Saudi Endodontic Journal

CASE REPORT
Year
: 2020  |  Volume : 10  |  Issue : 2  |  Page : 165--171

A multidisciplinary approach to an unusual cystic lesion of the maxilla – A case report


Farhana Omar1, Tahir Yusuf Noorani2, Huwaina Abd Ghani2, Nik Rozainah Nik Abdul Ghani2, Noor Hayati Abdul Razak3, Nur Diyanah Ab Wahid4,  
1 Conservative Dentistry Unit, School of Dental Sciences, Universiti Sains Malaysia, Health Campus, 16150 Kubang Kerian, Kota Bharu, Kelantan, Malaysia
2 Conservative Dentistry Unit, School of Dental Sciences; Conservative Dentistry Unit, Hospital Universiti Sains Malaysia, Health Campus, 16150 Kubang Kerian, Kota Bharu, Kelantan, Malaysia
3 Oral and Maxillofacial Surgery Unit, School of Dental Sciences, Universiti Sains Malaysia; Oral and Maxillofacial Surgery Unit, Hospital Universiti Sains Malaysia, Health Campus, 16150 Kubang Kerian, Kota Bharu, Kelantan, Malaysia
4 Oral and Maxillofacial Surgery Unit, School of Dental Sciences, Universiti Sains Malaysia, Health Campus, 16150 Kubang Kerian, Kota Bharu, Kelantan, Malaysia

Correspondence Address:
Dr. Tahir Yusuf Noorani
Conservative Dentistry Unit, School of Dental Sciences, Universiti Sains Malaysia, Health Campus, 16150 Kubang Kerian, Kota Bharu, Kelantan
Malaysia

Abstract

A large cyst in the maxilla in relation to a nonvital tooth is not a common occurrence. The present case report demonstrates an unusual case of a giant cyst in the right maxillary antrum region extending from the anterior to posterior right maxilla with no intraoral bony and soft tissue expansion of a healthy 31-year-old female. However, bone erosion was noted and integrity of important anatomical structures of the right maxillary sinus was affected. The adjacent teeth were nonvital without any evidence of root resorption. Conventional root canal treatment together with surgical enucleation of the affected teeth at 6 months follow-up resulted in the successful resolution of the cyst with complete bone regeneration.



How to cite this article:
Omar F, Noorani TY, Ghani HA, Abdul Ghani NR, Abdul Razak NH, Ab Wahid ND. A multidisciplinary approach to an unusual cystic lesion of the maxilla – A case report.Saudi Endod J 2020;10:165-171


How to cite this URL:
Omar F, Noorani TY, Ghani HA, Abdul Ghani NR, Abdul Razak NH, Ab Wahid ND. A multidisciplinary approach to an unusual cystic lesion of the maxilla – A case report. Saudi Endod J [serial online] 2020 [cited 2020 Jul 12 ];10:165-171
Available from: http://www.saudiendodj.com/text.asp?2020/10/2/165/283140


Full Text

 Introduction



Cystic lesions of the maxilla are benign lesions of either odontogenic or nonodontogenic origin. Many classifications have been developing based on the lesion etiologic or pathologic condition. The World Health Organization in 1992[1] classified cyst of the jaw into odontogenic and nonodontogenic origin; either developmental or inflammatory.[1] Further classification has been broadly developed by Shear and Speight who further classified cysts into three main groups which are cysts of the jaws, cysts associated with the maxillary antrum, and cysts of the soft tissues of the mouth, face, neck, and salivary glands.[2]

A dome-shaped radiopaque shadow, commonly seen on the floor of the maxillary sinus, is sometimes inaccurately referred to as antral mucoceles. Their histologic appearance is that of normal or inflamed maxillary sinus lining. Less commonly, epithelium-lined retention cysts, similar to those of the minor salivary glands, are found, but mucoceles of the type found in the oral cavity apparently do not occur in the maxillary sinus.[3] Pseudocyst of maxillary antrum or previously called as mesothelial cyst or interstitial cyst or lymphangiectatic cyst or false cyst could also be considered as one of the diagnosis based on the clinical, radiographic, and histologic features of the case.[3],[4] Clinically, presence of toothache and spontaneous disappearance and reemergence of the lesion have been reported among manifestations of pseudocyst which could also be seen in this case.[4] Radicular cyst, on the other hand, which may extend into the maxillary sinus is caused by host defense to microbial challenge from infection extending from the root canals of a necrotic tooth.[5]

The choice of treatment may be determined by some factors such as extension of the lesion, relation with noble structures, evolution, origin, clinical characteristics of the lesion, cooperation, and systemic condition of the patient. Treatment of radicular cyst usually involves conventional nonsurgical root canal treatment when lesion is localized.[6] However, the management of large cystic lesions has been the subject of prolonged debate, but surgical treatment such as enucleation, marsupialization, or decompression may be the preferred approach to treating a large cyst.[7],[8],[9],[10],[11]

Most of cysts associated with the maxillary antrum spontaneously regressed or showed no significant change in size over the long term and were rarely related to odontogenic pain.[12] This is one of an unusual and unique case of cyst associated with maxillary antrum being managed dentally and was associated with nonvital affected teeth unlike several other case reports.[13],[14]

The present case report demonstrates an unusual case of a giant cyst in the right maxillary antrum region extending from the anterior to posterior right maxilla with no intraoral bony and soft-tissue expansion.

 Case Report



A 31-year-old female was referred from oral and maxillofacial surgery unit after informed consent was obtained for endodontic treatment of upper right lateral incisor, canine, first premolar, and second premolar (teeth #12, 13, 14, and 15, respectively). History revealed that the patient complained of right facial swelling since several months which gradually increased in size and was associated with pain. There was a history of fever during the 1st week when swelling started to appear. Right facial swelling was associated with right upper toothache, but the patient was unable to locate the offending tooth. The dental history revealed repeated prescription of antibiotics and analgesics at private dental clinics for the same recurrent swelling for the last 6 months. Medical history was unremarkable. Extraorally, lymph nodes were nonpalpable. There was a small mesial proximal composite restoration on tooth #12, while the rest of teeth in the first quadrant were sound. Teeth #12, 13, 14, and 15 were nonresponsive to pulp sensibility testing and #13 presented with grayish-to-blackish discoloration [Figure 1]. The buccal and palatal mucosa was devoid of any swelling or sinus drainage. The patient's oral hygiene was generally fair.{Figure 1}

 Investigations



The patient was advised for orthopantomography (OPG) radiographic examination, which revealed a large unilocular radiolucency with well-defined dome-shaped radiopaque border on the right maxilla area associated with the roots of upper right anteriors and premolars [Figure 2]. To investigate the lesion three-dimensional (3D) radiography, cone-beam computed tomography (CBCT) was done. Coronal view of CBCT [Figure 3]a showed the lesion in the right maxilla and lesion expanded mesiodistally causing displacement of the right maxillary sinus superiorly. Sagittal view [Figure 3]b showed direct communication of the lesion with the root apex of tooth #13. Axial view [Figure 3]c showed expansion of the lesion laterally and distally. 3D view [Figure 3]d showed bony fenestration on the right maxilla. On the basis of the clinical and radiographic findings, a differential diagnosis of cysts of maxillary antrum, mucocele, retention cyst, pseudocyst and postoperative maxillary cyst, or inflammatory cyst of odontogenic origin (radicular cyst) was made. A biopsy was considered for further investigation. Under local anesthesia, a small incision was made at the most fluctuant right buccal mucosal area, and lesion fluid was aspirated using fine-needle aspiration biopsy and sample sent for histopathology investigation. The result was nonspecific inflammatory infiltrate.{Figure 2}{Figure 3}

Treatment

A diagnosis of pulp necrosis with radicular cyst for teeth #12, 13, 14, and 15 was established and conventional endodontic treatment was initiated for teeth #12, 13, 14, and 15 as an emergency management by the oral and maxillofacial surgery team, to address the patient's complaint of pain before the patient was referred. In the endodontics unit, periapical radiographs were taken [Figure 4] to serve as preoperative baseline radiographs. Following rubber dam isolation, teeth #12–15 were reaccessed, cleaning and shaping was done using ProTaper Universal Rotary Files (DENTSPLY, UK), 5.25% sodium hypochlorite (HUSM Pharmacy, Malaysia) was used as irrigating agent during cleaning and shaping. Finally, the canals were rinsed with 17% ethylenediaminetetraaceticacid (Dentonics Inc., USA) and dried with paperpoints. However, for tooth #13, direct communication with the periapical lesion resulted in inability to dry the root canal with continuous straw-like fluid discharging out from the root canal. As a result, the severity of the discoloration of tooth #13 increased and tooth could not be obturated. Hence, the tooth was medicated with nonsetting calcium hydroxide (Apexcal® Calcium Hydroxide, Ivoclar Vivadent). Nevertheless, teeth #12, 14, and 15 were obturated by a combination of warm vertical condensation technique and injectable gutta-percha backfill technique using System B™ Cordless (Kerr Dental, Germany). AH Plus (Dentsply, Germany) resin-based root canal sealer was used in combination with gutta-percha. Increase in the discoloration of tooth #13 could be suggested due to direct communication of the root tip of the tooth with the lesion causing continuous exposure of the lesion fluid with nonviable pulp tissue in the root canal of tooth #13. Continuous medication of tooth #13 with calcium hydroxide for 3 months did not reduce the discharge into its root canal. Hence, surgical lesion exploration and enucleation were done under general anesthesia [Figure 5]. The lesion was enucleated and sent for histopathology examination [Figure 6]. As the lesion was extensive, it caused perforation of the maxillary sinus near the roots of the right maxillary first molar (#16). Hence, the sinus perforation was repaired with a membrane (Pericard® LEMB, USM, Malaysia) which was sutured in place with vicryl 4/0 suture (Ethicon, USA). Surgical apicectomy was then performed at the root tips of teeth #12, 13, 14, and 15 using a straight fissure bur in slow-speed straight handpiece and 3 mm of the root end cut. The apical root end was then prepared to receive a root end filling. A 3-mm deep apical preparation was done on all affected teeth using microsurgery ultrasonic tip. White mineral trioxide aggregate (ProRoot® MTA, Dentsply Sirona, Canada) was placed at the prepared apical root end and packed densely [Figure 7]. All the excess MTA were cleaned and removed. Granular synthetic bone graft (GranuMas®, GranuLab, Malaysia) was placed at the large bony defect which as a result of removal of the cyst. The surgical area was then sutured with vicryl 4/0. The patient was then reviewed for up to 6 months postsurgery.{Figure 4}{Figure 5}{Figure 6}{Figure 7}

Due to inability to dry the root canal of tooth #13 even after aspiration of the cystic fluid from the root canal, decision to postpone the obturation of #13 was made. Root canal treatment was continued for tooth #13 until obturation stage after 1 month of surgical cyst enucleation. Tooth #13 was obturated by a combination of warm vertical condensation technique and injectable gutta-percha backfill technique using System B™ Cordless (Kerr Dental, Germany). AH Plus (Dentsply, Germany) resin-based root canal sealer was used in combination with gutta-percha. Nonvital tooth bleaching was then done to enhance the color of the tooth. Internal tooth bleaching for tooth #13 was done using (Opalescence® Endo, Ultradent, America) placed in the pulp chamber for 5 days.[15] Baseline color was recorded, and the color changes after bleaching were compared with the baseline. Bleaching procedure was repeated two times until the patient was satisfied with the color changes.[15] Upon review appointment, the patient came with a complaint of pain and tenderness on the right maxillary central incisor (#11). Examination and investigations revealed tenderness to percussion without any sign of swelling or sinus tract formation. However, pulp sensibility test gave no response, and radiographic investigations revealed widening of periapical periodontal space. Hence, conventional root canal treatment was done on tooth #11 as well using the same technique as previously described for other teeth.

Outcome and follow-up

Histological examination of the lesion revealed the presence of an inflamed cyst on the wall of the maxillary antral region [Figure 8]. However, a confirmed diagnosis and origin of the cyst could not be given after histological examination as the findings were not typical of any particular cyst. At 6 months follow-up, no recurrence was observed [Figure 9], orthopantomogram and periapical radiograph revealed new bone formation at the site of cystic lesion [Figure 10]. The other teeth remained vital.{Figure 8}{Figure 9}{Figure 10}

 Discussion



The unique intraoral manifestation in this case is association of the lesion with nonvital right anteriors and premolars. Unlike other cases reported, several nonvital teeth were encountered in this case.[16],[17] All these teeth were sound and with no previous evidence of trauma yet were found to be associated with such a big lesion. Surprisingly, tooth #13 had direct communication with the lesion and act as passage for the cystic fluid drainage causing development of tooth discoloration which is rarely seen in the other cases.[3],[4] Indeed, pulp necrosis of tooth #13 could be the cause of the lesion. Periapical lesions of endodontic origin are followed by infection and necrosis of the pulp.[5] Pulp necrosis is commonly associated with either previous trauma, restorations, or previous history of pain or caries. However, tooth #13 was sound, and there was no history of trauma, and hence, a cause of pulp necrosis of tooth #13 could not clearly be identified. Furthermore, histologic examination did not reveal features of a particular cyst, and hence, lesion is less likely to be of endodontic origin even though a direct communication between the apex of tooth #13 and the lesion existed. Nevertheless, the lesion is likely to be of nonodontogenic origin, which expanded, leading to compression of the neurovascular bundles, causing necrosis of the involved teeth.[5] In addition, the length of the maxillary canine root and its proximity to the maxillary sinus (as it can been seen from the preoperative OPG for the left side) could have resulted in a direct communication between the canine root tip and the lesion.

In contrast to other cases with periapical lesions, which were successfully treated with conventional root canal treatment, this case was different.[6],[18],[19] Progression of the lesion healing after conventional root canal treatment was not evident in this case which warranted further investigations as different etiologic nonodontogenic origin of the lesion was suspected.

Treatment options for radicular cysts can be conventional nonsurgical root canal treatment when lesion is localized or surgical treatment such as enucleation, marsupialization, or decompression when the lesion is large.[6],[20] Surgical enucleation of the lesion in this case was decided based on lesion criteria both histologic and nonhistologic.[21],[22] In this current case report, the decision for surgical cyst enucleation was made in conjunction with conventional root canal treatment due to no sign of healing of the periapical radiolucency after completion of the root canal treatment on tooth #12, 14 and 15.[23] Furthermore, continuous exudates into the root canal of tooth #13, which had a direct communication with the cyst, warranted surgical enucleation. Paucity in evidence exists with regard to the appropriate treatment of cases with such large periapical cyst. Limited evidence, mainly from case reports, are available [Table 1]. Hence, combination of conventional and surgical treatment was proposed in this case based on clinical judgment.{Table 1}

Nonsurgical option involved root canal treatment on affected nonvital teeth #12, 13, 14, and 15 by conventional root canal treatment.[6],[18] This conventional root canal treatment was done prior to surgical cyst enucleation. The decision to delay the obturation of tooth #13 was due to inability to achieve canal disinfection, thus complete elimination of the root canal biofilms could not be achieved.[24],[25]

The resultant nonvital tooth #11 could potentially be due to pressure from the cyst lesion or trauma introduced during surgical cyst enucleation.[26],[27] Since the cystic lesion was extending to the mesial surface of root of #11, enucleation of the cyst lining against the root surface area may cut the neurovascular bundles which lead to loss of vitality of the tooth.[27]

Internal bleaching was done as thefirst-line conservative treatment to treat tooth discoloration.[28],[29] Discoloration on tooth #13 during endodontic treatment could be related to the intrinsic discoloration due to incomplete removal of necrotic pulp tissues that interact with cystic fluid which directly drained from the apex of tooth #13.[30]

 Conclusion



Although the origin of the lesion could not be identified, a multidisciplinary treatment approach involving endodontic therapy of the involved teeth and surgical enucleation of the lesion resulted in successful resolution with complete bone regeneration.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Acknowledgment

The authors would like to thank the management of the Hospital Universiti Sains Malaysia, Kubang Kerian, Kelantan, for allowing the use of space and assets belonging to the Hospital during the treatment of the presented case.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

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