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Aneurysmal bone cyst of the maxilla: case report and review of the literature
Journal of Pediatric Otorhinolaryngology, 25 (1993) 211-226 0 1993 Elsevier Scientific Publishers Ireland Ltd. All rights reserved 0165-5876/93/$06.00
International
211
PEDOT 00858
Aneurysmal bone cyst of the maxilla: case report and review of the literature Bruce H. Matt Department
of Otolaryngology
-Head
& Neck Indianapolis,
Surgery, Indiana IN (USA)
University
School
of Medicine,
(Received 19 February 1992) (Revised version received 28 May 1992) (Accepted 30 May 1992)
Key words:
Aneurysmal bone cyst; Head and neck tumor; Jaw tumor; Tumor of the maxilla; Bone tumor, diagnosis and treatment
Abstract Aneurysmal bone cyst (ABC) is a rare benign lesion of the head and neck. A case of an ABC in a 12-year-old female is presented, the twentieth reported such case in the maxilla. Literature review shows ABC are common in the long bones; only 2% are in the head and neck. Of the 77 lesions in the head and neck found in the literature, 31 (40%) are in the mandible, 20 (26%) are in the maxilla (including this case> and the remainder mainly in the skull. Current recommended treatment is curettage with enucleation first if technically possible.
Case report A 12-year-old white female presented in 1990 with a 2 month history of sinusitis and had received antibiotics twice. She denied prior facial trauma. Purulent tears developed and a golf ball sized mass developed in the right midface over a 2 week period. (Fig. 1) The palate was displaced inferiorly, including the anterior right maxillary teeth. This area was tender but had no associated erythema, diplopia or epistaxis. Family history was significant for a history of a ‘calcium tumor’ in a maternal aunt, and the mother and maternal aunts had histories of hypoglycemia. Physical examination revealed a 6 cm mass bulging into the gum, nasal airway, and Correspondence to: Bruce H. Matt, M.D., Department of Otolaryngology-Head & Neck Surgery, J.W. Riley Hospital for Children, Suite 0860, 702 Bamhill Drive, Indianapolis, IN 46202-5230, USA.
218
Fig. 1. Hard swelling of right midface.
anterior cheek. The area was firm to palpation. Right epiphora was present. The right nasal vault was obstructed by a medial bulging of the lateral nasal wall. Cranial nerves were intact. CT showed a large destructive soft-tissue mass in the right maxilla displacing and thinning bone at the lateral nasal wall, floor and anterior wall of the maxillary sinus. (Fig. 2) A soft-tissue density rounded mass consistent with a mucocele was seen in the contralateral maxillary sinus. EKG, serum calcium, urinalysis and chest X-ray were normal. In the operating room via a Caldwell-Luc approach, the right maxilla was exposed. The bone was spherically enlarged with the apparent nidus at the junction of the lateral nasal wall and maxillary sinus floor just posterior to the pyriform aperture (anterior face of maxilla). Breaking through the thinned bone a dark burgundy tumor which bleed freely without arterial ‘pumpers’ was found. This was firm and perhaps slightly gritty. The tumor was curetted away so no obvious gross mass was left. The tumor had not violated the mucoperiosteum posteriorly into the maxillary sinus proper. Packing was placed, the wound closed.
219
Fig. 2. CT scan demonstrating lesion in right midface with deformation of palate and maxilla.
The tumor had blood-filled cyst-like spaces with walls of varying thickness and prominent cellularity. The walls had exuberantly proliferating fibroblasts accompanied by varying concentrations of osteoclast-like giant cells. Mitoses were rare and without atypia. Hemosiderin pigment was present and lacy chondroid was prominent. Osteoid, with some osteoblasts was distributed through out (Fig. 3). The patient did well postoperatively and is without recurrence at 1 year.
Aneurysmal bone cyst (ABC) is a subset of unicameral cysts. This entity appears to first have been described as an ossifying hematoma by Van Arsdale in 1893 [14]. In 1910, Bloodgood reported a lesion which he called a periosteal hematoma [14]. In 1922, Barrie presented the term hemorrhagic osteomyelitis. Other authors have noted the similarity to giant cell tumors and have used terms such as giant cell tumor of bone, atypical giant cell tumor, and subperiosteal giant-cell tumor. Other
Fig. 3. (A) Histologic section with prominent cellularity and multinucleated tumor.
giant cells. (B) Osteoid in
221 TABLE
I
Date
Author
Age, race sex
Jaw
Side
No.
1940 1957 1958 1958 1959 1959 1959 1959 1960 1962 1964 1964 1964 1965 1968 1968 1968 1968 1968 1968 1968 1968 1969 1970 1970 1970 1971 1971 1972 1972 1972 1972 1973 1973 1975 1975 1976 1977 1978 1978 1978 1978 1978 1979 1979 1980 1981 1983 1983
Potts [341 Sherman [42] Bemier [2] Bemier [2] Bhaskar [4] Bhaskar [4] Bhaskar [4] Bhaskar [4] Wang [47] Vianna [46] Ebling [ 131 Yarington [48] Lucas [29] Koticha [25] Gruskin [ 181 Gruskin [ 181
6m 9 11 wf 59bf 9wf llwf 7bm 22wf 8wf 18 m 19wm 48 f 29 m 20 f 8f 20 m 12wf 8jm
mandible mandible mandible mandible mandible mandible mandible maxilla maxilla maxilla mandible maxilla maxilla mandible mandible maxilla mandible maxilla maxilla mandible mandible mandible maxilla mandible mandible maxilla mandible mandible maxilla maxilla mandible maxilla mandible mandible maxilla maxilla mandible maxilla zygoma maxilla maxilla mandible mandible maxilla mandible mandible mandible mandible mandible
right
1 1 1 1 1 1 1 1 1 1 1 1 1
Hoppe[231 Nosaka [31] Tillman [45] Tillman [45] Nobler [30] Ostrowski [33]
Byrd PI Costas [lo] Biesecker [5] LeJeune [26] Daugherty [ll] Buraczewski [7] Ellis [ 151 Romaniuk [38] Romaniuk [38] Komorn [24] Oliver [32] Beny [31 Powell [35] Roa [37] Gaillard [ 171 Ruiter [39] Evson [16] Cornyn [9] Reyneke [36] Hillerup [22] Steidler [43] f-kwd [61 Bwd t61 El Deeb [14] Saltzman [40] Shear [41] Harris [21]
13 m 25 w f 17bf 22 f 17 f 17 w m 26 f 17bm 10 m 14 f 26 f 20wf 18 f 16wf 12wm 9f 17wf 10 m 18 b m 10f 21wf 27 m 23 m 19wm 22 f 15 b m
right right right left right midline left right left left left left left right right left left right left right right left left left right right left left left left right right right left left left left left right
1
1 1 1 1 1 1 1 1 1 1 2 1 1 1 1 1
1 1 1 1 1 1 1 1 1 1 1 1 1 1 1 1 1 1 1
222 TABLE
I (continued)
Date
Author
Age, race sex
Jaw
Side
No.
1989 1991
Stimson [44] Matt
18 m 12wf
mandible maxilla
midline right
1 1
N.B. Blanks in the table indicate information was not obtainable from the reference. Key: Age is expressed in years; b = black; w = white; j = japanese; m = male; f = female.
names are osteitis fibrosa cystica, expansile hemangioma [14] and aneurysmal giant cell tumor [8]. Definition
ABCs are benign lesions of bone with an internal structure composed of multinucleated giant cells and osteoid. Cavernous spaces of varying size are filled with blood. Generally the ABC occurs in the realm of the orthopedic surgeon, with lesions in the long bones or vertebrae [1,14]. Epidemiology
Only 2% of ABCs are in the head and neck. Seventy-seven were reported in the head and neck region over 51 years with 51 in the jaws (66%). Including this case, of 52 cases in the facial skeleton 31 (60%) were in the mandible, 20 (38%) were in the maxilla and one was in the zygoma (2%) (Table I>. Outside the facial bones, other lesions in the head and neck were mainly in skull. Levine has reported one in the cricoid [27]. 0.1% of jaw cysts are ABC [41]. Several series have noted a slight predominance in females (60: 40). [5] The mean age of presentation is 18 years 1411. The age range reported is 6 to 63 [5,11]. In the facial skeleton, when side was mentioned (43 of 52), 18 were on the right, 22 on the left and 3 in the midline. Pathogenesis
The origin of the ABC is unknown. Some have proposed development from an alteration in hemodynamics [12,281. Other have suggested ABC are reparative replacements of a hematoma in the medullary cavity. One end of the spectrum may be ABC and if no circulatory connection is maintained then a giant cell granuloma occurs [21. ABC may be a hemangioma of the bone [20]. Other lesions found with ABC have been ‘non-ossifying fibroma, chondroblastoma, giant cell reparative granuloma, fibrous dysplasia and fibromyxoma’ [14]. Biesecker and others [5,14] believe that ABC is preceded by a primary lesion of the bone. Tillman [451 has listed a number of bony lesions with areas histologic features similar to ABC: fibrous dysplasia, osteoblastoma, chondroblastoma, bony lesions hyperparathyroidism and giant cell tumors. Most authors believe trauma is not important in the genesis of the ABC [5,19,281. Tillman [451 found no increased history of trauma than in other other bone lesions.
223
In the long bones, however, giant cell tumors arises near the epiphysis but in the jaws are usually far away from the condyles, [ll] the growth center of the mandible suggesting a difference in the development of ABC in the jaws versus the long bones. Symptoms
Wang reviewed ABC in all sites and found 71% of patients gave a history of prior trauma. [44,47] The two most frequent symptoms were nondescript pain in 55% and swelling in 24%. [4] Less common were throbbing pain in 16%, 9% with tenderness, and 8% with decreased range of motion. One lesion in the tibia was physically hot and 2 cysts had pulsations. [5] Radiography
The radiographic appearance is suggestive but not diagnostic. Usually an expansile mass of soft-tissue density is seen. The cyst is often unilocular and well circumscribed, however multilocular lesions have been reported. With increasing size of the lesion, the surrounding cortical bone is pushed out with a ‘ballooning or soap bubble appearance’. [231 Normal structures can be displaced and resorption of a tooth root can occur [14]. Differential radiographic diagnosis includes ameloblastoma, apical periodontitis, odontogenic keratocyst, odontogenic myxoma, central giant cell granuloma, malignant metastasis and central neurogenic tumor (neurofibroma, neurolemmoma) [14,441. Differential
diagnosis
Differential diagnosis includes giant cell reparative granuloma both peripheral and central, traumatic bone cyst, brown (osteoclastoma) tumor of hyperparathyroidism, myxoma, fibrous dysplasia, desmoplastic fibroma, fibrous histiocytoma, hemangioma, osteogenic sarcoma, globulomaxillary cysts, hemangioendothelioma and hemangiopericytoma. [8] Diagnosis
Aspiration of the mass may yield viscous, dark red or brown liquid. The tissue is soft, brown red, likened to a blood filled sponge. [44] Definitive diagnosis is histologic. Histologic examination
The lesion resembles a blood-filled sponge with pools of venous blood communicating in the honeycombed stroma. [l] Lacy chondroid foci are essentially pathognomic for aneurysmal bone cysts. Fibrous septa contain cartilage, fibroblasts, dilated capillaries, osteoid, extravasated blood cells, hemosiderin, and infrequently, multinucleated giant cells. The cavities are not lined by endothelium and are therefore not true cysts. The stroma is indistinguishable from that of giant cell granuloma and solid and vascular portions can be inhomogeneously distributed in the specimen [1,8,14,32]. Blood chemistries are normal (alkaline phosphatase, phosphorus, calcium etc) [44].
224
Treatment The ABC does not infiltrate,
so usually a thin shell of cortical bone is present.
P31 Resection, cryosurgery, and irradiation have been used, but current practice is curettage with enucleation first if technically possible [1,46,47]. El Deeb [14] proposes enucleation, curettage with bone chips placed in the cavity. They recommend cryosurgery if some residual ABC is left. Besides not being effective [14], radiation poses the risk of later malignancy (e.g., sarcoma> and is therefore contraindicated [11,14,45]. In the long bones, curettage with bone grafting is commonly used [141. Technically, good surgical exposure, control of feeding vessels close to the lesion and preservation of vital structures facilitate excision and minimize blood loss [14]. Prognosis Prognosis is excellent for aneurysmal bone cysts. El Deeb found a recurrence rate of 26% with curettage and enucleation for jaw lesions [14]. Most recurrences were within one year of treatment. Biesecker found a 59% recurrence with curettage, but only 2 of their 66 cases were in the jaws [5]. The highest recurrence rate has been for those under age 15 years with lesions less than 5 cm [141.
Acknowledgements I wish to thank the following for translations: Michael Agostino, M.D. (Spanish), Stan Garus (Polish), Markus Gapany, M.D. (Russian), and Reiko Yonogi, Ph.D. (Japanese).
References 1 Bat&is, J.G. (1979) Tumors of the Head and Neck: Clinical and Pathological Considerations, 2nd Edn., Williams &Wilkins, Baltimore, 393-4. 2 Bemier, J.L. and Bhaskar, S.N. (1958) Aneurysmal bone cyst of the mandible, Oral Surg. Oral Med. Oral Pathol. 11, 1018-1028. 3 Berry, M., Krishan, A. and Bhargava, S. (1973) An aneurysmal bone cyst of the mandible. Aust. Radiol. 17, 196-198. 4 Bhaskar, S.N., Bemier, J.L. and Godby, F. (1959) Aneurysmal bone cyst and other giant cell lesions of the jaws: report of 104 cases. J. Oral Surg. Anesthesia Hosp. Dent. Service 17, 30-41. 5 Biesecker, J.L., Marcove, R.C., Huvos, A.G. and Mike, V. (1970) Aneurysmal bone cysts: a clinicopathological study of 66 cases. Cancer, 26, 61.5-625. 6 Boyd, R.C. (1979) Aneurysmal bone cysts of the jaws. Br. J. Oral Surg. 16, 548. 7 Buraczewski, J. and Dabska, M. (1971) Pathogenesis of aneurysmal bone cyst: relationship between the aneurysmal bone cyst and fibrous dysplasia of bone. Cancer 28, 597-604. 8 Byrd, D.L., Allen, J.W., Kindrick, R.D. et al. (1969) Aneurysmal bone cyst of the maxilla. J. Oral Surg. 27,296-300. 9 Cornyn, J. and Morris, D.R. (1978) Different and unusual perspectives in diagnosis, Fifth annual continuing education program.‘Am. Acad. Dent. Radio]. (case No. 8).
225 10 Costas, J.B. and Peitropinto, J. (1970) Quiste Oseo-aneurismal del maxilar inferior. Ann. Esp. Odontstomat. 29, 462-467. 11 Daugherty, J.W. and Eversole, L.R. (1971) Aneurysmal bone cyst of the mandible. J. Oral Surg. 29, 737-741. 12 Donaldson, W.F. (1962) Aneurysmal bone cyst, J. Bone Joint Surg. 44A, 25-29. 13 Ebling, H. and Wagner, J.E. (1964) Aneurysmal bone cysts of the mandible: report of a case. Oral Surg. 18, 646-652. 14 ElDeeb, M., Sedano, H.O. and Waite, D.E. (1980) Aneurysmal bone cyst of the jaws. Int. J. Oral Surg. 9, 301-311. 15 Ellis, D.L. and Walters, P.J. (1978) Aneurysmal bone cysts of the maxilla. Oral Surg. 34, 26-32. 16 Evson, J.W., Moos, K.F. and Macdonald, D.G. (1978) Aneurysmal bone cysts of the zygomatic arch. Br. J. Oral Surg. 15, 259-264. 17 Gaillard, A., Nicouleau, P. and Courtay, D. (1976) Kyste an&uymsal de las branche motante de las mandibule chez l’enfant : apropos d’un cas. Rev. Stomat. Chirg. Maxillofac. 76, 26-28. 18 Gruskin, SE. and Dahlin, D.C. (1968) Aneurysmal bone cysts of the jaws. J. Oral Surg. 26,.523-528. 19 Jaffe, J.L. (1950) Aneurysmal bone cyst. Bull. Hosp. Joint Dis. 11, 3-13. 20 Hadders, H.N. and Osterdoom, H.J. (1956) The identification of the aneurysmal bone cyst with hemangioma of the skeleton. J. Pathol. Bact. 71, 193-200. 21 Harris, V.J., Lawrence, F.R. and Rodrigeuz, J. (1983) Lesions of the jaw bone: radiographic features. Warren H. Green, Inc, St. Louis, MO, pp. 42-51. 22 Hillerup, S. and Hjorting-Hansen, E. (1978) Aneurysmal bone cyst, simple bone cyst, two aspects of the same pathologic entity? Int. J. Oral Surg. 7, 16-22. 23 Hoppe, W. (1968) An aneurysmal bone cyst of the mandible: report of a case. Oral Surg. 25, l-5. 24 Komorn, R.M. (1972) Management of vascular tumors of the head and neck. South. Med. J. 65, 1106-1112. 25 Koticha, H.K.J. (1965) Aneurysmal bone cyst. Indian J. Med. Sci. 19, 315-319. 26 LeJeune, F.E. and Bordelon, J.P. (1970) Aneurysmal bone cyst of the maxillary antrum. Eye Ear Nose Throat 49, 216-217. 27 Levine, H.L. and Tubbs, R. (1986) Nonsquamous neoplasms of the larynx. Otolatyngol. Clinic. North Am. 19, 475-488. 28 Lichtenstein, L. (1950) Aneurysmal bone cyst: a pathological entity commonly mistaken for giant-cell tumor and occasionally for hemangioma and osteogenic sarcoma. Cancer 3, 279-289. 29 Lucas, R.B. (1964) Pathology of the Tumors of the Oral Tissue, J&A Churchill, Ltd., London, pp. 338-340. 30 Nobler, M.P., Higinbotham, N.L. and Phillips, R.F. (1968) The cure of aneurysmal bone cyst. Radiology 90, 1185-1192. 31 Nosaka, Y. and Nakazawa, A. (1968) Aneurysmal bone cyst of the maxillary bone. Otolaryngol. (Japan) 40,389-394. 32 Oliver, L.P. (1973) Aneurysmal bone cyst: report of a case. Oral Surg. 35, 67-76. 33 Ostrowski, L. et al. (1968) Aneurysmatic mandibular cyst, Czds. Stomat. (Poland) 21, 1379-1383. 34 Potts, W.J. (1940) Subperiosteal giant-cell tumor, J. Bone Joint Surg. 22, 417-420. 35 Powell, J.O. and Glaser, J.S. (1975) Aneurysmal bone cyst of the orbit. Arch. Ophthalmol. 93, 340-342. 36 Reyneke, J.P. (1978) Aneurysmal bone cyst of the maxilla. Oral Surg. 45, 441-447. 37 Roa, S.H. (1975) Aneurysmal bone cyst in the maxilla following the extraction of the left maxillary first molar. In: Archer, W.H. (Ed.). Textbook of Oral and Maxillo-Facial Surgery, Vol. 1, 5th Edn., WB Saunders Co, Philadelphia, pp. 654-655. 38 Romaniuk, K. and Becker, R. (1972) Two cases of aneurysmal bone cysts of the jaw. Int. J. Oral Surg. 1,48-52. 39 Ruiter, D.J., van Rijssel, T.G. and van der Velde, E.A. (1977) Aneurysmal bone cysts a clinicopathological study of 105 cases. Cancer 39, 2231-2239. 40 Saltzman, E.I. and Jun, M.Y. (1981) Aneurysmal bone cyst of the mandible: report of a case. J. Surg. Oncol. 17, 385-394. 41 Shear, M. (1983) Cysts of the oral regions. Wright PSG, Boston, 151-159.
226 42 Sherman, R.S. and Soong, K.Y. (1957) Aneurysmal bone cyst: its roentgen diagnosis. Radiology 68, 54-64. 43 Steidler, N.E., Cook, R.M. and Reade, P.C. (1978) Aneurysmal bone cysts of the jaws: a case report and review of the literature. Br. J. Oral Surg. 16, 254-261. 44 Stimson, P.G. and McDaniel, R.K. (1989) Traumatic bone cyst, aneurysmal bone cyst, and central giant cell granuloma-pathogenetically related lesions? J. Endodontics 15, 164-167. 45 Tillman, B.P., Dahlin, D.C., Lipscomb, P.R. and Stewart, J.R. (1968) Aneurysmal bone cyst: an analysis of 95 cases. Mayo Clinic Proc. 43, 178-495. 46 Vianna, M.R. and Horizonte, B. (1962) Aneurysmal bone cyst in maxilla: report of a case. J. Oral Surg. 20, 432-434. 47 Wang, S.Y. (1960) An aneurysmal bone cyst in the maxilla. Plast. Reconstr. Surg. 25, 62-72. 48 Yarington, C.T. Jr., Abbott, J. and Raines, D. (1964) Aneurysmal bone cyst of the maxilla: association with giant cell reparative granuloma. Arch. Otolaryngol. 80, 313-317.
International
211
PEDOT 00858
Aneurysmal bone cyst of the maxilla: case report and review of the literature Bruce H. Matt Department
of Otolaryngology
-Head
& Neck Indianapolis,
Surgery, Indiana IN (USA)
University
School
of Medicine,
(Received 19 February 1992) (Revised version received 28 May 1992) (Accepted 30 May 1992)
Key words:
Aneurysmal bone cyst; Head and neck tumor; Jaw tumor; Tumor of the maxilla; Bone tumor, diagnosis and treatment
Abstract Aneurysmal bone cyst (ABC) is a rare benign lesion of the head and neck. A case of an ABC in a 12-year-old female is presented, the twentieth reported such case in the maxilla. Literature review shows ABC are common in the long bones; only 2% are in the head and neck. Of the 77 lesions in the head and neck found in the literature, 31 (40%) are in the mandible, 20 (26%) are in the maxilla (including this case> and the remainder mainly in the skull. Current recommended treatment is curettage with enucleation first if technically possible.
Case report A 12-year-old white female presented in 1990 with a 2 month history of sinusitis and had received antibiotics twice. She denied prior facial trauma. Purulent tears developed and a golf ball sized mass developed in the right midface over a 2 week period. (Fig. 1) The palate was displaced inferiorly, including the anterior right maxillary teeth. This area was tender but had no associated erythema, diplopia or epistaxis. Family history was significant for a history of a ‘calcium tumor’ in a maternal aunt, and the mother and maternal aunts had histories of hypoglycemia. Physical examination revealed a 6 cm mass bulging into the gum, nasal airway, and Correspondence to: Bruce H. Matt, M.D., Department of Otolaryngology-Head & Neck Surgery, J.W. Riley Hospital for Children, Suite 0860, 702 Bamhill Drive, Indianapolis, IN 46202-5230, USA.
218
Fig. 1. Hard swelling of right midface.
anterior cheek. The area was firm to palpation. Right epiphora was present. The right nasal vault was obstructed by a medial bulging of the lateral nasal wall. Cranial nerves were intact. CT showed a large destructive soft-tissue mass in the right maxilla displacing and thinning bone at the lateral nasal wall, floor and anterior wall of the maxillary sinus. (Fig. 2) A soft-tissue density rounded mass consistent with a mucocele was seen in the contralateral maxillary sinus. EKG, serum calcium, urinalysis and chest X-ray were normal. In the operating room via a Caldwell-Luc approach, the right maxilla was exposed. The bone was spherically enlarged with the apparent nidus at the junction of the lateral nasal wall and maxillary sinus floor just posterior to the pyriform aperture (anterior face of maxilla). Breaking through the thinned bone a dark burgundy tumor which bleed freely without arterial ‘pumpers’ was found. This was firm and perhaps slightly gritty. The tumor was curetted away so no obvious gross mass was left. The tumor had not violated the mucoperiosteum posteriorly into the maxillary sinus proper. Packing was placed, the wound closed.
219
Fig. 2. CT scan demonstrating lesion in right midface with deformation of palate and maxilla.
The tumor had blood-filled cyst-like spaces with walls of varying thickness and prominent cellularity. The walls had exuberantly proliferating fibroblasts accompanied by varying concentrations of osteoclast-like giant cells. Mitoses were rare and without atypia. Hemosiderin pigment was present and lacy chondroid was prominent. Osteoid, with some osteoblasts was distributed through out (Fig. 3). The patient did well postoperatively and is without recurrence at 1 year.
Aneurysmal bone cyst (ABC) is a subset of unicameral cysts. This entity appears to first have been described as an ossifying hematoma by Van Arsdale in 1893 [14]. In 1910, Bloodgood reported a lesion which he called a periosteal hematoma [14]. In 1922, Barrie presented the term hemorrhagic osteomyelitis. Other authors have noted the similarity to giant cell tumors and have used terms such as giant cell tumor of bone, atypical giant cell tumor, and subperiosteal giant-cell tumor. Other
Fig. 3. (A) Histologic section with prominent cellularity and multinucleated tumor.
giant cells. (B) Osteoid in
221 TABLE
I
Date
Author
Age, race sex
Jaw
Side
No.
1940 1957 1958 1958 1959 1959 1959 1959 1960 1962 1964 1964 1964 1965 1968 1968 1968 1968 1968 1968 1968 1968 1969 1970 1970 1970 1971 1971 1972 1972 1972 1972 1973 1973 1975 1975 1976 1977 1978 1978 1978 1978 1978 1979 1979 1980 1981 1983 1983
Potts [341 Sherman [42] Bemier [2] Bemier [2] Bhaskar [4] Bhaskar [4] Bhaskar [4] Bhaskar [4] Wang [47] Vianna [46] Ebling [ 131 Yarington [48] Lucas [29] Koticha [25] Gruskin [ 181 Gruskin [ 181
6m 9 11 wf 59bf 9wf llwf 7bm 22wf 8wf 18 m 19wm 48 f 29 m 20 f 8f 20 m 12wf 8jm
mandible mandible mandible mandible mandible mandible mandible maxilla maxilla maxilla mandible maxilla maxilla mandible mandible maxilla mandible maxilla maxilla mandible mandible mandible maxilla mandible mandible maxilla mandible mandible maxilla maxilla mandible maxilla mandible mandible maxilla maxilla mandible maxilla zygoma maxilla maxilla mandible mandible maxilla mandible mandible mandible mandible mandible
right
1 1 1 1 1 1 1 1 1 1 1 1 1
Hoppe[231 Nosaka [31] Tillman [45] Tillman [45] Nobler [30] Ostrowski [33]
Byrd PI Costas [lo] Biesecker [5] LeJeune [26] Daugherty [ll] Buraczewski [7] Ellis [ 151 Romaniuk [38] Romaniuk [38] Komorn [24] Oliver [32] Beny [31 Powell [35] Roa [37] Gaillard [ 171 Ruiter [39] Evson [16] Cornyn [9] Reyneke [36] Hillerup [22] Steidler [43] f-kwd [61 Bwd t61 El Deeb [14] Saltzman [40] Shear [41] Harris [21]
13 m 25 w f 17bf 22 f 17 f 17 w m 26 f 17bm 10 m 14 f 26 f 20wf 18 f 16wf 12wm 9f 17wf 10 m 18 b m 10f 21wf 27 m 23 m 19wm 22 f 15 b m
right right right left right midline left right left left left left left right right left left right left right right left left left right right left left left left right right right left left left left left right
1
1 1 1 1 1 1 1 1 1 1 2 1 1 1 1 1
1 1 1 1 1 1 1 1 1 1 1 1 1 1 1 1 1 1 1
222 TABLE
I (continued)
Date
Author
Age, race sex
Jaw
Side
No.
1989 1991
Stimson [44] Matt
18 m 12wf
mandible maxilla
midline right
1 1
N.B. Blanks in the table indicate information was not obtainable from the reference. Key: Age is expressed in years; b = black; w = white; j = japanese; m = male; f = female.
names are osteitis fibrosa cystica, expansile hemangioma [14] and aneurysmal giant cell tumor [8]. Definition
ABCs are benign lesions of bone with an internal structure composed of multinucleated giant cells and osteoid. Cavernous spaces of varying size are filled with blood. Generally the ABC occurs in the realm of the orthopedic surgeon, with lesions in the long bones or vertebrae [1,14]. Epidemiology
Only 2% of ABCs are in the head and neck. Seventy-seven were reported in the head and neck region over 51 years with 51 in the jaws (66%). Including this case, of 52 cases in the facial skeleton 31 (60%) were in the mandible, 20 (38%) were in the maxilla and one was in the zygoma (2%) (Table I>. Outside the facial bones, other lesions in the head and neck were mainly in skull. Levine has reported one in the cricoid [27]. 0.1% of jaw cysts are ABC [41]. Several series have noted a slight predominance in females (60: 40). [5] The mean age of presentation is 18 years 1411. The age range reported is 6 to 63 [5,11]. In the facial skeleton, when side was mentioned (43 of 52), 18 were on the right, 22 on the left and 3 in the midline. Pathogenesis
The origin of the ABC is unknown. Some have proposed development from an alteration in hemodynamics [12,281. Other have suggested ABC are reparative replacements of a hematoma in the medullary cavity. One end of the spectrum may be ABC and if no circulatory connection is maintained then a giant cell granuloma occurs [21. ABC may be a hemangioma of the bone [20]. Other lesions found with ABC have been ‘non-ossifying fibroma, chondroblastoma, giant cell reparative granuloma, fibrous dysplasia and fibromyxoma’ [14]. Biesecker and others [5,14] believe that ABC is preceded by a primary lesion of the bone. Tillman [451 has listed a number of bony lesions with areas histologic features similar to ABC: fibrous dysplasia, osteoblastoma, chondroblastoma, bony lesions hyperparathyroidism and giant cell tumors. Most authors believe trauma is not important in the genesis of the ABC [5,19,281. Tillman [451 found no increased history of trauma than in other other bone lesions.
223
In the long bones, however, giant cell tumors arises near the epiphysis but in the jaws are usually far away from the condyles, [ll] the growth center of the mandible suggesting a difference in the development of ABC in the jaws versus the long bones. Symptoms
Wang reviewed ABC in all sites and found 71% of patients gave a history of prior trauma. [44,47] The two most frequent symptoms were nondescript pain in 55% and swelling in 24%. [4] Less common were throbbing pain in 16%, 9% with tenderness, and 8% with decreased range of motion. One lesion in the tibia was physically hot and 2 cysts had pulsations. [5] Radiography
The radiographic appearance is suggestive but not diagnostic. Usually an expansile mass of soft-tissue density is seen. The cyst is often unilocular and well circumscribed, however multilocular lesions have been reported. With increasing size of the lesion, the surrounding cortical bone is pushed out with a ‘ballooning or soap bubble appearance’. [231 Normal structures can be displaced and resorption of a tooth root can occur [14]. Differential radiographic diagnosis includes ameloblastoma, apical periodontitis, odontogenic keratocyst, odontogenic myxoma, central giant cell granuloma, malignant metastasis and central neurogenic tumor (neurofibroma, neurolemmoma) [14,441. Differential
diagnosis
Differential diagnosis includes giant cell reparative granuloma both peripheral and central, traumatic bone cyst, brown (osteoclastoma) tumor of hyperparathyroidism, myxoma, fibrous dysplasia, desmoplastic fibroma, fibrous histiocytoma, hemangioma, osteogenic sarcoma, globulomaxillary cysts, hemangioendothelioma and hemangiopericytoma. [8] Diagnosis
Aspiration of the mass may yield viscous, dark red or brown liquid. The tissue is soft, brown red, likened to a blood filled sponge. [44] Definitive diagnosis is histologic. Histologic examination
The lesion resembles a blood-filled sponge with pools of venous blood communicating in the honeycombed stroma. [l] Lacy chondroid foci are essentially pathognomic for aneurysmal bone cysts. Fibrous septa contain cartilage, fibroblasts, dilated capillaries, osteoid, extravasated blood cells, hemosiderin, and infrequently, multinucleated giant cells. The cavities are not lined by endothelium and are therefore not true cysts. The stroma is indistinguishable from that of giant cell granuloma and solid and vascular portions can be inhomogeneously distributed in the specimen [1,8,14,32]. Blood chemistries are normal (alkaline phosphatase, phosphorus, calcium etc) [44].
224
Treatment The ABC does not infiltrate,
so usually a thin shell of cortical bone is present.
P31 Resection, cryosurgery, and irradiation have been used, but current practice is curettage with enucleation first if technically possible [1,46,47]. El Deeb [14] proposes enucleation, curettage with bone chips placed in the cavity. They recommend cryosurgery if some residual ABC is left. Besides not being effective [14], radiation poses the risk of later malignancy (e.g., sarcoma> and is therefore contraindicated [11,14,45]. In the long bones, curettage with bone grafting is commonly used [141. Technically, good surgical exposure, control of feeding vessels close to the lesion and preservation of vital structures facilitate excision and minimize blood loss [14]. Prognosis Prognosis is excellent for aneurysmal bone cysts. El Deeb found a recurrence rate of 26% with curettage and enucleation for jaw lesions [14]. Most recurrences were within one year of treatment. Biesecker found a 59% recurrence with curettage, but only 2 of their 66 cases were in the jaws [5]. The highest recurrence rate has been for those under age 15 years with lesions less than 5 cm [141.
Acknowledgements I wish to thank the following for translations: Michael Agostino, M.D. (Spanish), Stan Garus (Polish), Markus Gapany, M.D. (Russian), and Reiko Yonogi, Ph.D. (Japanese).
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