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Idiopathic bilateral central giant cell reparative granuloma of jaws: A case report and literature review
International Journal of Pediatric Otorhinolaryngology 74 (2010) 547–552
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International Journal of Pediatric Otorhinolaryngology journal homepage: www.elsevier.com/locate/ijporl
Case report
Idiopathic bilateral central giant cell reparative granuloma of jaws: A case report and literature review ¨ zlem Ekici b, Bu¨lent Erdog˘an a Erkan Orhan a,*, Songu¨l Erol a, Orgun Deren a, Asuman Sevin a, O a b
Department of Plastic and Reconstructive Surgery, Ankara Numune Training and Research Hospital, Ankara, Turkey Department of Pathology, Ankara Numune Training and Research Hospital, Ankara, Turkey
A R T I C L E I N F O
A B S T R A C T
Article history: Received 5 September 2009 Received in revised form 7 February 2010 Accepted 9 February 2010 Available online 12 March 2010
Objectives: The central giant-cell reparative granuloma has been defined as a localized benign but sometimes aggressive osteolytic proliferation consisting of fibrous tissue with hemorrhage and hemosiderin deposits, presence of osteoclast-like giant cells and reactive bone formation. It is a benign lesion usually appears as solitary, multilocular, radiolucencies, located in the mandible and maxilla. Multiple CGCRGs of the jaw bones is very rare and, if it occurs, it is usually associated with hyperparathyroidism in majority of the cases .This report presents an unusual case of a 12-year-old girl who has idiopatic, bilateral giant cell granulomas of the angulus mandible. Methods: A 12 year-old girl was admitted to our department with complain of swelling on both right and left side of her lower jaw. There was no history of trauma, dental problem or neck infection. Blood chemistry, including calcium, alkaline phosphatase and inorganic phosphorus was normal. Patient had not family history, clinical apperiance like cherubism or noonan sydrome and systemic anomalies. MRI showed, in right ramus mandible, 37 mm 35 mm 28 mm size mass and in lenf ramus mandible, an expansile, 30 mm 38 mm 12 mm size mass. The patient underwent surgical curettage of the lesion through an intraoral approach under general anesthesia. Result: The histopathologic examination of the lesion was reported as ‘giant cell reparative granuloma. Conclusion: Our patient had multiple CGCRG in her jaw. In literature there is several reports about multiple CGCRG but unlike of that report our patient had no syndromes like Cherubism, Noonan syndrome, neurofibromatosis type-1 and systemic disease like hyperparathyroidism ,fibrous dysplasia. So we define this case as Idiopatic bilaterally central giant cell reparative granuloma of jaw. ß 2010 Elsevier Ireland Ltd. All rights reserved.
Keywords: Giant cell Granuloma Jaw bone
The central giant cell reparative granuloma (CGCRG), has been first described by Jaffe in 1953 [1]. Jaffe used the ‘central giant cell reparative granuloma’ (CGCRG) term to describe a reactive lesion to an intra-osseous hemorrhage, which was clinically different from a ‘true’ giant cell tumor [1]. The World Health Organization has defined it as a localized benign but sometimes aggressive osteolytic proliferation consisting of fibrous tissue with hemorrhage and hemosiderin deposits, presence of osteoclast-like giant cells and reactive bone formation [2]. It is an uncommon benign lesion usually located in the mandible and maxilla [3] but rare cases of CGCRG have been reported in the bones of the skull, including the orbit, paranasal sinuses, cranial vault, temporal, sphenoid, and ethmoid bones, and in the small bones of the hands and feet [4].
* Corresponding author at: 60. SK. 132/11 Emek, Ankara 06510, Turkey. Tel.: +90 312 508 45 32/312 213 83 64/532 500 41 92; fax: +90 312 427 09 06/312 68 76. E-mail address: [email protected] (E. Orhan). 0165-5876/$ – see front matter ß 2010 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijporl.2010.02.006
The true nature of this lesion is unknown, and there is controversy in the literature about whether it is a true neoplasm or a reactive response [5–7]. Chuong et al. and Ficarra et al. have suggested to separate the jaw giant cell lesions into aggressive and nonaggressive types based on clinical and radiologic consideration. Aggressive types have aggressive features such as rapid growth, root resorption, paresthesia, or pain and have very high recurrence rates [8,9]. Although there is a female predominance of both maxillary and mandibular CGCRG [6,7,10–13], in one series in which only mandibular CGCRG was included, female-to-male ratio of 1:1 was found [14]. Whitaker and Bouquot investigated the correlation between hormonal influence and female predominance, their findings suggested that factors other than ovarian hormones, estrogen and progesterone are responsible for the development of CGCRG [15]. The CGCRG may occur at any age but it is most commonly seen (between 42% and 72%) before the age of 30 [10,11,14,16] and initial presentation beyond age 50 was unusual [10].
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Previous studies have shown that mandible is the most common location for the CGCRG. It occurs at least twice as often in the mandible than in the maxilla [6,10,11]. Most lesions (75%) in the maxilla appeared in the anterior region [11,14], whereas in the mandible the lesions are more equally distributed between the anterior and posterior region [6,10,11,16]. In Eisenbud et al.’s series, 62% of the cases have been measured over 30 mm [10]. CGCRGs usually present as an expansile radiolucency (87.5%) in X-ray films, but radiologic features vary from ill-defined destructive lesions to a well-defined, multilocular appearance [17]. In the series of de Lange, a multilocular aspect was present in 15.7% of lesions and 84.3% of lesions were unilocular [16] but in series of Whitaker and Waldron 61% of lesions were multiocular and 39% of lesions were unilocular in appearance. Teeth or tooth displacement and root resorption can be seen and root resorption characterizes the potential for the lesion to be benign locally aggressive [11]. In the series of de Lange root resorption was seen in 13.5% of the lesions and displacement of teeth was seen in 18.0% of lesions [16]. In the series of Whitaker and Waldron root resorption was seen in 43% of cases [11]. The features seen on computed tomography are clearer than those seen on plain film [12]. The radiologic appearance of the lesion is not pathognomonic and may be confused with that of many other lesions of the jaws [18,19]. Although the majority of cases were asymptomatic [11,16], the most common feature was a painless smooth swelling in the face or in the oral cavity [16]. The lesion does not invade the perineural sheets so paresthesia is not usually observed in these patients [3]. The other symptoms and signs are facial asymmetry, impaired nasal breathing, loosing or displacement of teeth, and pathologic fracture [3]. Histologically, multinucleated giant cells, in a cellular vascular stroma, and often-new bone formation are demonstrated. The osteoclast-like giant cells have a patchy distribution and are usually associated with areas of hemorrhage. Ultrastructurally, the proliferating cells include spindle- shaped fibroblasts, myofibroblasts, and inflammatory mononuclear cells [20]. Kruse-Losler et al. [21] proved that the aggressive variant of CGCRG presented a high number of giant cells, an increased mitotic activity, and a high fractional surface area. However, other studies have not been able to predict the clinical course of CGCRGs from known histological or immunohistochemical features [22]. The etiopathogenesis of CGCRG is uncertain, Jaffe’s [1] originally proposed CGCRG is a local reparative process in response to a trauma-induced intra-osseous hemorrhage, However, many patients with CGCRG have no history of trauma. Infectious and developmental etiologies have been suggested for etiopathogenesis of CGCRG [10,23]. CGCRG of the jaw is usually unifocal, the occurrence of multifocal lesions in patients with CGCG is reported to be extremely rare [24,25]. The differential diagnosis of CGCRG includes other giant cell lesions, such as true giant cell tumor of bone, aneurysmal bone cyst, chondroblastoma. Because of their different biologic behaviors, making different diagnosis between CGCRG and giant cell tumor (GCT) has important for therapeutic implications. True giant cell tumor of bone usually occurs in the epiphyses of long bones and is rare in the skull but CGCRG usually occurs in mandible and maxilla. Both GCRG and GCT appear as lytic defects on radiographic examination and must be distinguished by histology [26]. Hirschl and Katz defined major histological criteria in order to distinguish between the two lesions [23].
On histologic examination, GCT has larger and more rounded giant cells with a greater number of nuclei that cells are more uniformly dispersed than in CGCRG. GCT has less foci of osteoid and new bone and has rare hemorrhage, hemosiderin deposits or fibrosis. GCT’s stroma shows composed of plump, round and oval cells together [27]. Aneurysmal bone cyst may be differential from CGCRG by its microscopic appearance that reveals a honeycomb network of thin-walled cavities filled with blood [23]. Chondroblastoma has unique histologic presentations [28]. The traditional therapy of CGCRG has been local curettage, and this has been associated with a high success rate (80%) [29]. Several surgical methods have been described for removal of more aggressive CGCRGs, such as curettage and cryosurgery [30], curettage and peripheral osteotomy, excision and then reconstruction by using an autologous iliac crest bone graft, osseo-integrated implants, and an ‘‘over denture’’ prosthesis [31]. Some authors suggested using laser or cryoprobe for the sterilization of the CGCRG margins [3]. The CGCRG is a very vascular lesion so considerable blood loss may occur during surgery and blood products should be available [10]. Surgical treatment of CGCRGs can be associated with recurrence and serious facial mutilation and loss of teeth and tooth germs. To avoid such disadvantages, a number of alternative nonsurgical therapies including interferon alpha-2a, calcitonin and intralesional corticosteroid injection have been advocated for the management of CGCRG [32–35]. Non-surgical treatment of CGCRG is probably a good treatment option for small slowly enlarging lesions. Successful treatment of painful, large, and rapidly growing lesions is more likely achieved by surgical removal. In the literature, recurrence rates vary between 11% and 35% [4,8–10,36]. Whitaker and Waldron reported a mean interval between initial treatment and treatment of a recurrence was 21 months, and stated that very few recurrences were manifested after 2 years of initial treatment [11]. There have been reports of recurrence as late as 22 years [37]. Chuong et al. and Ficarra et al. reported 72% recurrence rate in the aggressive lesions, 3% recurrence rate in the nonaggressive lesions [8,9]. Whitaker–Waldron reported 46.1% recurrence rate in aggressive lesions [11]. Age of patients at the time of initial treatment may be a factor in the frequency of recurrence [4,8–11]. In male patients, the percentage disease free survival was significantly better for older than for younger patients and no significant differences in survival rates were observed between older and younger female patients [36]. Whitaker and Waldron have a distinct impression that lesions over 3.0 cm in greatest diameter were more likely to recur than smaller lesions [11]. According to de Lange, there was no significant difference in recurrence rates between the mandible and the maxilla [36]. This report presents an unusual case of a 12-year-old girl who has idiopathic, bilateral giant cell granulomas of the angulus mandible. 1. Case A 12-year-old girl was admitted to our department with complain of swelling on both right and left side of her lower jaw (Fig. 1a–c). Her parents stated that the swelling has been developed gradually for 6 months. The painless swellings resulted in facial asymmetry. There was no history of trauma, dental problem or neck infection None of the family members had similar lesions. Routine physical examination and ear-nose-throat examination was normal except for a 4 cm 3 cm fixed painless mass in the right
E. Orhan et al. / International Journal of Pediatric Otorhinolaryngology 74 (2010) 547–552
ramus of the mandible and a 3 cm 2 cm fixed painless in the left ramus of the mandible. The patient did not have any motor or sensory deficit. There was no intraoral swelling. Blood chemistry, including calcium, alkaline phosphatase and inorganic phosphorus was normal. Panoramic radiography revealed bilateral partially corticated, radiolucent lesions on mandible. MRI showed, in right ramus mandible, an expansile, 37 mm 35 mm 28 mm size mass which made cortical irritation, spread to tooth root and surrounded by a thin bony shell and in left ramus mandible, an expansile, 30 mm 38 mm 12 mm size mass which made cortical irritation, spread to tooth root and surrounded by a thin bony shell (Fig. 2a–c). The patient underwent surgical curettage of the lesion through an intraoral approach under general anesthesia. After the thin bony shell was removed, the reddish, friable and granulomatous material was curetted and excised. Then the cavity was drilled with diamond burr until obtaining a clear surgical margin. After surgical curettage the stabilization of mandible was very well but there was wide cortical bone defect on mandible so the cavity reconstruction was done with cortical bone grafts from iliac crest
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and graft stabilization was made with biodegradable plate and screw (which was made polylactic acid/polyglycolic acid combination). Then the wound was closed with interrupted sutures. The post-operative course was uneventful, and she was discharged on post-operative day 3. The histopathologic examination revealed cellular vascular connective tissue with osteoclast-type giant cells having multiple nuclei and granulation tissue rich in mononuclear inflammatory cells (Fig. 3a and b). The lesion was reported as ‘giant cell reparative granuloma’. No evidence of clinical and radiological recurrence was observed during follow-up for 10 months and the grafts were integrated during this time. 2. Discussion Most of the multifocal lesion of CGCRGs are associated with some form of inherited syndrome or systemic disease, such as brown tumor of hyperparathyroidism [38], fibrous dysplasia, ossifying fibroma [39], paget disease, fibro-osseous lesion or odontogenic fibroma [40].
Fig. 1. (a–c) Preoperative image of patients.
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Fig. 2. (a–c) MR image of patients.
Multiple CGCRGs of the jaw bones usually associated with hyperparathyroidism in the majority of cases [17]. Brown tumors of hyperparathyroidism may occur at any age [25]. This can occur either primarily, due to hyperplasia or neoplasia of the parathyroid glands, or secondarily in association with renal osteodystrophy [41]. Hypercalcemia, hypophosphatemia, and elevated parathyroid hormone blood levels characterize hyperparathyroidism. In our patient, an endocrine workup was unremarkable, effectively excluding hyperparathyroidism. Only a few cases of multiple CGCRG of the jaws in the absence of hyperparathyroidism have been reported in the English literature [42,43]. Bloom et al. and Hjorting-Hansen and Worsoe-Petersonzhave presented cases of multiple recurrence giant cell lesions involving the jaws and long bones [44]. These cases were probably giant cell tumors or low-grade osteosarcomas and are histologically dissimilar to giant cell granuloma. Davis and Tideman reported a case of CGCRG in the mandible and the maxilla [45]. The maxillary lesion was detected 4 months after the mandibular lesion was first seen. Cassatly et al. presented case of CGCRG in the mandible [43], and Smith et al. presented a case of CGCRGs in both maxillary sinuses [42].
A number of syndromes have been reported to be associated with multiple giant cell lesions of the jaws, including cherubism, Noonan syndrome [46,47] neurofibromatosis type-1 [24,48], Ramon syndrome (gingival fibromatosis, hypertrichosis, epilepsy, mental and somatic retardation, and cherubism-like lesions) [49], Noonan-like syndrome, cherubism, and polyarticular pigmented villonodular synovitis (multiple lesions of pigmented villonodular synovitis affecting multiple bones, commonly the knees and ankles) [47]. Cherubism, an autosomal dominantly inherited condition with variable expressivity, is characterized by multiquadrant radiolucent lesions of the jaws and a progressive and clinically, symmetrical enlargement of the mandible and/or the maxilla. There is usually a family history of similarly affected family members. Regression is often seen following puberty [50]. Noonan syndrome is a relatively common congenital syndrome: cardiac anomalies, short stature and atypical face like a broad or webbed neck, low set and posteriorly angulated ears, ptosis, hypertelorism, and downward-slanting eyes [51]. Similar to cherubism, Noonan syndrome is an autosomal dominantly inherited syndrome with variable expressivity. Our case has no family history, no clinical appearance like cherubism or Noonan syndrome (Fig. 1a–c), no
E. Orhan et al. / International Journal of Pediatric Otorhinolaryngology 74 (2010) 547–552
Fig. 3. (a and b) Histological appearances of lesions; nest of osteoclast-type giant cell alternate with fibrovascular stroma. The lamellar bone on the left side is seen.
systemic anomalies and swelling has been developed gradually for the last 6 months. 3. Conclusion Our patient had bilateral CGCRG in her jaw. In literature there are several reports about multiple CGCRG but unlike of that report our patient had no syndromes like cherubism, Noonan syndrome, neurofibromatosis type-1 and systemic disease like hyperparathyroidism, fibrous dysplasia. So we define this case as idiopathic bilaterally central giant cell reparative granuloma of jaw. References [1] H.L. Jaffe, Giant-cell reparative granuloma, traumatic bone cyst, and fibrous (fibroosseous) dysplasia of the jawbones, Oral Surg. Oral Med. Oral Pathol. 6 (1953) 159–175. [2] L. Barnes, J.W. Eveson, P. Reichart, Pathology and genetics of head and neck tumour, in: P. Kleihues, L.H. Sobin (Series Eds.), World Health Organization Classification of Tumours, IARC Press, Lyon, France, 2005, p. 324. ¨ stu¨ndag˘, M. I˙s¸eri, G. Keskin, B. Mu¨ezzinog˘lu, Central giant cell granuloma, Int. [3] E. U J. Pediatr. Otorhinolaryngol. 65 (2002) 143–146. [4] P.L. Auclair, P. Cuenin, F.J. Kratochvil, L.J. Slater, G.L. Ellis, A clinical and histomorphologic comparison of the central giant cell granuloma and the giant cell tumor, Oral Surg. Oral Med. Oral. Pathol. 66 (1988) 197–208. [5] L.T. Austın Jr., D.C. Dahlın, R.Q. Royer, Giant cell reparative granuloma and related condition affecting the jawbones, Oral Surg. Oral Med. Oral Pathol. 12 (1959) 1285–1295. [6] I. Kaffe, L. Ardekian, S. Taicher, M.M. Littner, A. Buchner, Radiologic features of central giant cell granuloma of the jaws, Oral Surg. Oral Med. Oral Pathol. Oral Radiol. Endod. 81 (1996) 720–726. [7] C.A. Waldron, W.G. Shafer, The central giant cell reparative granuloma of the jaws: an analysis of 38 cases, Am. J. Clin. Pathol. 45 (1966) 437–447.
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Case report
Idiopathic bilateral central giant cell reparative granuloma of jaws: A case report and literature review ¨ zlem Ekici b, Bu¨lent Erdog˘an a Erkan Orhan a,*, Songu¨l Erol a, Orgun Deren a, Asuman Sevin a, O a b
Department of Plastic and Reconstructive Surgery, Ankara Numune Training and Research Hospital, Ankara, Turkey Department of Pathology, Ankara Numune Training and Research Hospital, Ankara, Turkey
A R T I C L E I N F O
A B S T R A C T
Article history: Received 5 September 2009 Received in revised form 7 February 2010 Accepted 9 February 2010 Available online 12 March 2010
Objectives: The central giant-cell reparative granuloma has been defined as a localized benign but sometimes aggressive osteolytic proliferation consisting of fibrous tissue with hemorrhage and hemosiderin deposits, presence of osteoclast-like giant cells and reactive bone formation. It is a benign lesion usually appears as solitary, multilocular, radiolucencies, located in the mandible and maxilla. Multiple CGCRGs of the jaw bones is very rare and, if it occurs, it is usually associated with hyperparathyroidism in majority of the cases .This report presents an unusual case of a 12-year-old girl who has idiopatic, bilateral giant cell granulomas of the angulus mandible. Methods: A 12 year-old girl was admitted to our department with complain of swelling on both right and left side of her lower jaw. There was no history of trauma, dental problem or neck infection. Blood chemistry, including calcium, alkaline phosphatase and inorganic phosphorus was normal. Patient had not family history, clinical apperiance like cherubism or noonan sydrome and systemic anomalies. MRI showed, in right ramus mandible, 37 mm 35 mm 28 mm size mass and in lenf ramus mandible, an expansile, 30 mm 38 mm 12 mm size mass. The patient underwent surgical curettage of the lesion through an intraoral approach under general anesthesia. Result: The histopathologic examination of the lesion was reported as ‘giant cell reparative granuloma. Conclusion: Our patient had multiple CGCRG in her jaw. In literature there is several reports about multiple CGCRG but unlike of that report our patient had no syndromes like Cherubism, Noonan syndrome, neurofibromatosis type-1 and systemic disease like hyperparathyroidism ,fibrous dysplasia. So we define this case as Idiopatic bilaterally central giant cell reparative granuloma of jaw. ß 2010 Elsevier Ireland Ltd. All rights reserved.
Keywords: Giant cell Granuloma Jaw bone
The central giant cell reparative granuloma (CGCRG), has been first described by Jaffe in 1953 [1]. Jaffe used the ‘central giant cell reparative granuloma’ (CGCRG) term to describe a reactive lesion to an intra-osseous hemorrhage, which was clinically different from a ‘true’ giant cell tumor [1]. The World Health Organization has defined it as a localized benign but sometimes aggressive osteolytic proliferation consisting of fibrous tissue with hemorrhage and hemosiderin deposits, presence of osteoclast-like giant cells and reactive bone formation [2]. It is an uncommon benign lesion usually located in the mandible and maxilla [3] but rare cases of CGCRG have been reported in the bones of the skull, including the orbit, paranasal sinuses, cranial vault, temporal, sphenoid, and ethmoid bones, and in the small bones of the hands and feet [4].
* Corresponding author at: 60. SK. 132/11 Emek, Ankara 06510, Turkey. Tel.: +90 312 508 45 32/312 213 83 64/532 500 41 92; fax: +90 312 427 09 06/312 68 76. E-mail address: [email protected] (E. Orhan). 0165-5876/$ – see front matter ß 2010 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijporl.2010.02.006
The true nature of this lesion is unknown, and there is controversy in the literature about whether it is a true neoplasm or a reactive response [5–7]. Chuong et al. and Ficarra et al. have suggested to separate the jaw giant cell lesions into aggressive and nonaggressive types based on clinical and radiologic consideration. Aggressive types have aggressive features such as rapid growth, root resorption, paresthesia, or pain and have very high recurrence rates [8,9]. Although there is a female predominance of both maxillary and mandibular CGCRG [6,7,10–13], in one series in which only mandibular CGCRG was included, female-to-male ratio of 1:1 was found [14]. Whitaker and Bouquot investigated the correlation between hormonal influence and female predominance, their findings suggested that factors other than ovarian hormones, estrogen and progesterone are responsible for the development of CGCRG [15]. The CGCRG may occur at any age but it is most commonly seen (between 42% and 72%) before the age of 30 [10,11,14,16] and initial presentation beyond age 50 was unusual [10].
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Previous studies have shown that mandible is the most common location for the CGCRG. It occurs at least twice as often in the mandible than in the maxilla [6,10,11]. Most lesions (75%) in the maxilla appeared in the anterior region [11,14], whereas in the mandible the lesions are more equally distributed between the anterior and posterior region [6,10,11,16]. In Eisenbud et al.’s series, 62% of the cases have been measured over 30 mm [10]. CGCRGs usually present as an expansile radiolucency (87.5%) in X-ray films, but radiologic features vary from ill-defined destructive lesions to a well-defined, multilocular appearance [17]. In the series of de Lange, a multilocular aspect was present in 15.7% of lesions and 84.3% of lesions were unilocular [16] but in series of Whitaker and Waldron 61% of lesions were multiocular and 39% of lesions were unilocular in appearance. Teeth or tooth displacement and root resorption can be seen and root resorption characterizes the potential for the lesion to be benign locally aggressive [11]. In the series of de Lange root resorption was seen in 13.5% of the lesions and displacement of teeth was seen in 18.0% of lesions [16]. In the series of Whitaker and Waldron root resorption was seen in 43% of cases [11]. The features seen on computed tomography are clearer than those seen on plain film [12]. The radiologic appearance of the lesion is not pathognomonic and may be confused with that of many other lesions of the jaws [18,19]. Although the majority of cases were asymptomatic [11,16], the most common feature was a painless smooth swelling in the face or in the oral cavity [16]. The lesion does not invade the perineural sheets so paresthesia is not usually observed in these patients [3]. The other symptoms and signs are facial asymmetry, impaired nasal breathing, loosing or displacement of teeth, and pathologic fracture [3]. Histologically, multinucleated giant cells, in a cellular vascular stroma, and often-new bone formation are demonstrated. The osteoclast-like giant cells have a patchy distribution and are usually associated with areas of hemorrhage. Ultrastructurally, the proliferating cells include spindle- shaped fibroblasts, myofibroblasts, and inflammatory mononuclear cells [20]. Kruse-Losler et al. [21] proved that the aggressive variant of CGCRG presented a high number of giant cells, an increased mitotic activity, and a high fractional surface area. However, other studies have not been able to predict the clinical course of CGCRGs from known histological or immunohistochemical features [22]. The etiopathogenesis of CGCRG is uncertain, Jaffe’s [1] originally proposed CGCRG is a local reparative process in response to a trauma-induced intra-osseous hemorrhage, However, many patients with CGCRG have no history of trauma. Infectious and developmental etiologies have been suggested for etiopathogenesis of CGCRG [10,23]. CGCRG of the jaw is usually unifocal, the occurrence of multifocal lesions in patients with CGCG is reported to be extremely rare [24,25]. The differential diagnosis of CGCRG includes other giant cell lesions, such as true giant cell tumor of bone, aneurysmal bone cyst, chondroblastoma. Because of their different biologic behaviors, making different diagnosis between CGCRG and giant cell tumor (GCT) has important for therapeutic implications. True giant cell tumor of bone usually occurs in the epiphyses of long bones and is rare in the skull but CGCRG usually occurs in mandible and maxilla. Both GCRG and GCT appear as lytic defects on radiographic examination and must be distinguished by histology [26]. Hirschl and Katz defined major histological criteria in order to distinguish between the two lesions [23].
On histologic examination, GCT has larger and more rounded giant cells with a greater number of nuclei that cells are more uniformly dispersed than in CGCRG. GCT has less foci of osteoid and new bone and has rare hemorrhage, hemosiderin deposits or fibrosis. GCT’s stroma shows composed of plump, round and oval cells together [27]. Aneurysmal bone cyst may be differential from CGCRG by its microscopic appearance that reveals a honeycomb network of thin-walled cavities filled with blood [23]. Chondroblastoma has unique histologic presentations [28]. The traditional therapy of CGCRG has been local curettage, and this has been associated with a high success rate (80%) [29]. Several surgical methods have been described for removal of more aggressive CGCRGs, such as curettage and cryosurgery [30], curettage and peripheral osteotomy, excision and then reconstruction by using an autologous iliac crest bone graft, osseo-integrated implants, and an ‘‘over denture’’ prosthesis [31]. Some authors suggested using laser or cryoprobe for the sterilization of the CGCRG margins [3]. The CGCRG is a very vascular lesion so considerable blood loss may occur during surgery and blood products should be available [10]. Surgical treatment of CGCRGs can be associated with recurrence and serious facial mutilation and loss of teeth and tooth germs. To avoid such disadvantages, a number of alternative nonsurgical therapies including interferon alpha-2a, calcitonin and intralesional corticosteroid injection have been advocated for the management of CGCRG [32–35]. Non-surgical treatment of CGCRG is probably a good treatment option for small slowly enlarging lesions. Successful treatment of painful, large, and rapidly growing lesions is more likely achieved by surgical removal. In the literature, recurrence rates vary between 11% and 35% [4,8–10,36]. Whitaker and Waldron reported a mean interval between initial treatment and treatment of a recurrence was 21 months, and stated that very few recurrences were manifested after 2 years of initial treatment [11]. There have been reports of recurrence as late as 22 years [37]. Chuong et al. and Ficarra et al. reported 72% recurrence rate in the aggressive lesions, 3% recurrence rate in the nonaggressive lesions [8,9]. Whitaker–Waldron reported 46.1% recurrence rate in aggressive lesions [11]. Age of patients at the time of initial treatment may be a factor in the frequency of recurrence [4,8–11]. In male patients, the percentage disease free survival was significantly better for older than for younger patients and no significant differences in survival rates were observed between older and younger female patients [36]. Whitaker and Waldron have a distinct impression that lesions over 3.0 cm in greatest diameter were more likely to recur than smaller lesions [11]. According to de Lange, there was no significant difference in recurrence rates between the mandible and the maxilla [36]. This report presents an unusual case of a 12-year-old girl who has idiopathic, bilateral giant cell granulomas of the angulus mandible. 1. Case A 12-year-old girl was admitted to our department with complain of swelling on both right and left side of her lower jaw (Fig. 1a–c). Her parents stated that the swelling has been developed gradually for 6 months. The painless swellings resulted in facial asymmetry. There was no history of trauma, dental problem or neck infection None of the family members had similar lesions. Routine physical examination and ear-nose-throat examination was normal except for a 4 cm 3 cm fixed painless mass in the right
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ramus of the mandible and a 3 cm 2 cm fixed painless in the left ramus of the mandible. The patient did not have any motor or sensory deficit. There was no intraoral swelling. Blood chemistry, including calcium, alkaline phosphatase and inorganic phosphorus was normal. Panoramic radiography revealed bilateral partially corticated, radiolucent lesions on mandible. MRI showed, in right ramus mandible, an expansile, 37 mm 35 mm 28 mm size mass which made cortical irritation, spread to tooth root and surrounded by a thin bony shell and in left ramus mandible, an expansile, 30 mm 38 mm 12 mm size mass which made cortical irritation, spread to tooth root and surrounded by a thin bony shell (Fig. 2a–c). The patient underwent surgical curettage of the lesion through an intraoral approach under general anesthesia. After the thin bony shell was removed, the reddish, friable and granulomatous material was curetted and excised. Then the cavity was drilled with diamond burr until obtaining a clear surgical margin. After surgical curettage the stabilization of mandible was very well but there was wide cortical bone defect on mandible so the cavity reconstruction was done with cortical bone grafts from iliac crest
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and graft stabilization was made with biodegradable plate and screw (which was made polylactic acid/polyglycolic acid combination). Then the wound was closed with interrupted sutures. The post-operative course was uneventful, and she was discharged on post-operative day 3. The histopathologic examination revealed cellular vascular connective tissue with osteoclast-type giant cells having multiple nuclei and granulation tissue rich in mononuclear inflammatory cells (Fig. 3a and b). The lesion was reported as ‘giant cell reparative granuloma’. No evidence of clinical and radiological recurrence was observed during follow-up for 10 months and the grafts were integrated during this time. 2. Discussion Most of the multifocal lesion of CGCRGs are associated with some form of inherited syndrome or systemic disease, such as brown tumor of hyperparathyroidism [38], fibrous dysplasia, ossifying fibroma [39], paget disease, fibro-osseous lesion or odontogenic fibroma [40].
Fig. 1. (a–c) Preoperative image of patients.
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Fig. 2. (a–c) MR image of patients.
Multiple CGCRGs of the jaw bones usually associated with hyperparathyroidism in the majority of cases [17]. Brown tumors of hyperparathyroidism may occur at any age [25]. This can occur either primarily, due to hyperplasia or neoplasia of the parathyroid glands, or secondarily in association with renal osteodystrophy [41]. Hypercalcemia, hypophosphatemia, and elevated parathyroid hormone blood levels characterize hyperparathyroidism. In our patient, an endocrine workup was unremarkable, effectively excluding hyperparathyroidism. Only a few cases of multiple CGCRG of the jaws in the absence of hyperparathyroidism have been reported in the English literature [42,43]. Bloom et al. and Hjorting-Hansen and Worsoe-Petersonzhave presented cases of multiple recurrence giant cell lesions involving the jaws and long bones [44]. These cases were probably giant cell tumors or low-grade osteosarcomas and are histologically dissimilar to giant cell granuloma. Davis and Tideman reported a case of CGCRG in the mandible and the maxilla [45]. The maxillary lesion was detected 4 months after the mandibular lesion was first seen. Cassatly et al. presented case of CGCRG in the mandible [43], and Smith et al. presented a case of CGCRGs in both maxillary sinuses [42].
A number of syndromes have been reported to be associated with multiple giant cell lesions of the jaws, including cherubism, Noonan syndrome [46,47] neurofibromatosis type-1 [24,48], Ramon syndrome (gingival fibromatosis, hypertrichosis, epilepsy, mental and somatic retardation, and cherubism-like lesions) [49], Noonan-like syndrome, cherubism, and polyarticular pigmented villonodular synovitis (multiple lesions of pigmented villonodular synovitis affecting multiple bones, commonly the knees and ankles) [47]. Cherubism, an autosomal dominantly inherited condition with variable expressivity, is characterized by multiquadrant radiolucent lesions of the jaws and a progressive and clinically, symmetrical enlargement of the mandible and/or the maxilla. There is usually a family history of similarly affected family members. Regression is often seen following puberty [50]. Noonan syndrome is a relatively common congenital syndrome: cardiac anomalies, short stature and atypical face like a broad or webbed neck, low set and posteriorly angulated ears, ptosis, hypertelorism, and downward-slanting eyes [51]. Similar to cherubism, Noonan syndrome is an autosomal dominantly inherited syndrome with variable expressivity. Our case has no family history, no clinical appearance like cherubism or Noonan syndrome (Fig. 1a–c), no
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Fig. 3. (a and b) Histological appearances of lesions; nest of osteoclast-type giant cell alternate with fibrovascular stroma. The lamellar bone on the left side is seen.
systemic anomalies and swelling has been developed gradually for the last 6 months. 3. Conclusion Our patient had bilateral CGCRG in her jaw. In literature there are several reports about multiple CGCRG but unlike of that report our patient had no syndromes like cherubism, Noonan syndrome, neurofibromatosis type-1 and systemic disease like hyperparathyroidism, fibrous dysplasia. So we define this case as idiopathic bilaterally central giant cell reparative granuloma of jaw. References [1] H.L. Jaffe, Giant-cell reparative granuloma, traumatic bone cyst, and fibrous (fibroosseous) dysplasia of the jawbones, Oral Surg. Oral Med. Oral Pathol. 6 (1953) 159–175. [2] L. Barnes, J.W. Eveson, P. Reichart, Pathology and genetics of head and neck tumour, in: P. Kleihues, L.H. Sobin (Series Eds.), World Health Organization Classification of Tumours, IARC Press, Lyon, France, 2005, p. 324. ¨ stu¨ndag˘, M. I˙s¸eri, G. Keskin, B. Mu¨ezzinog˘lu, Central giant cell granuloma, Int. [3] E. U J. Pediatr. Otorhinolaryngol. 65 (2002) 143–146. [4] P.L. Auclair, P. Cuenin, F.J. Kratochvil, L.J. Slater, G.L. Ellis, A clinical and histomorphologic comparison of the central giant cell granuloma and the giant cell tumor, Oral Surg. Oral Med. Oral. Pathol. 66 (1988) 197–208. [5] L.T. Austın Jr., D.C. Dahlın, R.Q. Royer, Giant cell reparative granuloma and related condition affecting the jawbones, Oral Surg. Oral Med. Oral Pathol. 12 (1959) 1285–1295. [6] I. Kaffe, L. Ardekian, S. Taicher, M.M. Littner, A. Buchner, Radiologic features of central giant cell granuloma of the jaws, Oral Surg. Oral Med. Oral Pathol. Oral Radiol. Endod. 81 (1996) 720–726. [7] C.A. Waldron, W.G. Shafer, The central giant cell reparative granuloma of the jaws: an analysis of 38 cases, Am. J. Clin. Pathol. 45 (1966) 437–447.
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