Oral

Alternative treatments for a traumatic : A longitudinal case report Tracy M. Dellinger, DDS*/Ray Holder, MS, DMD**/H. Mark Livingstoti, Willie J, Hill, DDS****

A patient presented with a large, inultilocidar, refractory traumatic bone cyst. The radiolucency had in- creased in dimension since her last recall. Over ¡ I years, therapy had included needle aspiration biop- sies followed by simple curettage and closure, the most common therapy for traumatic bone cysts. However, all treatment had proved unsuccessful for this patient. It was decided to treat tiie patient with a slightly unique method. After curettage of the iesiiin, the traumatic bone cy.îf was packed with a mixmie of autogenous blood, harvested autogenous bone diips, and hydroxyapatite. (Quintessence Int I998-29- 497-502)

Key words: altemative treatments, curettage, florrid osseous dysplasia. hard tissue lesions, traumatic bone cyst

raumatic bone cysts were first noted in the literature rarely displacement of the involved dentition or expan- Tin 1929 but were considered quite rare until com- sion of the involved cortical plate in this condition.'' The mon use of panoramic radiographs demonstrated an in- lesion does not affect the vitality of the involved denti- creased frequency of this benign lesion.'- Traumatic tion.' However, an interesting patient subset was identi- bone cysts are normally associated with the posterior fied: There is a tendency for traumatic hone cysts to but on rare occasion involve the maxilla.- present at multiple sites in older black women.* Ad- Patients are usually in their second decade; males ex- ditionally, the simultaneous occurrence of simple bone hibit a slightly higher predilection than females. The pa- cysts and florid osseous dysplasia has heen reported.^' tient usually presents with a history of trauma to the The term traumatic bone cyst is a misnomer hecause . Radiographie examination reveáis a unilocular or no epithelial lining is associated with the lesion.^ More niultilocular, well-delineated radioliicency with scal- appropriately, it might be referred to as an idiopathic loped margins that extend between the roots of teeth.^ bone cavity, but the terms solitary bone cyst, hemor- The size and shape of the lesion may vary. There is rhagic cyst, extravasation cyst, unicameral bone cyst. and simple bone cyst have all been used to describe this phenomenon/ Diagnosis cannot truly he established •Denial Research E-"eI)ow, Deparimeni of Veterans Affairs, Jackson. until the lesion has been exposed by surgical access, Mississippi. whereupon usual findings include the lack of an epithe- **Assatiale Professor and Director, Advanced Education in Dentistry lial lining, walls of histologically normal bone sur- Residency, University of Mississippi, School of Denlistry, Jackson. rounding the cavity, and a space commonly devoid of Mississippi. any visible contents except for a minimal amount of '•'Assistant Professor, Department of Restorative Dentislry, University of Mississippi. School of Denlistry, Jackson, Mississippi. variously colored fluid.^'' •••••Professor and Chairman, Department of Oral and Maxillofacial The etiology of the traumatic bone cyst is unknown Surgery, University of Mississippi, School of Dentistry, Jackson, but may be related to trauma.'' Heubner and Turlington- Mississippi. delineated the following probable causes: (1) trauma, Reprint requesis; Dr Tracy M. Deilinger, University of Mississippi, resulting in intramedullary hemorrhage; (2) infarction School of Dentislry, CDRC Room D-202, 2500 North State Street. Jackson. Mississippi 39215. E-mail: den[al96@aoLcom of bone marrow or cancellous bone; <3) loss of the This article is a work of Ihe US governmeni and may he reprinted without blood supply of" a hemangioma. a lymphangioma, or an permission. The opinions or assertions contained herein are private views angiomatous cyst; (4) cystic degeneration of tumors; (5) of the authors and are not to be conslrued as official or as reflecting the blockade of osteogenic activity; (6) cystic areas of focal views of the Department of Veterans Affairs or any other department or agency of the US government. infection that result from their being "walled-off ' and

Ouintessence International 497 Deilinger et al

Fig 1 A mixed radiopaque-radioiuoent iesion is associated with Fig 2 A definitive, multilocular mixed lesion extends from the dis- the mandibuiar teeth, from the iett second premoiar to the right tal aspect of the left second premolar to the right first premolar canine (1984). (1990].

graduaily imbibing tluid; and (7) faulty caicium nietab- mandibular teeth all tested vital utilizing electric pulp oiism. Unfortunately, research has .still not pinpointed tester (EPT), coid, and heat. The oral surgery and oral the mechanism for traumatic hone cyst formation. pathology attending faculty were then consulted, and Although traumatic hone cysts are benign, resolution of the patient was rescheduled for biopsy. At this time, the the lesion is preferred. differentiai diagnosis included periapical cémentai dys- In the foilowing case, a patient presented to the clinic plasia. fiorid osseous dyspiasia, fibrous dysplasia, cen- with a large, multilocular traumatic bone cyst. This case trai ossifying or cementifying fihroma. calcifying ep- is quite informative because serial examinations and itheiial (Gorlins cyst}, calcifying radiographs aiiowed a longitudinal evaluation of the epithelial (Pindborg tumor), odonto- progression ofa traumatic bone cyst.'" genic keratocyst. and centrai giant-cell granuloma.'** The patient presented for biopsy of the lesion. A Case report small amount of straw-coiored exúdate was aspirated; it had a negative culture. A mucogingival flap was re- A 51-year-old black woman first came to the University flected, and access to the lesion was made through the of Mississippi School of Dentistry in 1984 for treatment thin cortical bone. The cavily was hollow, held a small planning and oral heaith care. The only .significant med- amount of straw-colored fluid, and had bony walls. The ical history included medically treated hypertension and iesion was curetted on both sides of the mandibular hormone replacement therapy for her postmenopausai midline, and the gingival fiap was ciosed. It was deter- status. At this time, a panoramic radiograph was taken mined on exarnination that the lesion was a traumafic (Fig I): however, no notation was made concerning the bone cyst and idiopathic bone cavity (Fig 3). The pa- mixed radiolucent-radiopaque area in the mandibie. She tient was released without complications with a pre- was then treated with periodontal therapy, restorafions, scription for antibiotics and pain medication. Sutures maxillary fixed partial dentures, muitiple extractions, were removed 1 week later, and the pafient's soft tissue and a mandibular removable partial denture. An oral hy- was healing without compiication. giene recall protocol was recommended. The pafient was recalled 6 months later; a panoramic The pafient decided !o seek outside dental care for radiograph revealed some radiopaque fill in the area the next 3 years. She returned to the clinic in 1990 with (Fig 4). The patient was monitored for 2 years, and the compiaint of paresthesia on the left side of the there was no additionai bone formation in the defect mandible following extraction of her mandibular left (Fig 5). In fact, the radiolucency had extended its bor- second molar by a private dentist. The clinical examina- ders along the right mandihie to include the region be- fion showed the mandible to be enlarged in the area of tween the left second premolar and right second premo- the left alveolar ridge. A panoramic radiogtaph was then lar. It was decided to reenter the bone cavity in 1993 taken, and a large multilocular mixed iesion involving and the same procedure and protocols previously dis- all mandibular anterior teeth was identified (Fig 2). The cussed were followed.

498 Volume 29, Ni.imber 8, 199S Deilinger et ai

Fig 3 in 1991 the lesion was accessed and diagnosed as a trau- Fig 4 From 1991 to 1993, the patient's traumatic bone cyst was matic bone cyst. The lesion consists of a hoiiow cavity surrounded monitored on a 6-month recaii system. by bony walis. No epitheiial iining is present. A small amount of straw-coiored fiuid was aspirated.

Fig 5 An additionai radiograph reveáis increased radiopacity on Fig G The mandibular anterior lesion has increased in both di- the ieft borde' oí the iesion and the continued focal areas of mension and radiolucency ¡1996). At this time, alternative therapy radiopacity within the lesion, most significantiy from the left iaterai was pianned. incisor to the right first premolar (1991).

The patient continued to attend her recall oral hy- that a more aggressive therapy than the conventional giene appointments. In 1996, the patient complained of curettage would be used to treat the lesion after a final continued pare.sthesia in the left mandihie. The patient biopsy confirmed that the lesion was, as originally diag- maintained excellent oral hygiene and had minimal nosed, a traumatic bone cyst. plaque and calculus accumulation. Also, during prophy- The patient was brought to the oral surgery clinic. A laxis, the hygienist noted moderate mohility gen- small amount of straw-colored exúdate, which con- eralized throughout the mandibular dentition. The hy- tained white and red blood cells, was withdrawn; how- gienist brought the patient to the graduate program for ever, the cavity appeared to he hollow. After a mucogin- evaluation. gival flap was reflected to reveal the huccal surface of Oral examination verified expansion of the buccal the anterior mandible, a small window was placed plate of the anterior mandible. All mandibular teeth through the thin cortical plate to gain access to the le- demonstrated +2 mobility and tested vital (utilizing sion. The lesion did not appear to have any type of epi- EPT, cold, and heat stimuli). A panoramic radiograph thelial lining, only thin hony margins. An interesting, (Fig 6) and assorted periapical radiographs were taken. but expected, finding was that the mandihular canal was The radiographs showed the radiolucent area depicted not contained within the traumatic hone cyst. The infe- in Fig 4. It was determined and explained to the patient rior alveolar nerve was intact but was more lingually

Quintessence international 499 Dellinger et al

Fig 7 The radiopaque mixture placed during oral suigery con- Fig 8 A 6-month pos'.optjra'.ije panoramic radiograph reveáis tains both autogenous har^iested bone chips and h yd roxy apatite generaiized increased radiopacity, less delined borders, and re- mixed with autogenous blood (1997) turn of trabeculation within the iesiûn site.

positioned than expected, possibly accounting for the and cheek, which was confirmed by pain stimuli; paresthesia the patient had been experiencing in her left however, sensation remained altered. The mucogingival mandible. After curettage, the lesion was closed, and the flap had healed within normal parameters, and the su- patient was given prescriptions for antibiotics and pain tures were retnoved. A 0.12% chlorhexidine regimen medication. was prescrihed. The patient was asked to return in 6 to The patient returned to the clinic 1 week later for su- S weeks for radiographie evaluation of the traumatic ture removal. At this time, it was discovered that the en- hone cyst. tire mandibular anterior .segment that was involved with Six months after the surgical treatment of the bone the lesion had depressed the involved teeth approxi- cyst, the patient reported back to the dental clinic for a mately 1.5 mm, further demonstrating that the thin re- follow-up evaluation and recall visit. She reported con- maining bone was not able to distribute stress in the af- tinued improved sensation in her left cheek and lower fected load-h earing regions. lip, which she referred to as "'feeling normal." Intraoral The patient was administered a local anesthetic. A examination revealed a well-healed surgical site with- mucogingival flap across the mandibular anterior mid- out abnormalities. Probing depths had remained un- line was reflected. A window was made by inserting an changed since the last visit; however, mobility had osteotome into the lesion; this window was enlarged uti- greatly decreased and only two teeth exhibited greater lizing rongeurs to gain access to all bony margins of the than normal movement. A panoramic radiograph re- traumatic bone cyst. The cavity was then thoroughly vealed an increased radiopacity within the lesion site, curetted. Most of the hone fragments were left within less defined borders of the traumatic bone cyst, sfight the cavity. However, a few fragments were removed and diffusion of the hydroxyapaiite-blood mixture within placed aside for later use. the cavity, and return of defined landmarks within the Approximately 5 mL of whole blood was withdrawn area (mainly the mandibular canals and tnental fora- from the patient's right antecuhital region. The hlood men) (Fig 8). was mixed with 6 mL of hydroxyapatite and the hone fragments that had heen set aside earlier. This mixture Discussion was gently injected into the mandibular cavity to slight excess. The mucogingival flap wa.s replaced, and inter- Following the ftrst mandibular biopsy of the patient in rupted silk sutures were used for primary closure. A 1990, confirming the diagnosis of a traumatic bone cyst, panoramic radiograph confirmed the piacement of the florid osseous dysplasia (FOD) remained a possihle un- injected mixture in the hone cavity (Fig 7). The patient derlying condition that required further considerafion. was prescribed a 7-day course of 5Û0 rng penicillin VK, Florid osseous dysplasia, also identified as focal taken every 6 hours, to prevent postoperative infection. cemento-osseous-dysplasia. is a condition characterized The patient returned for postoperative evaluation 1 by a primary osteolytic phase, typified by a radiolucent week following the oral surgery. The patient stated that radiographie appearance, and then followed by a mixed she could detect additional sensation in her left lower radiolucent and opaque lesion of the jaws." Florid

500 Volume 29, Number ! Dellinger et al osseous dysplasia is usually located in the mandibular placement of freeze-dried homographs of caneellous anterior region when identified in its target population, bone chips with traditional curettage and closure,'' the hlack women over the age of 40 years,'" placement of hydroxyapatite in the lesion after curet- In 1984. the patient first presented with the nnixed le- tage.'" packing the cyst cavity with a thrombin- sion in the anterior portion of her mandible. However, penicillin-impregnated Gelfoam," or the injection of a longitudinal panoramic films revealed that the lesion small amount of autogenous hlood into the cyst cavity nol only increased dramatically in the radiolucent com- during traditional therapy, as discussed by Precious and ponent but also expanded in size. A second difference is McFadden.' All of or each of these therapies led to suc- the contents; this lesion contained a straw-colored exú- cessful resolution of their respective lesions, which date, whereas the content.^ of an FOD lesion typically were refractory to conventional therapy. It was decided include a fibrous stroma, irregular hony traheculac, and to perform a comhination of some of the aforemen- cementumlike material.'" tioned techniques for this aggressive hard tissue lesion. Various authors have reported traumatic bone cysts Whole blood contains the progenitor cells necessary associated with FOD but all mention that this is a rare for osteogenesis.'" An injection into the bone defect of finding.'-'-'^ These studies suggest that simple bone atJtogenous blood mixed witb bone cbips from tbe cysts associated with FOD may have a different etiol- curettage of a bone cyst provides osteoinductive ogy than bone cysts not associated with this ahnormal- propetlies to instigate osteoblast activity so tbat bone ity_7,12.13 One theory suggests that local bone destruction formation ultimately occurs within the cavity. Hydroxy- within the traumatic bone cyst may be secondary to ob- apatite. a bioactive ceramic, bas been reported as an ef- struction of interstitial fluid drainage, thus leading to fective osteoconductive impetus tbat encourages new cyst formation.'- bone formation witbin tbe cavity."''''--^ Additionally, By itself, FOD is a benign condition rarely requiring hydroxyapatite has been mixed with autogenous bone surgical intervention because of its self-limiting nature; graft to furtber ensure osteogenic activity within bone however, traumatic bone cysts are commonly treated defects. '" surgically because of their tendency to expand and cause pathologic fractures.^" In addition, among 34 pa- Conclusion tients with florid osseous dysplasia. traumatic bone cysts were found in 14; three of the patients attained It was theorized that the combination of the osteocon- only partial resolution of the cysts following conven- ductive forces of hydroxyapatite and the osteoinductive tional surgical curettage.'-''' abilities of autogenous blood and bone would provide It is important that the pathogenesis of a solitary an optimal environment for bone regeneration. Initial bone cyst, either unicameral or traumatic bone cy,sts, be results seem promising. The bone density increased clarified for its treatment to be effective; however, the within the site of the traumatic bone cyst, and the denti- pathogenesis is stiil unknown and speculative.'^ The tion exhibited decreased mobility. most common therapy for bone cysts con,sists of simple Although there has not been any scientific evidence curettage and closure, which induces hemorrhage into stibstantiating the patient's anecdotal claim of increased the defect. Osteohlastic activity typically initiates sensation in tbe lower lip to be associated with the reso- mesynchymal cells found in the autogenous blood to lution of the traumatic bone cyst, it is possible that the differentiate into cells with osteogenic potential.' lack of bony support surrounding the inferior alveolar Complete resolution of the defect via bony regeneration nerve did cause some altered sensation. Along the same usually occurs within a short period following curet- reasoning, it is possihle that the added bone formation tage.'-* This therapy had proved unsuccessful for this relieved mechanical stress. Possible etiologies of me- patient, as demonstrated by the results from the proce- chanical stress include impingement of the inferior alve- dures performed in 1990 and 1993. Although some olar nerve and occlusion of the associated vessels, re- osteoblastic activity may have been initiated by the sulting in ischemia to the distal portion of the nerve. It conventional therapy, resolution of the lesion was is possihle that once mechanical support to the nerve incomplete. and associated vessels was returned, normal conduction To date, there have been several documented cases of sensations resumed. of "suspected recurrence" of traumatic bone cysts fol- The patient will be monitored every 6 months to fol- lowing curettage; however, the lesions were actually re- low the progression of healing. Recurrence of the fractory to resolution via the traditional surgical ther- traumatic bone cyst is not expected; however, con- apy.'"'After traditional bone cyst therapy has proved sidering the patient's histor>', periodic oral evaluation is unsuccessful, other treatment options have included the warranted.

Quinlessence Intetnalioral 501 Deilingeret al

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502 Volume 29, Numbere, 1998