J.Bio.Innov4(1),pp:01-11,2015|ISSN2277-8330 (Electronic) Kumar Nilesh et al.

UNUSUALLY LARGE RADICULAR CYSTS OF MAXILLA : STEPS IN DIAGNOSIS & REVIEW OF MANAGEMENT

Dr. Kumar Nilesh*, Dr. Anuj S Dadhich1, & Dr. Pramod R Chandrappa

Department of Oral & Maxillofacial Surgery, School of Dental Sciences, KIMSDU, Karad 1 Department of Oral & Maxillofacial Surgery, Rural Dental College & Hospital, Loni

Email:[email protected]

(Received on Date: 13th November 2014 Date of Acceptance: 2nd December 2014)

ABSTRACT Radicular cystsare common of jaw bone which invariably presents as well- defined radiolucency around the apex of the involved tooth. It is diagnosed on routine radiography or causes a slow growing swelling, limited to apical region of a tooth. Radicular cyst presenting as unusually large maxillary lesions is uncommon. This paper reports two cases of large radicular cyst involving maxilla. The first case presented as a single lesion occupying almost the entire anterior maxilla, while the second case presented a large cyst occupying the entire left half of the maxilla and the maxillary sinus. Unusually large radicular cyst of maxilla require stepwise clinical, radiological and laboratory evaluation to reach a definitive diagnosis. Although no consensusexist regarding the precise treatment modality for large radicular cyst of maxilla, both cases were successfully treated by surgical removal of the cyst following endodontic therapy.

Keywords: Large, Radicular cyst, Maxilla, Enucleation

No. of Figures: 8 No. of References : 28

2015 January Edition |www.jbino.com | Innovative Association J.Bio.Innov4(1),pp:01-11,2015|ISSN2277-8330 (Electronic) Kumar Nilesh et al.

INTRODUCTION canal is completely sealed, all cell Radicular Cysts are believed to be formed components participating in inflammatory from epithelial cell rests of Malassez (ERM), reaction gradually resolve (10). which are remnants of Hertwig’s epithelial Subsequently majority of inflammatory root sheath, present within the periodontal cells, endothelial cells and in the ligament. Proliferation of these epithelial paeriapical lesion are deleted by cell rests is frequently associated with apoptosis or programmed cell death (11). stimuli from periapical periodontal However surgical removal following secondary to pulpitis (1). endodontic treatment is required for cases During periapical inflammation, host cells in not amenable to conservative therapy the periapical tissues release many (12). Presence of unusually large inflammatory mediators, proinflammatory periapical lesion may often lead to , and growth factors which diagnostic dilemma. Such cases need to induce proliferation of the ERM in all be evaluated in a step wise manner to directions to form a three-dimensional ball reach a definite diagnosis. This article mass(2,3). As the epithelial mass grows, the reports two cases of large radicular cyst of central cells move further away from their maxilla thepresentation, steps in diagnosis source of nutrition and undergo and management of these lesions are also and liquefaction degeneration, forming discussed. central cystic cavity lined by epithelial wall. Following its formation, radicular cysts Case Report: grows by periapical bone resorption Case 1:A 34 year old female presented mediated by prostagladins and cytokines. with complaint of painless swelling over It is interesting to note that most roof of the mouth. The patient first noticed inflammatory mediators which induce the swelling about 6 months back, which proliferation of epithelial cell rests also had grown gradually to its present size. No mediate bone resorption in inflammatory history of trauma or tooth was periapical lesion (4-9). However there is no reported. Extraoral examination revealed time-lapse study showing that the size of no facial swelling or obvious facial apical cysts gradually increases as asymmetry. Intraoral examination showed periapical bone destruction increases. a palatal swelling starting distal to right 1st Clinically radicular cyst presents as premolar and extending anteriorly, across periapical lesion, ranging from 0.5 to 1.5 the midline to the contra laterallateral cm in size. Smaller periapical cysts can incisor, measuring about 4 x 3.5 cm in size often be treated conservatively by non- (Figure 1a). Swelling was non-tender and surgical endodontic therapy. Proper firm on palpation. The overlying palatal endodontic therapy of the involved teeth mucosa appeared normal. Maxillary removes irritants in the canals by chemo- anterior teeth were grade one mobile, with mechanical instrumentation. As the root normal depth of gingival sulcus on diagnostic probing. All the maxillary

2015 January Edition |www.jbino.com | Innovative Association J.Bio.Innov4(1),pp:01-11,2015|ISSN2277-8330 (Electronic) Kumar Nilesh et al.

anteriors were non-vital on electric percha. The cyst was approached through testing. The clinical presentation was labial gingiva mucoperiosteal flap and suggestive of a benign intraosseous tumor removed by enucleation. Apicoectomy or an odontogenic cyst. Clinical differential and retrograde root end filling for maxillary diagnosis included central giant cell lesion, anterior teeth was done with MTA to obtain radicular cyst, odontogenic Keratocyst, apical seal. The labial and interdental globulomaxillary cyst and ameloblastoma. bone support around the maxillary anterior Aspiration of the lesion yielded yellow color teeth appeared to be compromised due fluid which was subjected to cytochemical to bone resorption caused by the evaluation (Figure 1b). The fluid consisted periapical lesion. After through irrigation of of cholesterol crystal and numerous the bone cavity & hemostasis, the bone inflammatory cells, suggestive of an defect was grafted with platelet rich inflammatory cystic lesion. concentrate to facilitate periapical wound A cone beam computed tomography healing. Autologous platelet rich (sironaorthophos X3GD, Germany) was concentrate was prepared from 10 ml of advised, to study the size and extent of the patient’s own blood centrifuged at 2888 lesion. The 3D reconstruction showed a rpm for 12 minutes (Remi Lab centrifuge) large osteolytic lesion extending form (Figure 3). The defect was closed primarily maxillary right 1st premolar to maxillary left and cyst lining was sent for histopathology canine and superiorly till the floor of the examination, which confirmed the nasal fossa. The roots of the associated diagnosis of radicular cyst [Figure 4]. teeth were displaced and showed no Splinting of maxillary anterior teeth was resorption (Figure 2a). Axial section showed done for 3 weeks, using 22-gauge stainless 5 x 3.5 cm intraosseous radiolucent lesion steel wire stabilized with light cured with defined sclerotic borders involving composite in immediate postoperative almost entire anterior maxilla (Figure 2b). period. At 1 year follow up the patient was Diagnosis of inflammatory odontogenic asymptomatic and showed no sign of cyst was established based on the clinical, recurrence. There was no mobility of the radiological and aspirate evaluation. anterior teeth and imaging of the surgical Surgical enucleation of the cyst was site showed normal healing of the bone planned under general anesthesia after defect (Figure 5).Case 2:A thirty six year old root canal therapy of the involved female, presented with complain of maxillary teeth. Biomechanical painless swelling of eight months duration perpetration and irrigation (with saline & over left side of midface. The patient gave 1% sodium hypochlorite alternatively) of history of fall from bicycle during her root canals of all the maxillary anterior was childhood. On examination a large extra- done under antibiotic cover. Calcium oral swelling was evident, extending from hydroxide intracanal medication was left infraorbital region to level of corner of placed for period of 1 week. Subsequently mouth, measuring roughly 4 x 5 cm in size. the root canals were obturated with gutta- The nasolabial fold was obliterated along

2015 January Edition |www.jbino.com | Innovative Association J.Bio.Innov4(1),pp:01-11,2015|ISSN2277-8330 (Electronic) Kumar Nilesh et al.

with elevation of ala of left nose (Figure 6). incisors and placement of calcium The swelling was non-tender and firm on hydroxide intracanal dressing for one palpation. Intraoral examination showed week, the patient was prepared for soft fluctuant swelling localized over left surgical removal of the cyst under general maxillary vestibule in premolar-molar anesthesia. After the cyst was enucleated region. Maxillary left central and later in-toto, apicoectomy of the left maxillary incisors were non-responsive to electric incisors and retrograde root end filling with pulp vitality test. On radiological MTA was done. The cyst lining was evaluation, a large radiolucent lesion was submitted for microscopic evaluation evident involving the entire left maxilla. which confirmed diagnosis of radicular cyst Orthopantamogram showed a well- (Figure 8). The postoperative healing was defined lesion with sclerotic border, uneventful. The patient was symptom free extending from maxillary midline till the and showed healing intraosseous surgical maxillary tuberosity. The entire left maxillary defect, at 6 months follow-up.Radicular sinus appeared hazy on paranasal sinus cysts are the most common odontogenic view (Figure 7). The presenting features cyst affecting the human jaw (13). It is were suggestive of cyst of odontogenic or believed to be formed from proliferation of maxillary sinus origin. Content of the lesion epithelial residues in the periodontal was aspirated intra-orally under local ligament as a result of periapical anaesthesia using 18 gauge needle. Cyto- inflammation secondary to pulpitis. chemical examination of the straw color Although they are direct sequel to apical aspirate showed numerous inflammatory periodontitis, not every case of apical cells and cholestrol crystals. Based on the periodontitis causes formation of radicular history, clinical presentation, vitality test, cyst (14). Since radicular cyst form at the radiographic features and evaluation of apex of tooth root(s) it is also called aspirate, diagnosis of inflammatory . They may be associated odontogenic cyst following of with any tooth. However it commonly left maxillary incisors was made. However, involves maxillary anterior teeth and looking into the unusually large size of the seldom involves primary dentition. Their lesion, incision biopsy was planned to prevalence is highest in the third decade conform the diagnosis. The lesion was of life and are more commonly seen in accessed intraorally through Caldwell-Luc males than females (15). Position of the approach (by creating bony window in maxillary anterior teeth makes them more region of canine fossa). A portion of the prone to injury and subsequent pulp cyst lining was removed and sent for sent necrosis. This may be the possible for histopathological examination. explanation for their prevalence in males Diagnosis of radicular cyst was confirmed and in maxillary anterior region. One of our based on incision biopsy report. Following reported cases (case 2) gave history of chemo-mechanical preparation of pulp trauma to maxillary anterior teeth, which chambers of the involved left maxillary could have been the possible cause of

2015 January Edition |www.jbino.com | Innovative Association J.Bio.Innov4(1),pp:01-11,2015|ISSN2277-8330 (Electronic) Kumar Nilesh et al.

initiation of cyst formation. Periapical another important diagnostic test for cystic inflammation and pulpitis secondary to lesion. It helps to rule out any vascular dental caries is another cause of formation lesion and provides fluid specimen for of radicular cyst. Most of the radicular cysts cytochemical evaluation. In our reported are symptomless and discovered when cases the cyst content was aspirated periapical radiographs are taken for non- under local anaesthesia, using wide gauge vital teeth. They present as round to oval needle. The fluid was yellowish-white in radiolucency associated with root apices, color and contained cholesterol crystal varying in size from 0.5 to 1.5 cm. As the and numerous inflammatory cells. The cyst grows in size by resorption of the treatment of radicular cyst, as a disease of surrounding periapical bone, it causes root canal infection or inflammation, localized jaw swelling. Pain and pus consists of eradicating or substantially discharge are presenting feature of an reducing the microbial load and infected radicular cyst. Only few cases of inflammatory mediators from the canals radicular cysts involving large part of and periapical tissue. Numerous measures maxilla have been previously reported in have been described to reduce the literature (16-19). Interestingly the reported number of microorganisms in the root cases of large radicular cyst involve maxilla canal system, including the use of various and very often extended to occupy large irrigation regimens and intracanal portions of the maxillary sinus. A logical medicaments. Various root canal irrigants reasoning to explain this association can used include Sodium hypochlorite, be the relatively thin and cancellous ethylenediaminetetraacetic acid (EDTA), nature of the maxillary bone as compared citric acid and chlorhexidine. Sodium to dense mandible, which would be more hypochloride is the most commonly used resistant to expansion and resorption. Also endodontic irrigant (21). In our cases 1% extension of these periapical lesions into Sodium hypochloride and saline was used the maxillary sinus would allow its alternatively as root canal irrigant. Intra- unrestricted growth, leading to formation canal medicaments eliminate or reduce of giant radicular cyst. number of microorganisms, rendering Although intra-oral radiographs are canal contents inert. It helps in prevention sufficient for studying a periapical cysts, of post-treatmentpain, and enhances larger lesions often require more extensive anesthesia (22). Calcium hydroxide is most imaging to evaluate the location, size, widely used intracanal medicament. It also proximity to vital structures and extent of plays a major role as an inter-visit dressing the lesion (20). Both extra-oral radiographs in the disinfection of the root canal system. (including orthopantamogram¶nasal Reduction of inflammatory cells and sinus view) and three dimensional imaging mediators from roots of the involve tooth using cone beam computed tomography may often lead to resolution of smaller were used in our cases for radiological periapical lesion. However cases which do assessment. Aspiration of the cyst fluid is not respond to conservative management

2015 January Edition |www.jbino.com | Innovative Association J.Bio.Innov4(1),pp:01-11,2015|ISSN2277-8330 (Electronic) Kumar Nilesh et al.

require surgical intervention. Surgical intraosseous bony cavity is irrigated treatment is preferred approach to treat generously to remove all periapical large radicular cyst. For apical irritants. In our first case report we had radiolucency larger than 20 mm in grafted the bony cavity with autologous diameter or having cross-sectional area platelet rich concentrate to facilitate greater than 200 mm2, surgical removal healing of the large defect of anterior may be the best option (23). Surgical maxilla, which had caused significant treatment is also recommended when the bone loss around the maxillary anterior canal appears calcified or obstructed and teeth. The teeth were subsequently cannot be negotiated with endodontic splinted in immediate postoperative instruments. The surgical treatments for period. Platelet rich concentrate is known radicular cyst include total enucleation, to initiate osteoinduction process which is marsupialization or a combination of these mediated by growth factors present in techniques (24, 25). Removal of entire cyst platelets. High number of platelets in the allows histopathological evaluation of cyst concentrate produces large amount of lining, thereby helping in reaching a growth factors, initiating bone formation definitive diagnosis and ruling out any (27). Beside bone regeneration, it also aid malignant or dysplastic changes in large in healing of surrounding soft tissues (28). cyst. After the enucleation of the cyst, Both the reported cases were followed up apicoectomy of the involve teeth is done. for 6- 12 months and showed uneventful Apicoectomy involves sectioning of the healing of the lesion with no reoccurrence apex of the root and reterograde filling to or complication.Periappical attain a sound periapical seal. Historically secondary to pulp inflammation is a amalgam was the most frequently used common finding. However usually large material in apicoectomy procedure. IRM, periapical lesion require stepwise clinical, super EBA and MTA are newer and more radiological and laboratory evaluation to suitable materials, and give better results in reach a definitive diagnosis. Such large apicoectomy procedures than Amalgam lesion often warrants detailed radiological (26). Advantages of MTA include high assessment to study the location, size and biocompatibility and excellent sealing extent of the lesion. Cytochemical ability with low marginal leakage. However evaluation of the aspirate provides high cost of MTA is a major disadvantage valuable input regarding nature of the to its use in every clinical situation. All our cyst. Although no consensusexist regarding cases were treated with surgical the precise treatment modality for large enucleation of the periapical cyst along radicular cyst of maxilla, both cases were with apicoectomy and retrograde fillings of successfully treated by surgical removal of involved roots with MTA. the cyst following endodontic therapy of The bony defect left after the removal of the involved teeth. Enucletion of the cyst the cyst lining heal uneventfully after a provided the lining for complete proper apical seal is attained and the histological evaluation of the periapical

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pathology and to rule out any malignant large cysts. or dysplastic changes in these unusually

Figure 1: (Case 1) Swelling over anterior palate (block arrows)

Figure 2: (Case 1) (a) Cone beam 3D reconstruction showing large radiolucent lesion involving almost entire anterior maxilla (b) Axial section, at the level of Apical third of maxillary anterior teeth showing the dimensions of the cyst.

Figure 3: (Case 1) Platelet rich concentrate prepared from patients’ blood (centrifuged 10 ml blood at 2888 rpm for 12 minutes; Remi Lab centrifuge)

2015 January Edition |www.jbino.com | Innovative Association J.Bio.Innov4(1),pp:01-11,2015|ISSN2277-8330 (Electronic) Kumar Nilesh et al.

Figure 4: (Case 1) H & E stained Photomicrograph showing (a) Stratified squamous lining epithelium ( 4X magnification) (b) Cholestrol clefts (black arrow) and giant cells (red arrow) in the wall of the cyst lining (40X magnification).

Figure 5: (Case 1) One year post-operative radiological showing healing at the surgical site.

Figure 6: (Case 2) (a) Frontal (b) Lateral (c) Bird view of the extraoral swelling evident over left side of mid-face; (d) Intraoral swelling in the left maxillary vestibule

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Figure 7: (Case 2) (a) Orthopantamogram showing large radiolucent lesion with sclerotic borders (red arrows) occupying the entire left maxilla (b) Paranasal sinus view showing haziness of the left maxillary sinus (blue arrow), as compared to the normal radiolucency of right maxillary sinus (red arrow).

Figure 8: (Case 2) (a) Cyst removed in-toto (b) Photomicrograph showing (H & E stained; 10X magnification) showing cyst lining made up of stratified squamous epithelium (black arrow).

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