Authors: Soufiane Boussouni, Chloe Laporte, Martin Gaudinat, Anne Margaux Collignon, Thông Nguyen and Loredana Radoï
Department of Oral medicine, Louis Mourier Hospital, Paris Diderot University, AP-HP, France
Corresponding author: Prof. Loredana Radoï, Department of Oral medicine, Louis Mourier Hospital, Paris Descartes University, AP-HP, France. Email: loredana.radoi@parisdescartes.fr
ABSTRACT:
Dentigerous cysts are the second most frequent odontogenic cysts. They are usually solitary and associated with impacted third molars in young patients. Multiple cysts are rare and generally occur in syndromic patients. We present the surgical and endodontic management of a bilateral mandibular dentigerous cysts in a non-syndromic patient and a comprehensive literature review.
A 25-year-old woman was referred with pain in the right side of the mandible. Clinical, radiological and anatomopathological examinations led to the diagnosis of dentigerous cysts associated with impacted mandibular third molars. The patient’s follow-up was marked by the pulp necrosis of mandibular second molars, which were treated endodontically. Bone repair was complete at 18 months.
The literature review identified only 43 cases of multiple dentigerous cysts occurring in healthy patients reported over the past 10 years. The endodontic treatment of resorbed adjacent teeth was rarely attempted in the cases reported in the literature. Surgery for large bone lesions carries the risk of complications requiring a multidisciplinary approach.
INTRODUCTION
Dentigerous cysts belong to the developmental odontogenic cysts group. Derived from epithelial residues of the dental lamina, they develop at the enamel-cement junction of an impacted tooth. Dentigerous cysts represent the second most common odontogenic cysts after radicular cysts.1 They are often unique and associated, in order of frequency, with the inclusion of the mandibular third molar, maxillary canine and mandibular second premolar.2 Multiple localisations have rarely been reported in the literature in non-syndromic patients. We report a rare case of bilateral mandibular dentigerous cysts associated with adjacent teeth resorptions, managed by surgical and endodontic treatment in a non-syndromic patient, along with an updated review of the literature.
OBSERVATION
A 25-year-old woman was referred to the dental emergency department by her dentist for severe pain in the right side of the mandible (visual analogue scale score 80/100), accompanied by deterioration of general condition and asthenia. The pain was radiating to the temporal region and exacerbated when chewing or brushing.
The patient had no medical condition or surgical history apart from hepatitis A. She was a smoker (5 pack-years). General examination did not reveal any lymphadenopathy or facial asymmetry. The oral examination revealed a mucous cap partially covering the crown of the right mandibular second molar, whose gum was oedematous and erythematous. Pulp sensitivity tests were positive on all mandibular teeth, the percussion test was negative, and palpation of the buccal and lingual cortices was painful on the right side of the mandible. The teeth had physiological mobility.
The panoramic radiograph showed bilateral homogeneous radiolucent lesions with well-defined borders, extending from the mandibular angles to the distal roots of first molars, and from the alveolar ridge to the basilar border of the mandible (Figure 1). The lesions were associated with the crowns of the impacted mandibular third molars, the right third molar being driven back towards the mandibular rising branch. The Cone Beam Computed Tomography (CBCT) examination showed thinning of cortical plates and repression of the lower alveolar nerve (Figure 2). The apices of second mandibular molars adjacent to the lesions were resorbed, in particular the distal root of the one on the left. The presumptive diagnosis was bilateral mandibular dentigerous cysts with an infectious complication on the right side.
The two cysts were enucleated and both mandibular third molars were removed under general anaesthesia (Figure 3). The enucleation specimens were sent for anatomopathological analysis which confirmed the diagnosis of dentigerous cysts. The post-operative course was uneventful. The patient consulted two months later for facial cellulitis originating from pulp necrosis of the right mandibular second molar. Cold and electrical pulp sensitivity tests were also negative for the left mandibular second molar. The cellulitis was treated with antibiotic therapy (amoxicillin and metronidazole) and analgesics (paracetamol and tramadol). Both mandibular second molars were treated endodontically with the placement of a temporary filling based on calcium hydroxide, renewed every two weeks until the symptoms had disappeared and the canal had dried up; and this for 4 months. A CBCT was carried out four months after the intervention and before performing the final root canal fillings. It showed ongoing but late bone repair on the left side where drying of the root canals was more difficult to achieve. The right mandibular second molar was sealed with tricalcium silicate-based cement (Mineral Trioxide Aggregate, MTA®), indicated for its biocompatibility and easy manipulation. A gingival sulcular suppuration and a seepage of the canals were persistent on the left mandibular second molar, which led to pain at chewing. For this reason, antibiotic treatment with good bone diffusion (ofloxacin and clarithromycin) was prescribed until the symptoms improved and the canal drying was obtained (14 days). After stopping the suppuration, the left mandibular second molar was filled with MTA® (Figure 4). The patient was seen every three months until the end of the follow-up (at 18 months) to check bone repair and vitality of mandibular first molars (Figure 5).
FIGURES
Figure 1: Panoramic radiograph showing bilateral mandibular angular radiolucent lesions related to the impacted mandibular third molars.
Figure 2: Preoperative CBCT (coronal (A), axial (B), right and left, (C,D) sections) showing extensive bilateral osteolysis extending from the mandibular angles to the distal roots of the first molars, and apical root resorptions of the second molars.
Figure 3: Intraoperative clinical photographs.
Figure 4: Post-endodontic retro-alveolar radiographs of right (a) and left (b) mandibular second molars.
Figure 5: Panoramic radiograph at 18 months showing complete bone healing.
TABLES
Table 1 – Part 1: Summary of cases of non-syndromic bilateral or multiple dentigerous cysts reported between 2009 and 2019
Table 1 – Part 2: Summary of cases of non-syndromic bilateral or multiple dentigerous cysts reported between 2009 and 2019
Table 1 – Part 3: Summary of cases of non-syndromic bilateral or multiple dentigerous cysts reported between 2009 and 2019
Table 1 – Part 4: Summary of cases of non-syndromic bilateral or multiple dentigerous cysts reported between 2009 and 2019
DISCUSSION OF THE LITERATURE REVIEW
Dentigerous cysts are the second most common odontogenic cysts after radicular cysts, with a prevalence of approximately 21%; they are more frequent in the male population with a peak between the second and the fourth decade.3 They are mostly associated with the inclusion of the mandibular third molars. Their discovery is generally fortuitous except in the event of secondary infection or delayed tooth eruption.4 These are generally unique cysts in healthy patients. Indeed, multiple localisations have rarely been described in patients without major syndromes.4,5 In a series of 2,082 histologically confirmed dentigerous cysts, Zhang et al.6 found that multiple cysts not associated with any syndromes or systemic conditions represented 2.5% of cases. However, there is no clear evidence to suggest the cause of bilateral dentigerous cysts in non-syndromic patients.
According to the authors, the number of cases reported from 1943 (year of publication of the first case of non-syndromic multiple dentigerous cysts) to 2009 was small, varying from 117 to 42.8 However, as dentigerous cysts represent a common pathology, a publication bias cannot be excluded. Another explanation may be the underdiagnosis of these lesions before the use of new imaging methods such as CBCT permitting the identification of multiple lesions. These forms generally occur within certain syndromes such as Maroteaux-Lamy, Gardner and Gorlin syndromes or cleidocranial dysplasia.9.10 Medication (a combination of cyclosporin A and calcium channel blockers) has been associated with multiple dental cysts.5 In the reported case, a general disease and taking medication were ruled out after interview and general examination by patient’s attending physician.
We carried out a literature review from January 2009 to December 2019 on PubMed, Scopus and Google Scholar databases using the search equation (‘bilateral’ OR ‘multiple’ OR ‘bimaxillary’) AND (‘dentigerous cyst’) AND (‘jaw’ OR ‘mandible’ OR ‘maxilla’) and by performing a manual bottom-up search based on the selected publications; 53 articles in the English language, limited to humans, were identified; after reading the titles and abstracts, 39 articles presenting multiple, bilateral (mandible or maxilla) or bimaxillary dentigerous cysts were selected; among them, three articles were excluded because either they reported cases of single cysts associated with two or multiple included teeth or because they described multiple cysts in patients with syndromes or systemic conditions.
The synthesis of the 36 publications retained in the review.7,8,11-44 reporting 43 cases of multiple/bilateral/bimaxillary dentigerous cysts is presented in Table 1.
The mean age was 18.07 years-old (standard deviation, SD = 12.18), and 67.44% of patients were men (29 cases out of 43).
- Regarding the circumstances of cysts discovery, the diagnosis was fortuitous in 37.21% of cases (16 patients) or following infectious accidents in 39.53% of them (17 patients) or dental mobility in 6.98% of cases (3 patients). Data were not available for 16.28% of cases (7 patients)
- The affected arches were the mandible bilaterally in 53.49% of cases (23 patients); the maxilla bilaterally in 27.91% of cases (12 patients); the maxilla and mandible in 18.60% of cases (8 patients) with 2, 3 or 4 quadrants affected
- The dentigerous cysts were related to impacted mandibular second premolars in 14 patients (32.55%), maxillary canines in 13 patients (30.23%) and mandibular third molars in 12 patients (27.90%)
- Regarding the number of cysts, 79.06% of cases (34 patients) had 2 cysts, 9.30% (4 patients) presented 3 cysts, 9.30% (4 patients) had 4 cysts and 2.33% (1 patient) developed 5 cysts
- Concerning the size of the cysts, 25.58% of cases (11 patients) presented cysts smaller than 2cm; 47% (20 patients) presented at least one cyst between 2cm and 4cm; 11.63% (5 patients) presented at least one cyst larger than 4cm. Data were not available for 16.28% of cases (7 patients).
The anomalies of the teeth adjacent to the cysts and their treatments were as follow:
- 34.88% of cases (15 patients) had at least one resorption of the adjacent teeth
- Only two patients (4.65%) received an endodontic treatment of the resorbed tooth22,29
- Two patients (4.65%) were treated by marsupialization of the cysts with initial conservation of the resorbed teeth39,42, but in one of the cases, the resorbed tooth was removed later.42
- Nine patients presented resorptions of the deciduous teeth; for two patients, we assume that they have got extracted because the authors did not specify the fate of these teeth.25,26
- Teeth with resorption were therefore extracted in 80.0% of cases (12 patients of 15); in 20.0% of the patients (3 patients of 15) they could have been kept, as in the case we reported.
The treatment of cysts consisted of enucleation with the extraction of the impacted teeth for 65.11% of cases (28 patients), while 27.91% (12 patients) were treated with a marsupialisation technique, allowing the evolution of teeth and their eruption spontaneously or after orthodontic traction (in two cases).28,32 We noted that a patient received an orthodontic traction of the impacted tooth after marsupialisation of one of the cysts and enucleation and extraction of the affected teeth on the other side.22 The surgical interventions were performed under general anaesthesia for 41.86% of patients (18 patients of 43) and under local anaesthesia for 13.95% (6 patients of 43) (information was missing for the others).
The management of dental cysts mostly consists in the removal of the impacted teeth during the enucleation of the cysts. The anatomical pieces must be sent for anatomopathological analysis, the main issue of which is the differential diagnosis with ameloblastoma or other tumours. Indeed, Zhang et al.6 have shown that 0.5% of dentigerous cysts can co-exist with other more serious conditions, such as keratocystic odontogenic tumours or cystic ameloblastoma, indicating the importance of histologically confirming any jaw cyst, even when it presents clinically as a classic dentigerous cyst.
When the cysts are large and have proximity to noble anatomical structures or when the impacted teeth are important for occlusion (canines or premolar for example), the marsupialisation technique whether associated or not with orthodontic traction can be considered. In the reported case, the patient had refused the proposed marsupialisation treatment; despite the repression of the lower alveolar nerves by the cysts, no neurological complication was identified in the postoperative period. In the cases included in the literature review, no neurological complications were described despite the large size of some lesions.
In our case, the pulp necrosis of the mandibular second molars can be explained by a possible rupture of the vascular-nerve bundle during cysts enucleation. The literature review showed that, in most cases, the teeth resorbed on contact with the dentigerous cysts were extracted. In the presented case, despite the pulp necrosis of the mandibular second molars and the difficulty to obtain root canals drying for 4 months, drying required for the achievement of tight apical fillings, these teeth were preserved due to the young age of the patient. The enucleation of the cysts associated with the endodontic treatment of the adjacent affected teeth favoured bone repair, complete after 18 months.
In the cases included in the literature review, patients’ follow-up ranged from 10 days to 10 years and showed ongoing or complete mucosal and bone healing (depending on the length of follow-up). No recurrence of dentigerous cysts has been reported in the literature.
CONCLUSION
Multiple non-syndromic dentigerous cysts are rare in healthy patients. The literature review showed that they are more likely to affect young male patients and they are more frequently related to mandibular second premolars, maxillary canines and mandibular impacted third molars. Its discovery is mostly either fortuitous or caused by infectious complications. Adjacent teeth, even if they have significant resorptions, should be preserved whenever possible, but the endodontic treatment of these teeth was rarely attempted in the cases reported in the literature. Enucleation is the most frequently performed surgical treatment. Regular monitoring helps to control bone healing and to treat potential complications. Collaboration across different specialties is essential for the treatment of large lesions. Furthermore, systematic clinical examinations should be performed to rule out any associated syndrome.
CONFLICT OF INTEREST
The authors declare that they have no conflict of interest to declare. This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.
REFERENCES
1. Bilodeau EA, Collins BM. Odontogenic Cysts and Neoplasms. Surg Pathol Clin 2017; 10: 177-222. https://doi.org/10.1016/j.path.2016.10.006.
2. Brown LH, Berkman S, Cohen D, Kaplan AL, Rosenberg M. A radiological study of the frequency and distribution of impacted teeth. J Dent Assoc South Afr Tydskr Van Tandheelkd Ver Van Suid-Afr 1982; 37: 627-630.
3. Daley TD, Wysocki GP, Pringle GA. Relative incidence of odontogenic tumors and oral and jaw cysts in a Canadian population. Oral Surg Oral Med Oral Pathol 1994; 77: 276-280. https://doi.org/10.1016/0030-4220(94)90299-2.
4. Shear M, Speight PM. Cysts of the oral and maxillofacial regions. 4th ed., New Jersey: Blackwell Pub, Oxford; 2007: p. 59-75.
5. De Biase A, Ottolenghi L, Polimeni A, Benvenuto A, Lubrano R, Magliocca FM. Bilateral mandibular cysts associated with cyclosporine use: a case report. Pediatr Nephrol 2001; 16: 993-995. https://doi.org/10.1007/s004670100011.
6. Zhang LL, Yang R, Zhang L, Li W, MacDonald-Jankowski D, Poh CF. Dentigerous cyst: a retrospective clinicopathological analysis of 2082 dentigerous cysts in British Columbia, Canada. Int J Oral Maxillofac Surg 2010; 39: 878–882. https://doi.org/10.1016/j.ijom.2010.04.048.
7. Vasiapphan H, Christopher PJ, Kengasubbiah S, Shenoy V, Kumar S, Paranthaman A. bilateral dentigerous cyst in impacted mandibular third molars: a case report. Cureus 2018. https://doi.org/10.7759/cureus.3691.
8. Cura N, Hanttash A, Inceoglu B, Orhan K, Oncul AMT. Dentigerous cysts in four quadrants of a nonsyndromic patient: case report and literature review. Oral Radiol 2015; 31: 49-58. https://doi.org/10.1007/s11282-014-0171-7.
9. Roberts MW, Barton NW, Constantopoulos G, Butler DP, Donahue AH. Occurrence of multiple dentigerous cysts in a patient with the Maroteaux-Lamy syndrome (mucopolysaccharidosis, type VI). Oral Surg Oral Med Oral Pathol 1984; 58: 169-175. https://doi.org/10.1016/0030-4220(84)90133-6.
10. Trimble LD, West RA, McNeill RW. Cleidocranial Dysplasia: comprehensive treatment of the dentofacial abnormalities. J Am Dent Assoc 1982; 105: 661-666. https://doi.org/10.14219/jada.archive.1982.0450.
11. Aher V, Ali F, Chander P, Chikkalingaiah R. Dentigerous cysts in four quadrants: A rare and first reported case. J Surg Tech Case Rep 2013; 5: 21. https://doi.org/10.4103/2006-8808.118607.
12. Akay C, Kaya M. Treatment of nonsyndromic dentigerous cysts in primary dentition. Clin Cosmet Investig Dent 2011; 17. https://doi.org/10.2147/CCIDEN.S17091.
13. AlKhudair B, AlKhatib A, AlAzzeh G, AlMomen A. Bilateral dentigerous cysts and ectopic teeth in the maxillary sinuses: A case report and literature review. Int J Surg Case Rep 2019; 55: 117-120. https://doi.org/10.1016/j.ijscr.2019.01.012.
14. Iyengar AR, Vasudev SB, Fatima R, Karibasappa GG, Patil S. Bilateral dentigerous cyst involving mandibular first molars- a case report. Int J Med Sci Clin Invent 2017; 4: 2727-2730.
15. Cury SE, Cury MD, Cury SE, Pontes FS, Pontes HA, Rodini C, et al. Bilateral dentigerous cyst in a nonsyndromic patient: case report and literature review. J Dent Child (Chic) 2009; 76: 92-96.
16. Devi P, Thimmarasa VB, Mehrotra V, Agarwal M. Multiple dentigerous cysts: a case report and review. J Maxillofac Oral Surg 2015; 14: 47-51. https://doi.org/10.1007/s12663-011-0280-3.
17. Dhupar A, Yadav S, Dhupar V, Mittal HC, Malik S, Rana P. Bi-maxillary dentigerous cyst in a non-syndromic child – review of literature with a case presentation. J Stomatol Oral Maxillofac Surg 2017; 118: 45-48. https://doi.org/10.1016/j.jormas.2016.12.001.
18. Gogula S, Nagaraj T, Sumana CK, Nigam H. Bilateral dentigerous cyst – A case report. J Med Radiol Pathol Surg 2018; 5: 9-11.
19. Grewal H, Batra. Non syndromic bilateral dentigerous cysts- a case report. Int J Dent Clinics, North America 2010; 2. Available at: <http://intjdc.com/index.php/intjdc/article/view/42>. Date accessed: 04 Apr. 2019.
20. Hansford JT, Kelsch RD, Wiltz M. Multiple Radiolucencies of the Jaws in a 6 year old. NY State Dent J 2015; 81: 40-43.
21. Imada TSN, Neto VT, Bernini GF, Silva Santos PS, Rubira-Bullen IRF, Bravo-Calderòn D, et al. Unusual bilateral dentigerous cysts in a nonsyndromic patient assessed by cone beam computed tomography. Contemp Clin Dent 2014; 5: 240-242. https://doi.org/10.4103/0976-237X.132366.
22. Ishihara Y, Kamioka H, Takano-Yamamoto T, Yamashiro T. Patient with nonsyndromic bilateral and multiple impacted teeth and dentigerous cysts. Am J Orthod Dentofac Orthop 2012; 141: 228-241. https://doi.org/10.1016/j.ajodo.2010.02.043.
23. Jeon J-Y, Park C-J, Cho SH, Hwang K-G. Bilateral dentigerous cysts that involve all four dental quadrants: a case report and literature review. J Korean Assoc Oral Maxillofac Surg 2016; 42: 123. https://doi.org/10.5125/jkaoms.2016.42.2.123.
24. Kannan N, Patil R, Sreenivasalu P. Bilateral maxillary dentigerous cysts a case report. Int J Dent Clin 2010; 2: 28-30.
25. Kanth S, Rajkumar K, Krishnana R, Kumar AR, Malini R, Nandhini G, Cherian E. Bilateral dentigerous cyst in a nine year old child. SRM Univ J Dent Sci 2011; 2: 76-78.
26. Kaushik A, Chaudhry A, Saluja P, Kumar M, Varshney M. Non-syndromic bilateral dentigerous cysts of maxillary and mandibular canines: A case series and review of literature. J Oral Maxillofac Surg Med Pathol 2015; 27: 562-566. https://doi.org/10.1016/j.ajoms.2014.09.008.
27. Khandeparker RV, Khandeparker PV, Virginkar A, Savant K. Bilateral maxillary dentigerous cysts in a nonsyndromic child: a rare presentation and review of the literature. Case Rep Dent 2018; 2018: 1-6. https://doi.org/10.1155/2018/7583082.
28. Reddy PM, Swamy KVN, Tavane PN, Malu K, Gupta PK. Non Syndromic bilateral dentigerous cysts: An unusual case report. Int J Clin Dent Sci 2011; 2: 74-76.
29. Mehdizadeh M, Hajisadeghi S, Lotfi A. Bilateral dentigerous cysts in a non-syndromic patient: literature review and report of a case. J Islam Dent Assoc Iran 2019; 31: 57-63. https://doi.org/10.30699/jidai.31.1.8.
30. Morais HHA de, Dias TG de S, Vasconcellos RJ de H, Vasconcelos BC do E, Melo AR, Gondim DA, et al. Bilateral mandibular dentigerous cysts: a case report. RGO – Rev Gaúcha Odontol 2014; 62: 299-304. https://doi.org/10.1590/1981-8637201400030000010641.
31. Moturi K, Kaila V. Management of non-syndromic multiple impacted teeth with dentigerous cysts: a case report. Cureus 2018. https://doi.org/10.7759/cureus.3323.
32. Pant B, Carvalho K, Dhupar A, Spadigam A. Bilateral nonsyndromic dentigerous cyst in a 10-year-old child: a case report and literature review. Int J Appl Basic Med Res 2019; 9: 58-61. https://doi.org/10.4103/ijabmr.IJABMR_205_18.
33. Prabhakar V, Sandhu SV. Nonsyndromic bilateral maxillary dentigerous cysts: Review of literature and report of an unusual case. Int J Pediatr Otorhinolaryngol Extra 2011; 6: 5-8. https://doi.org/10.1016/j.pedex.2009.11.003.
34. Prasad LK, Chakravarthi PS, Sridhar M, Ramakumar Y, Kattimani V. Nonsyndromic bilateral maxillary and unilateral mandibular multiple dentigerous cysts in a young girl: report of a rare case. Int J Clin Pediatr Dent 2010; 3: 219-223. https://doi.org/10.5005/jp-journals-10005-1082i.
35. Saluja J, Ramakrishnan M, Vinit G, Jaiswara C. Multiple dentigerous cysts in a nonsyndromic minor patient: Report of an unusual case. Natl J Maxillofac Surg 2010; 1: 168. https://doi.org/10.4103/0975-5950.79223.
36. Sanjay CJ, David CM, Kaul R, Shilpa PS. Kissing dentigerous cysts involving mandibular canines: report of unusual case with review of literature. J Calif Dent Assoc 2015; 43: 29-33.
37. Hiremath SS, Charantimath S, Byakodi R, Hiremath SS. Non-syndromic bilateral dentigerous cysts in a 7-year-old child: Report of a rare case. Int J Contemp Dent 2011; 2: 101104.
38. Sharma S, Chauhan JS. Bilateral ectopic third molars in maxillary sinus associated with dentigerous cyst—A rare case report. Int J Surg Case Rep 2019; 61: 298-301. https://doi.org/10.1016/j.ijscr.2019.07.072.
39. Shirazian S, Agha-Hosseini F. Non-syndromic bilateral dentigerous cysts associated with permanent second premolars. Clin Pract 2011; 1: 64. https://doi.org/10.4081/cp.2011.e64.
40. Sindi AM. Bilateral mandibular dentigerous cysts presenting as an incidental finding: a case report. Am J Case Rep 2019; 20: 1148-1151. https://doi.org/10.12659/AJCR.917943.
41. Tamgadge A, Tamgadge S, Bhatt D, Bhalerao S, Pereira T, Padhye M. Bilateral dentigerous cyst in a non-syndromic patient: Report of an unusual case with review of the literature. J Oral Maxillofac Pathol 2011; 15: 91. https://doi.org/10.4103/0973-029X.80017.
42. Tikekar S, Degwekar S, Bhowate R. Bilateral dentigerous cyst: an unusual case report and review of literature. J Indian Acad Oral Med Radiol 2010; 22: 116-118.
43. Vassiliou L-V, Lahiri S, Matthews SN. Non-syndromic bilateral dentigerous cysts with significant root resorption: a case report: Non-syndromic bilateral dentigerous cysts. Oral Surg 2015; 8: 59-62. https://doi.org/10.1111/ors.12121.
44. Yonel Z, McIntosh AP, Donaldson N, Murphy M, Taneja P. Bilateral dentigerous cysts: an updated literature review and report of a case with associated root resorption. Dent Update 2018; 45: 1063-1082. https://doi.org/10.12968/denu.2018.45.11.1063.
ABOUT THIS PAPER
Manuscript submitted: 7 July 2020
Manuscript accepted: 24 November 2020
DOI: https://doi.org/10.36161/FJDM.0007
