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 Table of Contents  
CASE REPORT
Year : 2014  |  Volume : 26  |  Issue : 3  |  Page : 342-346

Solitary bone cyst coexistent with tooth agenesis: Unique case report and brief review


1 Department of Oral Medicine and Radiology, Awadh Dental College and Hospital, Jamshedpur, Jharkhand, India
2 Departments of Maxillofacial Pathology and Microbiology, Panineeya Mahavidyalaya Institute of Dental Sciences and Research Centre, Hyderabad, Telangana, India
3 Departments of Orthodontics and Dentofacial Orthopedics, Panineeya Mahavidyalaya Institute of Dental Sciences and Research Centre, Hyderabad, Telangana, India

Date of Submission07-Nov-2014
Date of Acceptance12-Nov-2014
Date of Web Publication19-Nov-2014

Correspondence Address:
Ashalata Gannepalli
Professor, Department of Maxillofacial Pathology and Microbiology, Panineeya Mahavidyalaya Institute of Dental Sciences and Research Centre, Chaitanyapuri - 500 060, Hyderabad
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0972-1363.145024

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   Abstract 

Solitary bone cysts (SBCs) exist generally as asymptomatic solitary lesions and have no epithelial lining. They are uncommon in the jaws and are often polymorphic, show scalloped borders when located between the roots. They have higher predilection for the posterior mandible, mostly occurring near the mental foramen. Various pathomechanisms were proposed, but the triggering factor still remains to be ascertained. Currently, osteolytic pathogenesis is speculated, which may be due to vascular alteration leading to aseptic osseous necrosis. Fibro-osseous lesions are occasionally associated with SBCs, but concomitant presence with tooth agenesis has not been reported. We present a unique case of SBC with familial tooth agenesis in a 16-year-old female patient with a brief review.

Keywords: Solitary bone cyst, tooth agenesis, unilocular


How to cite this article:
Podduturi SR, Gannepalli A, Ayinampudi BK, Ksheerasagara P. Solitary bone cyst coexistent with tooth agenesis: Unique case report and brief review . J Indian Acad Oral Med Radiol 2014;26:342-6

How to cite this URL:
Podduturi SR, Gannepalli A, Ayinampudi BK, Ksheerasagara P. Solitary bone cyst coexistent with tooth agenesis: Unique case report and brief review . J Indian Acad Oral Med Radiol [serial online] 2014 [cited 2022 May 22];26:342-6. Available from: https://www.jiaomr.in/text.asp?2014/26/3/342/145024


   Introduction Top


Solitary bone cyst (SBC) was first described by Lucas and Blum in 1929 and later defined by Rushton in 1946 as a single cyst that has no epithelial lining, has an intact bony wall, is fluid filled, and has no evidence of acute or chronic inflammation. [1],[2] Various other terms were given by many authors, such as traumatic bone cyst, progressive bone cyst, hemorrhagic bone cyst, unicameral bone cyst, idiopathic bone cyst, and simple cyst. The numerous synonyms referring to these lesions reflect their uncertain nature. The term "traumatic bone cyst" has been recognized as a misnomer in that the incidence of prior trauma in patients with this entity is the same as in the general population. [3],[4]

Solitary bone cysts are asymptomatic and exist as unilocular solitary lesions. In the jaws, they are often polymorphic and show scalloped borders when located between the roots. Solitary bone cysts are occasionally associated with fibro-osseous lesions - Fibrous dysplasia, focal cement-osseous dysplasia, and florid osseous dysplasia; but a few associations are rare, i.e., central odontogenic fibroma (WHO type), ameloblastoma, and osteogenesis imperfecta. [5],[6],[7],[8],[9],[10] Concomitant presence of SBC and tooth agenesis has not been reported till date in the literature. We present a unique case of SBC coexistent with tooth agenesis, along with a brief account of the different pathomechanisms.


   Case Report Top


A 16-year-old female patient reported to us with the complaint of malaligned and deformed lower front teeth. She also complained of spacing in between her upper and lower front teeth. The patient was completely asymptomatic with no pain and discomfort. The medical history was not significant and there was no history of trauma. Family history revealed missing permanent mandibular central incisors in her father and brother. The findings of her extraoral clinical examination were noncontributory and unremarkable, with no asymmetry and lymphadenopathy. The intraoral examination revealed missing 12, 31, and 41, retained 53, 71, and 81, and peg-shaped 22. There was severe attrition in 71 and 81 with the orifices of the pulp canals clearly visible [Figure 1]. There were diastemas in between the upper anteriors. The oral hygiene status was good with no evidence of stains, calculus, and carious lesions. The vitality of all teeth was confirmed. The other intraoral findings were noncontributory and unremarkable.
Figure 1: Intraoral examination revealing retained deciduous 71 and 81 with no obvious swelling

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The patient was advised an orthopantomograph (OPG) and lateral cephalogram. The OPG revealed a well-defined radiolucency in relation to the apices of 43 and 44, and anodontia of 12, 31, and 41 indicating tooth agenesis. The radiolucency was oval to round and unilocular with well-defined sclerotic borders measuring approximately 1.5 cm in diameter [Figure 2]. In an intraoral periapical (IOPA) radiograph of 43 and 44 region the lamina dura around 43 and 44 was intact. The full superoinferior extent of the lesion, however, was not appreciated in the IOPA radiograph and only the superior part of the radiolucency was visible. For a detailed evaluation of the radiolucency, computed tomography (CT) scan was advised which showed the presence of a unilocular lesion with 1.25 cm diameter [Figure 3]a-d. Based on the clinical and radiographic presentation, an inflammatory lesion was ruled out. Since there was a concomitant presence of tooth agenesis, developmental odontogenic cysts and tumors were considered and a provisional diagnosis of keratocystic odontogenic tumor (KCOT) was made. The differential diagnoses of ameloblastoma, adenomatoid odontogenic tumor, lateral periodontal cyst, focal osteoporotic defect, and SBC were considered. For confirmative diagnosis, the proposed treatment included enucleation followed by histopathological examination.
Figure 2: OPG showing well-defi ned, unilocular periapical radiolucency at the apices of 43 and 44, retained 53, 71, and 81, and agenesis of 12, 31, and 41

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Figure 3: (a) - (d) CT showing unilocular lesion measuring 1.25 cm

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A complete blood and urine analysis was done and all indices were found to be within normal limits. Vestibular osteotomy of the region was done by gingival crevicular incision with anterior vertical release incision, elevating the periosteal flap. The buccal cortical plate was removed using bone drill and an empty intrabony cavity was noticed with thick fluid [Figure 4]. Curettage was done till the entire necrotic bone was removed, provoking fresh hemorrhage, and care was taken to preserve the apical vascularization of 43. Thin delicate cystic lining with few pieces of bone were encountered, which were sent for histopathological examination. The section revealed thin cystic lining constituting delicate band of fibrous connective tissue, eosinophilic exudate, extravasated RBCs, and few neutrophils [Figure 5] and [Figure 6]. The histopathological findings were suggestive of SBC. The postoperative course was uneventful and follow-up after 6 months showed bony filling. The case was planned for orthodontic treatment with fixed mechanotherapy and extraction of 53, 71, and 81 with implant prosthesis.
Figure 4: Surgical exploration revealing empty intrabony cavity

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Figure 5: H and E (40×) image showing delicate, thin lining of the fibrous connective tissue

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Figure 6: H and E (40×) image showing extravasated RBCs with fragments of necrotic bone

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   Discussion Top


Solitary bone cysts are non-epithelial lined cavities occurring in many parts of the skeleton. They are frequently encountered in long bones with 90% incidence, and have an incidence of 5% in the jaw bones. They comprise only 1% of all jaw cysts. These cysts have an equal prevalence in both genders and present at a mean age of 18 years. Solitary bone cyst has a higher predilection for mandible than maxilla and for posterior mandible than the anterior region, mostly occurring near the mental foramen. [3],[11] Few atypical locations such as zygoma and condyle were also reported. [12],[13] As SBCs are asymptomatic, these are detected on routine radiographic examination and are classically located above the mandibular canal as round to oval radiolucencies with well-contoured borders. They are often unicystic, but sometimes have scalloped borders when they involve the roots of the teeth and, very rarely, multicystic cases are also seen with an incidence of 11%. Usually they are unilateral, but bilateral multifocal presence has also been documented. [3],[11],[14]

The present case is a 16-year-old female with clinical presentation in accordance with the literature, with no pathognomic clinical behavior and radiographic features. Hence, based on the gender predominance, site predilection, and vitality of teeth, the SBC was distinguished from focal osteoporotic bone marrow defect, inflammatory cysts, odontogenic cysts, and neoplasms in the present case. The lamina dura and vitality of 43 was preserved, ruling out an inflammatory lesion in the present case. Solitary bone cyst may have a similar appearance as KCOT, as it tends to grow along the bone with destruction of cancellous bone without much expansion. Since there was coexistent tooth agenesis, enucleation was recommended to rule out any aggressive lesion.

The most common etiologic factor evaluated is history of trauma. The histogenesis and origin of the tissue is still speculative, but a myriad of different pathomechanisms have been proposed. Traumatism was proposed by Blum in the year 1932 and was the most widely accepted hypothesis based on the occurrence of an intramedullary hemorrhage followed by hematoma after a trauma. [15] Many authors have questioned this mechanism, given that often there was no history of trauma in patients with SBC. [3],[14] Cohen proposed that the formation and existence of this cyst is due to blockage of the normal draining interstitial fluid, but such a mechanism should show an equal distribution of SBCs in all locations. [16]

The predilection for occurrence in preadolescence and adolescence, and development near osseous remodeling areas, i.e., near the cartilage growth plates of long bones, and mental foramen, which is the preferential zone for SBCs, have led authors to hypothesize a local abnormality of bone growth. Some authors proposed that there is an exuberant out-of-control remodeling process in such areas or an abnormality in osteogenesis and angiogenesis modified by local mechanical factors. [4],[11],[14] Mirra et al. proposed these cysts to be synovial cysts arising from entrapment of small nests of synovium intraosseously during the fetal development which retain some secretory function resulting in cystification. [17] But lack of fluid content in many SBCs and no evidence of synovial lining are against this theory.

Tumor degeneration was another mechanism put forth as SBCs were reported in fibrous dysplasia, cemental dysplasia, and central giant cell granuloma (CGCG). Solitary bone cysts in fibro-osseous lesions might be secondary to liquefaction or degeneration leading to microcyst formation or fibro-osseous tissue might be secondary to cavity thought to be a healing process. [5],[14],[18] But the prevalence of CGCG, fibrous dysplasia in the epiphysis and symphysial area is debated. Atypical simple bone cysts with radiopaque lesions were thought to be due to a long-standing cyst which may cause erosion of lamina dura leading to direct exposure of the roots to the cystic cavity, which, in turn, could trigger the production of cementum. [19] Currently, diverse factors and pathomechanisms are speculated leading to aseptic osseous necrosis with cyst formation, but the triggering factor still remains to be ascertained.

Our case was associated with a non-syndromic, genetic type of hypodontia. Agenesis or hypodontia of one or more permanent teeth is the most common congenital malformation in humans, which may be syndromic or non-syndromic. More than 20% of the population lacks one or more third molars and, most commonly, some of the second premolars or upper lateral incisors. The mandibular incisors are rarely affected, with a frequency of 6%. A higher incidence of tooth agenesis is seen in females, with a male to female ratio of 1:1.4. The inherited form could be either an autosomal-dominant, autosomal-recessive, or X-linked trait. For non-syndromic oligodontia, dominant mutations in the transcription factors MSX1 and PAX9 have been described. New researches have reported a higher presence of cancer in the family members of patients with hypodontia. More than 300 genes are involved in odontogenesis, and the genes that control the development of teeth also have important functions in other organs and body systems. [20],[21]

Since there are no cases reported of coexistence of SBC with tooth agenesis, it is difficult to ascertain the genetic role and the plausible reason could be locoregional factor. Whether the classic age group and site of occurrence (in the periapical region between 43 and 44, i.e., near mental foramen) of SBC in the present case, concomitant with agenesis is a coincidental finding or both have a related pathomechanism, needs to be ascertained.

Solitary bone cysts are suggested to be self-healing, but with their wide range of behavior, few cases of recurrences have been reported. [22],[23] It is advisable to confirm the diagnosis with simple exploration and curettage of bony walls, which might promote healing. The healing is proposed to be due to mechanical disruption of the cavity. Alternative treatments such as filling the cavity with autologous blood with bone, hydroxyapatite, and bovine lyophilized bone have also been proposed when conventional management fails. [24] The intralesional injection of methylprednisolone has been described as a treatment modality in long bones, but the mechanism of action of corticosteroid is complex. [25] The diagnosis of SBC relies on identification of empty cavity on surgery, with the presence of clear cystic fluid or an air-filled cavity. Microscopic examination plays a determining role to rule out any aggressive lesion. Histopathological features include no evidence of epithelium and presence of connective tissue cystic lining with or without tissue content, blood, or any cystic fluid. Fibroblasts, osteoclasts, bony trabeculae, congested capillaries, and cholesterol crystals also may be present. [3],[14]


   Conclusion Top


The present case is a rare association of SBC with tooth agenesis, and such rare associations are leading to more speculations regarding its etiopathogenesis. The surgical management has given good postoperative result, and in such cases, it is advisable to rule aggressive lesions.

 
   References Top

1.Lucas CD, Blum T. Do all cysts in the jaws originate from the dental system? J Am Dent Assoc 1929;16:647-61.  Back to cited text no. 1
    
2.Rushton MA. Solitary bone cysts in the mandible. Br Dent J 1946;81:37-49.  Back to cited text no. 2
    
3.Kaugers GE, Cale AE. Traumatic bone cyst. Oral Surg Oral Med Oral Pathol 1987;63:318-24.  Back to cited text no. 3
    
4.Kuhmichel A, Bouloux GF. Multifocal traumatic bone cysts: Case report and current thoughts on etiology. J Oral Maxillofac Surg 2010;68:208-12.  Back to cited text no. 4
    
5.Hara H, Ohishi M, Higuchi Y. Fibrous dysplasia of the mandible associated with large solitary bone cyst. J Oral Maxillofac Surg 1990;48:88-91.  Back to cited text no. 5
    
6.Mupparapu M, Singer SR, Milles M, Rinaggio J. Simultaneous presentation of focal cemento-osseous dysplasia and simple bone cyst of the mandible masquerading as a multilocular radiolucency. Dentomaxillofac Radiol 2005;34:39-43.  Back to cited text no. 6
    
7.Melrose RJ, Abrams AM, Mills BG. Florid osseous dysplasia. A clinical-pathologic study of thirty-four cases. Oral Surg Oral Med Oral Pathol 1976;41:62-82.  Back to cited text no. 7
    
8.Kawahira K, Fujinami Y, Hamasaki E, Kawashima K, Yamashita S. A case of simple bone cyst coexistent with ameloblastoma in the median mandibular region. Jap J Oral Maxillofac Surg 1979;25:157-60.  Back to cited text no. 8
    
9.Pushpanshu K, Kaushik R, Punyani SR, Jasuja V, Raj V, Seshadri A. Concurrent central odontogenic fibroma (WHO Type) and traumatic bone cyst: Report of a rare case. Quant Imaging Med Surg 2013;3:341-6.  Back to cited text no. 9
    
10.Jones AC, Baughman RA. Multiple idiopathic mandibular bone cysts in a patient with osteogenesis imperfecta. Oral Surg Oral Med Oral Pathol 1993;75:333-7.  Back to cited text no. 10
    
11.Harnet JC, Lombardi T, Klewansky P, Rieger J, Tempe MH, Clavert JM. Solitary bone cyst of the jaws: A review of the etiopathogenic hypotheses. J Oral Maxillofac Surg 2008;66:2345-8.  Back to cited text no. 11
    
12.Bradley JC. Solitary bone cyst of the zygomatic bone. Br Dent J 1982;152:203-4.  Back to cited text no. 12
    
13.Kim KA, Koh KJ. Recurrent simple bone cyst of the mandibular condyle: A case report. Imaging Sci Dent 2013;43:49-53.  Back to cited text no. 13
    
14.Jaffe HL, Lichtenstein L. Solitary unicameral bone cyst: With emphasis on the roentgen picture, the pathologic appearance and the pathogenesis. Arch Surg 1942;44:1004-25.  Back to cited text no. 14
    
15.Blum T. Unusual bone cavities in the mandible: A report of three cases of traumatic bone cysts. J Am Dent Assoc 1932;19:281-301.  Back to cited text no. 15
    
16.Cohen J. Etiology of simple bone cyst. J Bone Joint Surg Am 1970;52:1493-7.  Back to cited text no. 16
    
17.Mirra JM, Bernard GW, Bullough PG, Johnston W, Mink G. Cementum-like bone production in solitary bone cysts (so called "cementoma" of long bones). Report of three cases. Electron microscopic observations supporting a synovial origin to the simple bone cyst. Clin Orthop Relat Res 1978;135:295-307.  Back to cited text no. 17
    
18.Fischer AD. Bone cavities in fibro-osseous lesions. Br J Oral Surg 1976;14:120-7.  Back to cited text no. 18
    
19.Saito Y, Hoshina Y, Nagamine T, Nakajima T, Suzuki M, Hayashi T. Simple bone cyst. A clinical and histopathologic study of fifteen cases. Oral Surg Oral Med Oral Pathol 1992;74:487-91.  Back to cited text no. 19
    
20.Vastardis H. The genetics of human tooth agenesis: New discoveries for understanding dental anomalies. Am J Orthod Dentofac Orthop 2000;117:650-6.  Back to cited text no. 20
    
21.Lammi L, Arte S, Somer M, Jarvinen H, Lahermo P, Thesleff I, et al. Mutations in AXIN2 cause familial tooth agenesis and predispose to colorectal cancer. Am J Hum Genet 2004;74:1043-50.  Back to cited text no. 21
    
22.Sapp JP, Stark ML. Self-healing traumatic bone cysts. Oral Surg Oral Med Oral Pathol 1990;69:597-602.  Back to cited text no. 22
    
23.Feinberg SE, Finkelstein MW, Page HL, Dembo JB. Recurrent 'traumatic' bone cysts of the mandible. Oral Surg Oral Med Oral Pathol 1984;57:418-22.  Back to cited text no. 23
    
24.Dellinger TM, Holder R, Livingston HM, Hill WJ. Alternative treatments for a traumatic bone cyst: A longitudinal case report. Quintessence Int 1998;29:497-502.  Back to cited text no. 24
    
25.Wright JG, Yandow S, Donaldson S, Marley L; Simple Bone Cyst Trial Group. A randomized clinical trial comparing intralesional bone marrow and steroid injections for simple bone cysts. J Bone Joint Surg Am 2008;90:722-30.  Back to cited text no. 25
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]



 

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