Aneurysmal Bone Cyst of the Mandible: Report of 3 Cases

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16. Witt C, Borges AC, Klein K, et al: Radiographic manifestations of multiple myeloma in the mandible: A retrospective study of 77 patients. J Oral Maxillofac Surg 55:450, 1997 17. Epstein JB, Voss NJS, Stevenson-Moore P: Maxillofacial man- ifestations of multiple myeloma: An unusual case and review of the literature. Oral Surg Oral Med Oral Pathol 57:267, 1984 18. Furutani M, Ohnishi M, Tanaka Y: Mandibular involvement in patients with multiple myeloma. J Oral Maxillofac Surg 52:23, 1994 19. George ED, Sadovsky R: Multiple myeloma: Recognition and management. Am Fam Physician 59:1885, 1999 20. Zulian GB: Multiple myeloma: Clinical evaluation of plasma cell lymphoproliferative disorders and initial management. Semin Hematol 34:29, 1997 (suppl 1) 21. Baskaran RK, Krishnamoorthy SM, Smith M: Numb chin syn- drome—A reflection of systemic malignancy. World J Surg Oncol 4:52, 2006 22. Iniguez C, Mauri JA, Larrode P, et al: Mandibular neuropathy due to infiltration of the Gasser ganglion. Rev Neurol 25:1092, 1997 23. Thompson A, Pearce I, Walton G, et al: Numb-chin syndrome: An unusual presentation of metastatic prostate cancer. BJU Int 85:377, 2000 24. Lesnick JA, Zallen RD: Numb chin syndrome secondary to metastatic breast disease. J Colo Dent Assoc 78:11, 1999 25. Dispenzieri A, Kyle RA: Neurological aspects of multiple my- eloma and related disorders. Best Pract Res Clin Haematol 18:673, 2005 26. Rosado MF, Morgensztern D, Peleg M, et al: Primary diffuse large cell lymphoma of the mandible. Leuk Lymphoma 45: 1049, 2004 27. Marinella MA: Metastatic large cell lung cancer presenting with numb chin syndrome. Respir Med 91:235, 1997 28. Berenson JR, Lightenstein A, Knight RD, et al: Efficacy of pamidronate in reducing skeletal events in patients with ad- vanced multiple myeloma. N Engl J Med 334:488, 1996 29. Rosen LS, Gordon D, Seaman JJ: Long-term efficacy and safety of zoledronic acid compared with pamidronate disodium in the treatment of skeletal complications in patients with advanced multiple myeloma or breast carcinoma: A random- ized, double-blind, multicenter, comparative trial. Cancer 98:1735, 2003 30. Lacy MQ, Dispenzieri A, Kyle RA: Mayo Clinic consensus state- ment for the use of bisphosphonates in multiple myeloma. Mayo Clin Proc 81:1047, 2006 31. Wahner HW, Kyle RA, Beabout JW: Scintigraphic evaluation of the skeleton in multiple myeloma. Mayo Clin Proc 55:739, 1980 32. Dudeney S, Lieberman IH, Reinhardt MK, et al: Kyphoplasty in the treatment of osteolytic vertebral compression frac- tures as a result of multiple myeloma. J Clin Oncol 20:2382, 2002 33. LeGrand SB, Leskuski D, Zama I: Narrative review: Furosemide for hypercalcemia: An unproven yet common practice. Ann Intern Med 149:259, 2008 J Oral Maxillofac Surg 67:1996-2004, 2009 Aneurysmal Bone Cyst of the Mandible: Report of 3 Cases Ajoy Roychoudhury, MDS,* Ankur Rustagi, BDS,† Krushna Bhatt, BDS,‡ Ongkila Bhutia, MDS,§ and Ashu Seith, MD In 1942 Jaffe and Liechtenstein 1 first discovered an- eurysmal bone cyst (ABC) as a distinct entity when they described 2 cases of a “peculiar blood containing cyst of large size.” Only 1.9% of all ABCs occur in the jaws, representing 1.5% of all nonodontogenic cysts. 2 The term aneurysmal bone cyst was coined to de- scribe the characteristic ballooned appearance of the cortex on the radiograph. The lesion is not a true cyst because it lacks epithelial lining. Preoperative diagno- sis of ABC can be difficult because of similarity to other lesions such as ameloblastoma, giant cell tumor, hyperparathyroidism, myxoma, traumatic bone cyst, and odontogenic keratocyst. Aspiration of blood from the lesion should lead the clinician to suspect a vas- cular lesion or ABC. The absence of bruits, thrill, and lack of pulse pressure help to differentiate ABC from a vascular lesion clinically. It is essentially a diagnosis of exclusion. Definitive diagnosis can be made only after incisional biopsy. Biopsy should be attempted only when a vascular lesion has been ruled out. 3 We report a series of 3 extensive cases of ABC of the mandible (Table 1, Figs 1-3). Received from the All India Institute of Medical Sciences, Delhi, India. *Associate Professor, Department of Oral and Maxillofacial Sur- gery, CDER. †Junior Resident, Department of Oral and Maxillofacial Surgery, CDER. ‡Junior Resident, Department of Oral and Maxillofacial Surgery, CDER. §Assistant Professor, Department of Oral and Maxillofacial Sur- gery, CDER. Associate Professor, Department of Radio Diagnosis. Address correspondence and reprint requests to Dr Roy- choudhury: Department of Oral & Maxillofacial Surgery, All India Institute of Medical Sciences, Ansari Nagar, New Delhi, Delhi 110029, India; e-mail: [email protected] © 2009 American Association of Oral and Maxillofacial Surgeons 0278-2391/09/6709-0029$36.00/0 doi:10.1016/j.joms.2009.04.021 1996 ANEURYSMAL BONE CYST OF MANDIBLE

Transcript of Aneurysmal Bone Cyst of the Mandible: Report of 3 Cases

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1996 ANEURYSMAL BONE CYST OF MANDIBLE

6. Witt C, Borges AC, Klein K, et al: Radiographic manifestationsof multiple myeloma in the mandible: A retrospective study of77 patients. J Oral Maxillofac Surg 55:450, 1997

7. Epstein JB, Voss NJS, Stevenson-Moore P: Maxillofacial man-ifestations of multiple myeloma: An unusual case and reviewof the literature. Oral Surg Oral Med Oral Pathol 57:267,1984

8. Furutani M, Ohnishi M, Tanaka Y: Mandibular involvement inpatients with multiple myeloma. J Oral Maxillofac Surg 52:23,1994

9. George ED, Sadovsky R: Multiple myeloma: Recognition andmanagement. Am Fam Physician 59:1885, 1999

0. Zulian GB: Multiple myeloma: Clinical evaluation of plasma celllymphoproliferative disorders and initial management. SeminHematol 34:29, 1997 (suppl 1)

1. Baskaran RK, Krishnamoorthy SM, Smith M: Numb chin syn-drome—A reflection of systemic malignancy. World J SurgOncol 4:52, 2006

2. Iniguez C, Mauri JA, Larrode P, et al: Mandibular neuropathy dueto infiltration of the Gasser ganglion. Rev Neurol 25:1092, 1997

3. Thompson A, Pearce I, Walton G, et al: Numb-chin syndrome:An unusual presentation of metastatic prostate cancer. BJU Int85:377, 2000

4. Lesnick JA, Zallen RD: Numb chin syndrome secondary tometastatic breast disease. J Colo Dent Assoc 78:11, 1999

5. Dispenzieri A, Kyle RA: Neurological aspects of multiple my-eloma and related disorders. Best Pract Res Clin Haematol

18:673, 2005

Ashu Seith,

hatclaoaorm

ery, CDER.

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6. Rosado MF, Morgensztern D, Peleg M, et al: Primary diffuselarge cell lymphoma of the mandible. Leuk Lymphoma 45:1049, 2004

7. Marinella MA: Metastatic large cell lung cancer presenting withnumb chin syndrome. Respir Med 91:235, 1997

8. Berenson JR, Lightenstein A, Knight RD, et al: Efficacy ofpamidronate in reducing skeletal events in patients with ad-vanced multiple myeloma. N Engl J Med 334:488, 1996

9. Rosen LS, Gordon D, Seaman JJ: Long-term efficacy and safetyof zoledronic acid compared with pamidronate disodium inthe treatment of skeletal complications in patients withadvanced multiple myeloma or breast carcinoma: A random-ized, double-blind, multicenter, comparative trial. Cancer98:1735, 2003

0. Lacy MQ, Dispenzieri A, Kyle RA: Mayo Clinic consensus state-ment for the use of bisphosphonates in multiple myeloma.Mayo Clin Proc 81:1047, 2006

1. Wahner HW, Kyle RA, Beabout JW: Scintigraphic evaluation ofthe skeleton in multiple myeloma. Mayo Clin Proc 55:739,1980

2. Dudeney S, Lieberman IH, Reinhardt MK, et al: Kyphoplastyin the treatment of osteolytic vertebral compression frac-tures as a result of multiple myeloma. J Clin Oncol 20:2382,2002

3. LeGrand SB, Leskuski D, Zama I: Narrative review: Furosemidefor hypercalcemia: An unproven yet common practice. Ann

Intern Med 149:259, 2008

Oral Maxillofac Surg7:1996-2004, 2009

Aneurysmal Bone Cyst of the Mandible:Report of 3 Cases

Ajoy Roychoudhury, MDS,* Ankur Rustagi, BDS,†

Krushna Bhatt, BDS,‡ Ongkila Bhutia, MDS,§ and

MD�

n 1942 Jaffe and Liechtenstein1 first discovered an-urysmal bone cyst (ABC) as a distinct entity whenhey described 2 cases of a “peculiar blood containingyst of large size.” Only 1.9% of all ABCs occur in theaws, representing 1.5% of all nonodontogenic cysts.2

he term aneurysmal bone cyst was coined to de-cribe the characteristic ballooned appearance of theortex on the radiograph. The lesion is not a true cystecause it lacks epithelial lining. Preoperative diagno-is of ABC can be difficult because of similarity tother lesions such as ameloblastoma, giant cell tumor,

eceived from the All India Institute of Medical Sciences, Delhi,

ndia.

*Associate Professor, Department of Oral and Maxillofacial Sur-

ery, CDER.

†Junior Resident, Department of Oral and Maxillofacial Surgery, CDER.

‡Junior Resident, Department of Oral and Maxillofacial Surgery, CDER.

§Assistant Professor, Department of Oral and Maxillofacial Sur-

yperparathyroidism, myxoma, traumatic bone cyst,nd odontogenic keratocyst. Aspiration of blood fromhe lesion should lead the clinician to suspect a vas-ular lesion or ABC. The absence of bruits, thrill, andack of pulse pressure help to differentiate ABC fromvascular lesion clinically. It is essentially a diagnosisf exclusion. Definitive diagnosis can be made onlyfter incisional biopsy. Biopsy should be attemptednly when a vascular lesion has been ruled out.3 Weeport a series of 3 extensive cases of ABC of theandible (Table 1, Figs 1-3).

�Associate Professor, Department of Radio Diagnosis.

Address correspondence and reprint requests to Dr Roy-

houdhury: Department of Oral & Maxillofacial Surgery, All India

nstitute of Medical Sciences, Ansari Nagar, New Delhi, Delhi

10029, India; e-mail: [email protected]

2009 American Association of Oral and Maxillofacial Surgeons

278-2391/09/6709-0029$36.00/0

oi:10.1016/j.joms.2009.04.021

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athologic Features on Microscopy

All the specimens showed sinusoidal spaces filledith red blood cells in a fibrous matrix. Hemosiderin-

aden macrophages and multinucleated osteoclast-likeiant cells were also seen surrounding the sinusoidalpaces (Fig 4).

iscussion

ABC is most common in those parts of the skel-ton where there is a relatively high venous pres-ure and high marrow content.4 The skull bonesave low venous pressure. Thus the ABC is rare inhese areas. However, when present, the mandibles the most commonly affected (mandible-maxillaatio, 3:1), with a higher predilection for molar

Table 1. THREE CASES OF GIANT ABC OF MANDIBLE

Data Case 1

ge when firstreported (yr)

15 3

ender F Furation 1.5 yr 4eedle aspiration forblood

Positive P

revioushistopathologicdiagnosis

— S

revious treatment None C

linical findings Large spherical swellingmeasuring 9 � 7 � 6 cmover body–ramal regionextending to right corner ofmouth. Ballooning of cortexseen. Overlying skin appearsnormal. No pulsations felt.No associated pain (Fig 1A).

L

adiographicfindings

Multilocular radiolucency of leftangle–ramus–coronoid–condyle. Root resorption(present). Multiple septa seen(Figs 1B and 1C).

M

nterventionradiology

None N

anagement Resection plus titanium platereconstruction (Fig 1D)

R

ollow-up (yr) 3.4 (Fig 1E) 2ecurrence No N

oychoudhury et al. Aneurysmal Bone Cyst of Mandible. J Oral

reas.5,6 Bernier and Bhaskar7 described the first f

ase of ABC in the jaw. Involvement of other bonesf the face such as the zygoma and zygomatic archas also been reported.6,8,9 The mean age of in-olvement in the skull and facial region is reportedo be 14.3 years,10 with no gender predilection.5

he ABC occurs predominantly in young individu-ls aged under 30 years. In this series the molarreas of the mandible were affected, with a male-emale ratio of 1:2 and a mean age of 20 years.

Over the years, the pathogenesis of ABC has beenuestioned. Jaffe and Liechtenstein1 advanced theheory that ABC often arises from a pre-existing lesionf bone, most of which has been destroyed by hem-rrhage. On the other hand, Jaffe and Liechtensteinnd other authors9 proposed vascular disturbance inhe etiopathogenesis of ABC, which may be in the

Case 2 Case 3

14

M10 moPositive

riant ABC Reparative giant cellgranuloma (?)

ge Intralesional steroids(triamcinolonehydrochloride, 40 mg, 8doses)

void swelling withations measuring 6 �

cm over left anglen extending to area

corner of mouth withying skin appearingy. Lingual expansionPain on palpation

ent) (Fig 2A).

Large single ovoid nontenderswelling measuring 10 �6 � 4 cm over right angleregion of mandibleextending to area belowcorner of mouth. Significantexpansion of lower borderwith lingual extension seen(ballooning). Overlying skinappears healthy (Fig 3A).

ular radiolucency of leftangle–ramus. Root

ption (present). Fluidwith internal septa

on CT scan (Figs 2B and

Multilocular radiolucency ofright angle–body–symphysis.Root resorption (present).Fluid levels with internalsepta seen on CT scan (Figs3B and 3C).

Digital subtractionangiography with absorbablegelatin sponge embolizationof feeder artery

on plus titanium platestruction (Fig 2D)

Resection plus titanium platereconstruction (Fig 3E)

2E) 2 (Fig 3F)No

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1998 ANEURYSMAL BONE CYST OF MANDIBLE

FIGURE 1. A, Clinical photograph showing rounded, balloon-type swelling over leftmandible. B, Panoramic radiograph showing multilocular radiolucent lesion involvingleft ramus, condyle, and coronoid process of mandible. C, CT scan showing expansilelytic lesion involving left ramus–condyle region with internal septa and fluid levels. D,Postoperative panoramic radiograph showing reconstruction plate in situ. E, Postoper-ative clinical photograph of patient.

Roychoudhury et al. Aneurysmal Bone Cyst of Mandible. J Oral Maxillofac Surg 2009.

ROYCHOUDHURY ET AL 1999

FIGURE 2. A, Clinical photograph of patient showing well-defined swelling over left mandible. B, Panoramic radiographshowing expansile multilocular radiolucent lesion of left man-dible. C, CT scan showing multilocular lytic lesion over leftside of mandible with presence of fluid level. D, Postoperativeradiograph showing well-contoured reconstruction plate inposition. E, Postoperative clinical photograph of patient show-ing optimal contour and esthetics.

Roychoudhury et al. Aneurysmal Bone Cyst of Mandible.J Oral Maxillofac Surg 2009.

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2000 ANEURYSMAL BONE CYST OF MANDIBLE

ysm, the increased pressure resulting in a dilated,ngorged intra-bony venous bed. They believed thathe presence of giant cells in a fibrous matrix merelyepresents a physiologic process of resorption and

IGURE 3. A, Preoperative photograph showing rounded, balloon-tyultilocular lesion of right mandible. C, CT scan showing expansile, multiple fluid levels within cystic spaces at right side of mandible. D, Ranoramic radiograph showing well-contoured reconstruction plate.

oychoudhury et al. Aneurysmal Bone Cyst of Mandible. J Oral Ma

eplacement rather than a primary lesion. f

In 1978 Hillerup and Hjorting-Hansen11 proposed theheory that ABC, central giant cell granuloma (CGCG),nd simple bone cyst arise from some vascular defectnd that a trauma or some form of aneurysm leads to

lling over right side of mandible. B, Panoramic radiograph showinglar osteolytic lesion with multiple occurrences of internal septation andph of specimen showing multiple radiolucent areas. E, Postoperativeperative clinical photograph of patient.

Surg 2009.

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ematoma is in direct contact with a large vessel (suchs the inferior alveolar bundle), ballooning via bloodressure occurs and an ABC results.In our series all lesions were showing ballooningith a rapid course. This finding corroborates theypothesis of Struthers and Shear,9 who in 1984howed that the microcysts were the initiating lesionf the vascular spaces. The enlargement of these mi-rocysts via stromal collapse and the osmotic forcesause rupture of thin-walled vessels with hemorrhagento the lumen. As the expansion continues and only

thin shell remains, “blowout” of the lesion mayccur, lifting the periosteum, as proposed by Jaffe andiechtenstein.1 The erosion of cortices and blowoutneurysm may be responsible for such a rapid course.

Radiographic findings of ABC vary from unicystic ra-iolucencies or moth-eaten radiolucencies to extensiveultilocular lesions causing expansion and destruction

IGURE 4. A, B, Histopathologic slides showing sinusoidal spaces fil, Multinucleated osteoclast type of giant cells in fibrous tissue (heacrophages and multinucleated giant cells in areas surrounding sin

oychoudhury et al. Aneurysmal Bone Cyst of Mandible. J Oral Ma

f both cortices.12 A characteristic radiographic feature t

as been described as a “ballooning” distention of peri-steum with a thin outline of reactive, subperiostealone.13 All our cases presented as a multilocular radi-lucency with well-defined margins. The ballooningomponent was present in all our cases, which waselpful in diagnosis. Computed tomography (CT) scan isuperior to plain radiology in defining the extent andoft tissue extension of an ABC, particularly in the skull.ultiple small fluid levels are important characteristicsf ABC on CT scan, which represent sedimentation ofed blood cells within blood-filled cavities. The ABCshat do not show fluid levels on CT scan are oftenonhomogeneous and resemble some giant cell tu-ors.14 Multiple fluid-filled cavities were also present on

he CT scan in the cases in this series.Biesecker et al,15 in their review of 66 cases of ABC (2

n the mandible), found an association with ossifyingbroma, chondroblastoma, giant cell tumor, osteoblas-

red blood cells (hematoxylin-eosin stain, original magnification �4).lin-eosin stain, original magnification �20). D, Hemosiderin-ladenspaces (hematoxylin-eosin stain, original magnification �40).

Surg 2009.

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2002 ANEURYSMAL BONE CYST OF MANDIBLE

ameral bone cyst. They concluded that these associ-ted lesions are appreciable in only 1 or 2 sections, thusequiring close observation by the pathologist. The mis-iagnosis of the primary lesion would cause recurrence.GCG and ABC can coexist, or ABC and CGCG canccur concurrently, with transition of CGCG into ABC atome stage.9,11

Vergel De Dios et al10 reported that ABC can beadiographically distinguished from CGCG and low-rade osteosarcomas because CGCG has its epicenterocated at the end of bone whereas ABC involves the

etaphysis. With regard to the jaws, the ABC is anccentric lytic lesion, expansile in nature but with anntact outer rim of bone. The presence of internal septand fluid levels on a CT or magnetic resonance imagingcan further supports the diagnosis.1,10,14

Histologically, the giant cells are a regular feature ofoth lesions and seldom prove fruitful in differentiatinghe two. Vergel De Dios et al10 observed that the stromas more fibrogenic in ABC than in a giant cell lesion. Inddition, the presence of large vascular spaces oftenined with microcysts points toward the diagnosis of theormer.9 In addition, they observed that the entity “solidBC” shows marked histologic resemblance to “repara-

ive giant cell granuloma” and osteosarcoma. Althougholid ABC and reparative giant cell granuloma may actu-lly represent the same histologic entity, their differen-iation from osteosarcoma is extremely important and issually established with the presence of anaplasia intromal cells. Hirst et al16 in 1970 reported a case of aalignant variant of ABC of the long bones with meta-

tatic deposits to the lungs. A similar report was pub-ished by Levy et al17 in 1975, where 12 cases of ABC

ere coexistent with osteosarcoma and 14 were coex-stent with osteoclastoma.

According to the World Health Organization, ABC isn “expanding osteolytic lesion consisting of blood-filledpaces of variable size separated by connective tissueepta containing trabeculae of osteoid tissue and oste-clast giant cells.”18 The gross features of ABC usuallyonsists of a multicystic, sponge-like soft tissue masslled with considerable dark, venous blood. The micro-copic features correlate with the gross features. Numer-us cavernous, sinusoidal spaces filled with blood areurrounded by loose, fibrous connective tissue. Theonnective tissue septa contain small capillaries, mul-inucleated giant cells, inflammatory cells, extravasatedrythrocytes, and hemosiderin. The multinucleated, os-eoclast-like giant cells often aggregate adjacent to theinusoidal spaces. Trabeculae of reactive, woven bonere often evident within the connective tissue.19 Ouristologic finding in all the cases was consistent with thereviously mentioned features.Traditional management of ABCs has been conser-

ative therapy with enucleation and curettage, with

eliable results. Simple curettage has been associated r

ith high recurrence rates varying from 21% to0%.15,20,21 The aim of therapy should be to eliminatehe aneurysm either by ligation of distal and proximalessels or with surgical removal. Because both areechnically difficult, higher rates of recurrence can bexpected with simple procedures such as curettage.15

Considering that ABC generally affects young pa-ients (aged �20 years in most cases),1,9,15 surgeonsay be tempted to resort to a conservative mode of

reatment, curettage, with a view to maintain esthet-cs and function. However, recurring lesions are moreistressing for the patient and the surgeon alike. Vergele Dios et al,10 in their study of 238 cases of ABC

nvolving different regions, showed that the chances ofecurrence after curettage were high (up to 90%) espe-ially when the patient was young and aged less than 20ears. The use of radiotherapy as an alternative hashown efficacy in some cases, but the risk of the devel-pment of fibrosarcoma is too great to be ignored.22,23

Koskinen et al24 in 1976 evaluated curettage and resec-ion as treatment modalities for about 20 cases of ABC.hey concluded that curettage alone accounted for a highecurrence and deemed it inadequate. In addition, devel-pment of huge cysts was noted after curettage.Aegerter and Kirkpatric25 observed that ABC was fre-

uently superimposed on osteosarcoma. They indicatedhe possibility that high proportions of simple bone cystransform into ABC and, subsequently, osteosarcoma inate phases, by either fracture or surgery apparently dueo hematoma formation. Their observations, thoughone were made in relation to the jaws, hint towardossible malignant transformation after repeated surgi-al intervention. Whether repeated intervention associ-ted with recurrent lesions actually increases thehances of malignant transformation is not clear. How-ver, such interventions do cause repeated hemorrhagend intramedullary hematoma formation.

Embolization of the feeding vessels has been showno be effective as a preoperative procedure to reducereoperative bleeding, and after surgical failure, ABC

requently lacks large feeding vessels and thereforeay require repeated embolization procedures.26

ase 3 was huge and aggressive, and thus we decidedo opt for preoperative embolization to reduce thentraoperative bleeding.

Table 2 shows the findings of several studies usingesection as the treatment modality for ABC of theaws.4,27-34 Karabouta et al33 in 1991 reported a casef extensive ABC of the right mandible that was ade-uately managed with resection and reconstructionsing autogenous ribs and coralline porous hydroxy-patite. In another report involving 3 cases of man-ibular ABC, Wiatrak et al34 in 1995 managed a giant

esion in a 10-year-old boy with extensive resectionnd microvascular reconstruction of the defect. No

ecurrence was reported in either case.

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In our series cases 1 and 3 were so extensive thatost-curettage bone would have been paper thin,

eading to fracture/defect. Case 2 was a recurrentase. The previously mentioned considerations led tour decision to perform resection and reconstruction.Three cases of extensive ABCs of the mandible are

eported. Although traditional management includesurettage, these cases were treated with resectionnd reconstruction because, in extensive lesions, theallooning thins the available bone and renders itnusable for any load bearing. Curettage in extensive

esions causes severe blood loss intraoperatively. In aealth care setup where the per-capita income ofatients does not allow extensive follow-up, recur-ences will not be caught. A more predictable andermanent cure is the requirement, especially forxtensive and recurrent lesions.

eferences1. Jaffe HL, Liechtenstein L: Solitary unicameral bone cyst with

Table 2. RESECTION AS TREATMENT MODALITY FOR AB

Author Age (yr) Gender Location

otts (1940)27 6 M Angle of mandiblebling andWagner28

(1964)

19 F Left body of mandible

arington etal29 (1964)

48 F Right maxillary sinus

oticha30

(1965)20 F Mandibular ramus

oppe31

(1968)12 F Left body of mandible

ruskin andDahlin32

(1968)

20 M Left maxilla

oyd4 (1979) 23 M Left body of mandible

arabouta etal33 (1991)

22 F Right body–ramus ofmandible

iatrak et al(1995)34

10 M Left body of mandible

resent series 15 F Left angle–ramus–coronoid–condyle ofmandible

30 F Left body–ramus ofmandible

14 M Right ramus–symphysisof mandible

Abbreviations: NM, not mentioned; NR, no recurrence.

oychoudhury et al. Aneurysmal Bone Cyst of Mandible. J Oral

emphasis on the roentgen picture, the pathologic appearanceand pathogenesis. Arch Surg 44:1004, 1942

2. Eldeeb M, Sedano HO, Waite DE: Aneurysmal bone cyst of thejaws. Report of a case associated with fibrous dysplasia andreview of the literature. Int J Oral Surg 9:301, 1980

3. Rattan V, Goyal S: Aneurysmal bone cyst of the coronoid processof the mandible. J Indian Soc Pedod Prev Dent 24:155, 2006

4. Boyd RC: Aneurysmal bone cysts of the jaws. Br J Oral Surg16:248, 1979

5. Motamedi MH, Yazdi E: Aneurysmal bone cyst of the jaws:Analysis of 11 cases. Oral Maxillofac Surg 52:471, 1994

6. Struthers P, Shear M: Aneurysmal bone cyst of the jaws. (II).Pathogenesis. Int J Oral Surg 13:92, 1984

7. Bernier JL, Bhaskar SN: Aneurysmal bone cysts of the mandible.Oral Surg Oral Med Oral Pathol 11:1018, 1958

8. Eveson JW, Moos KF, MacDonald: Aneurysmal bone cyst of thezygomatic arch. Br J Oral Surg 15:259, 1978

9. Struthers P, Shear M: Aneurysmal bone cysts of the jaws. (I).Clinicopathologic features. Int J Oral Surg 13:85, 1984

0. Vergel De Dios AM, Bond JR, Shives TC, et al: Aneurysmal bonecyst. A clinicopathologic study of 238 cases. Cancer 69:2921,1992

1. Hillerup S, Hjorting-Hansen E: Aneurysmal bone cyst—Simplebone cyst, two aspects of the same pathologic entity? Int J OralSurg 7:16, 1978

2. Kalantar Motamedi MH: Aneurysmal bone cysts of the jaws: Clin-icopathological features. J Craniomaxillofac Surg 26:56, 1998

3. Dorfman HD, Czerniak B: Cystic lesions, in Bone Tumors. StLouis, MO, Mosby, 1998, p 857

4. Hudson TM: Fluid levels in aneurysmal bone cyst: A CT feature.

JAWS

Treatment Reconstruction Follow-up

ction NM NR, 2.5 yrpartialndibulectomy

NM NM

t partialxillectomy

NM NM

al mandibularection

NM NM

partialndibulectomy

NM NM

partialxillectomy

NM NR, 26 yr

partialndibulectomy

NM NR, 34 yr

t segmentalection

Autogenous ribs andcoralline poroushydroxyapatite

NR, 2 yr

mimandibulectomyMicrovascular

reconstruction via leftscapular myo-osseouscutaneous flap

NM

segmentalection witharticulation

Titanium reconstructionplate

NR, 3.4 yr

segmentalection

Titanium reconstructionplate

NR, 2.8 yr

tmimandibulectomythoutarticulation

Titanium reconstructionplate

NR, 2 yr

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2004 CROUZON SYNDROME AND OSAS

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Oral Maxillofac Surg7:2004-2009, 2009

Combined Internal and ExternalDistraction of the Midface for the

Treatment of Crouzon Syndrome andCritical Obstructive Sleep Apnea:

A Case Report

Hans-Christian Jacobsen, MD, DMD,* Peter Sieg, MD, DMD, PhD,†

MD, DMD, PhD‡

istraction osteogenesis has become more and moremportant within the past few years for surgical cor-ection of the hypoplastic midface.1 It is especially

eceived from the Department of Maxillofacial Surgery, University

ospital Schleswig-Holstein, Lübeck, Germany.

*Vice Director, Senior Maxillofacial Surgeon.

†Professor and Director.

‡Assistant Professor, Senior Maxillofacial Surgeon.

Address correspondence and reprint requests to Dr Jacobsen: De-

artment of Maxillofacial Surgery, University Hospital, Schleswig-

olstein, Lübeck Ratzeburger Allee 160, 23538 Lübeck, Germany;

-mail: [email protected]

2009 American Association of Oral and Maxillofacial Surgeons

278-2391/09/6709-0030$36.00/0

seful in severe cases and cases with difficult soft tissueonditions; it has meanwhile become the method of firsthoice to move the upper jaw or the midface forwardfter osteotomy. The advancement can be achievedither by pushing using internal distractors2,3 or byulling using external distraction devices.4-6 In the

iterature the advantages and disadvantages of bothethods are controversial and are still being dis-

ussed.1,7 External distraction has the advantage of aontrolled and correctable vector of long-distance dis-raction. However, patients have to wear the uncom-ortable distraction device for several weeks. Afteremoval of this device, the advancement reached haso be secured by osteosynthesis or additional treat-ent with a Delaire mask (eg, Ortho-Dent-SpecialsmbH, Kisdorf, Germany).7 Subcutaneous systems

ave the advantage that they are neither annoying nor