recurrence.
The pathogenesis and manifestations of actinomycetic osteomyelitis
Whether primary or recurrent OKC, cysts are susceptible to secondary infection if they penetrate the bone plate, invade soft tissue, or communicate with the external environment. This infection typically presents as soft tissue swelling and pus discharge while osteomyelitis has not been reported. In this study, multiple dissociate sequestrum in the alveolar bone of the mandibular anterior dental region were found using CBCT, which was considered to be the main cause of chronic osteomyelitis. Combined with pathological examination, actinomycetes clumps were found to support the diagnosis of actinomyctic osteomyelitis, which was extremely rare.
Actinomycosis of the jaws is a relatively uncommon infection. The principle cause of cervicofacial actinomycosis is Actinomyces israelii [11]. This endogenous parasite commonly resides in dental caries, periodontal pockets, and tonsil crypts in healthy individuals. When the normal oral mucosal barrier is damaged by infection, trauma or surgery, and the body’s resistance is weakened, actinomyces can invade adjacent tissues and spread to bone tissues, leading toosteomyelitis. Chronic osteomyelitis is typically characterized by granulomatous and suppurative lesions due to the slow reproduction and weak virulence of actinomyces [12]. Lesions are identified by the presence of light yellow actinomycelium or yellow sulfur particles within granulation tissue or pus. Imaging reveals bone destruction and clear areas of varying sizes. Some patients may also exhibit the formation of sequestra, periosteal hyperplasia, and maxillary swelling [5, 13].
This case primarily presented with bone destruction and detached sequestrum, but no periosteal hyperplasia was observed. This could be due to the periosteal of the affected area being destroyed by surgery or OKC. Considering the patient’s history, it was concluded that preoperative mucosal tissue damage and reduced defense function, resulting from either the OKC surgery or its recurrence, were the primary causes of actinomycotic osteomyelitis in this case.
Chose appropriate surgical and drug project to treatment OKC and actinomycetic osteomyelitis
Gonçalves et al. [14] conducted a systematic evaluation of six electronic databases and literature sources, identifying that the most common treatment for OKC was enucleation (including curettage or simple enucleation), followed by assisted technology enucleation and bagging.The average recurrence rate of OKC was 16.2%, and the highest recurrence rate of OKC was 43.2% after curettage or enucleation.
Crescente et al. [15] suggested curettage as the preferred treatment for OKC with initial onset, limited scope and no risk factors for recurrence. Petchiammal and Al-Moraissi et al. [16–18] suggested curettage combined with adjuvant therapy as a routine treatment for lesions with risk factors for recurrence or recurrent lesions. Guided tissue regeneration is also an acceptable treatment for the management of OKC after complete enucleation [19]. Maxillectomy was suitable for radical treatment of large or recurrent lesions. It should be noted that patients with infection need to control infection before surgery.
For the treatment of chronic osteomyelitis, Florian, Simre et al. suggested that sensitive antibiotics should be used as soon as possible, and the basic treatment principles were thorough debridement, elimination of ineffective cavity and wound protection [20]– [21]. Urish and Karan et al. [22]– [23] believed that the use of sensitive antibiotics for at least 4 weeks, otherwise it would easily lead to the recurrence of osteomyelitis. While Rod-Fleury et al. [24] pointed out that there was no direct correlation between the recurrence of osteomyelitis and the time of antibiotic use, and whether the dewound was thorough was the key factor determining the recurrence of chronic osteomyelitis. The standard of debridement is to remove infected or necrotic bone tissue until healthy bone tissue, and non-bleeding bone tissue needs to be completely removed.
In this case, the patient being a school student had high standards for aesthetics and occlusal function. To meet the patient’s needs and achieve optimal therapeutic results, the initial procedure involved fenestration and drainage, followed by curettage. This approach maintained a good aesthetic outcome, with no recurrence observed four years post-surgery. Following recurrence, the patient declined radical treatment involving extensive jaw resection and the removal of adjacent teeth. According to the current requirements, OKC curettage was performed, followed by cauterization of the residual bone cavity using an electroknife to deactivate remaining OKC cells as effectively as possible. For osteomyelitis, sensitive antibiotics were used both before and after surgery. The sequestrum, granulation tissue, and other infected tissues were thoroughly removed. The wound was then thoroughly cleaned with a benzalkonium chloride solution.No recurrence was observed within 1 year after surgery, and the aesthetic effect was good and the patient was satisfied.
Given the high recurrence rate of OKC, particularly for this patient who underwent conservative surgery, it is advisable to reduce the follow-up interval to detect recurrences early. Minimally invasive treatments like curettage should be considered to address both aesthetic and functional needs. In cases where recurrence is accompanied by infiltration, radical surgery may be necessary.
Conclusions
Mandibular polycystic OKC tends to recur after surgery and can damage the oral mucosal barrier in the surgical area, potentially leading to actinomyces infection and chronic osteomyelitis.
The primary clinical symptoms include gum swelling, pain, and pus discharge. Radiographic signs such as osteolytic destruction, sequestra, and soft tissue swelling, along with the detection of actinomycetes, mycelium, or sulfur granules, are crucial for aiding in diagnosis. Surgical treatment of primary or secondary OKC involves thorough curettage of inflammatory lesions, along with the standard use of antibiotics before and after surgery to prevent infection. This approach is essential for effective treatment.However, it should still be noted that OKC and osteomyelitis both have a certain recurrence, and standardized follow-up after surgery is very necessary, especially for patients who have not been treated with radical surgery.
Acknowledgements
The authors would like to thank Central Laboratory, Jinan Key Laboratory of Oral Tissue Regeneration, Jinan Stomatological Hospital, Jinan, China.
Abbreviations
- OKC
Odontogenic keratocyst
- NBCCS
Nevoid Basal Cell Carcinoma Syndrome
- CBCT
Cone-beam Computed Tomography
Authors’ contributions
Yuxin Ma and Lin Yang contributed equally to this study.H, M,and Y were responsible for manuscript writing and revision. Z and M contributed to the evaluation of CBCT and pathological images.All authors read and approved the final manuscript.
Funding
Not applicable.
Data availability
The dataset used and/or analyzed during the current case report are available from the corresponding author on reasonable request.
Declarations
Ethics approval and consent to participate
This case report was approved by the Ethics Committee of Jinan Stomatological Hospital, Shandong, China. All procedures were explained to the patient, and the written informed consent was signed by the patient.
Consent for publication
We have acquired informed consent from the patient, who has given written consent that their personal or clinical details and any identifying images will be published in this study.
Competing interests
The authors declare no competing interests.
Footnotes
Publisher’s Note
Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.
Yuxin Ma and Lin Yang contributed equally to this work.
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Associated Data
This section collects any data citations, data availability statements, or supplementary materials included in this article.
Data Availability Statement
The dataset used and/or analyzed during the current case report are available from the corresponding author on reasonable request.