Simultaneous occurrence of an odontogenic keratocyst and giant cell granuloma-like lesion in the mandible

Yoon, Jung Hoon; Kim, S.G; Lee, S.H; Kim, J

doi:10.1016/j.ijom.2003.09.007

Cited by 13 publications

(8 citation statements)

References 8 publications

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“…Dysplastic dentin or cystic-like characteristics, however, are absent [17]. Rarely, CGCL have also been identified in proximity to other odontogenic lesions including ameloblastoma [28,29] and keratocystic odontogenic tumor (KCOT, odontogenic keratocyst) [30], probably representing an epiphenomenon.…”

Section: Discussionmentioning

confidence: 99%

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Clinicopathologic and Molecular Characteristics of Familial Cherubism with Associated Odontogenic Tumorous Proliferations

Argyris

Gopalakrishnan

et al. 2017

Head and Neck Pathol

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Cherubism is a rare autosomal dominant condition affecting the jaws and caused by mutations in the gene encoding for the adapter protein SH3BP2 that maps to chromosome 4p16.3. Cherubism is characterized by symmetrically developing bone lesions in the maxilla and mandible. The lesions have been radiographically and histopathologically well-described. Here, we present a family with cherubism with two of its members featuring odontogenic tumorous proliferations in association with persistent central giant cell lesions (CGCL). Specifically, the proband, a 25-year-old male, developed a radiolucent lesion characterized histologically by central odontogenic fibroma-like proliferation in association with a CGCL component, while his mother, at age 57, was diagnosed with primary intraosseous odontogenic carcinoma with areas of benign fibro-osseous lesions. In both patients the lesions occurred in the anterior mandible and presented with clinical enlargement. The son underwent incisional biopsy and did not have additional treatment. His mother underwent extensive mandibulectomy due to widespread tumor. The son has two affected children with classic cherubism while a third child at age 5, had not shown any features of the disease. Mutation analysis of three affected members resulted in the identification of a heterozygous mutation in SH3BP2 (c.1244G>C; p.Arg415Pro). To the best of our knowledge, association of cherubism with odontogenic neoplastic lesions has hitherto not been reported in the literature, thus suggesting a relationship between cherubism with disturbed odontogenesis.

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Section: Discussionmentioning

confidence: 99%

“…In addition, separate bilateral radiolucent lesions were observed in the maxilla. An incisional biopsy was performed between second right mandibular premolar and first molar (teeth # [29][30].…”

Section: Clinico-pathologic Characteristicsmentioning

confidence: 99%

Clinicopathologic and Molecular Characteristics of Familial Cherubism with Associated Odontogenic Tumorous Proliferations

Argyris

Gopalakrishnan

et al. 2017

Head and Neck Pathol

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“…The most striking feature here is the presence of numerous multinucleated giant cells in the connective tissue capsule. After its first report by Yoon et al in 2004, 3 only single attempt has been made to explain the pathogenesis of multinucleated giant cell component in KCOT. 4 The paucity of such reported cases only adds to the confusion regarding the presence of such giant cells in KCOT.…”

Section: Discussionmentioning

confidence: 99%

“…These lesions are relatively common in their own terms, but a lesion showing features of both is found to be reported only twice in the literature. 3,4 We report third such case of KCOT showing focal areas of GCG like lesion.…”

Section: Introductionmentioning

confidence: 87%

“…Although there are several case reports of KCOT associated with traumatic bone cyst, 6 ameloblastoma, 7 and KCOT containing cartilage or KCOT associated with intramandibular chondroma, 8 there are only two cases of KCOT with GCG features reported till date in literature, 3,4 likewise GCG is reported to occur with aneurysmal bone cyst, ossifying fibroma, osteoblastoma, chondroblastoma and odontogenic fibroma, 9 but not very commonly with KCOT. Hence, our report stands distinct from others.…”

Section: Jcdpmentioning

confidence: 99%

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Collision Lesion of Mandible—Coexistence of Keratocystic Odontogenic Tumor with Central Giant Cell Granuloma: A Rare Case Report

Ravi

Prashanthi²,

Karun³

et al. 2013

The Journal of Contemporary Dental Practice

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Central giant cell granuloma: treatment with calcitonin, triamcinolone acetonide, and a cystic finding 3 years and 6 months after the primary treatment

Sampieri

Yaedú

Santos

et al. 2012

Oral Maxillofac Surg

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Due to the diagnosis and the age of the patient, we chose a conservative treatment, administering subcutaneous injections of calcitonin. During this treatment, no reduction to the lesion was observed. Therefore, we chose to treat the lesion with triamcinolone acetonide. Monthly follow-ups demonstrated good lesion reduction and the absence of any clinical symptoms during the first 2 years. After a 3-year follow-up, the patient returned, presenting mobility of the lower incisors. A significant increase in the size of the lesion was observed. After a biopsy, with the removal of tissue which had the appearance of a cyst capsule, microscopic analyses were found to be compatible with a secondarily infected cyst. Two months following this procedure, the patient did not present tooth mobility anymore and the oral mucosa presented a normal aspect. Following a radiographic exam, full lesion repair was observed. These conservative treatments should be the first option in cases of central giant cell granuloma and the patient must be observed for a long period of time, until no further clinical or radiographic signs of lesions are observed.

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Simultaneous occurrence of an odontogenic keratocyst and giant cell granuloma-like lesion in the mandible

Cited by 13 publications

References 8 publications

Clinicopathologic and Molecular Characteristics of Familial Cherubism with Associated Odontogenic Tumorous Proliferations

Clinicopathologic and Molecular Characteristics of Familial Cherubism with Associated Odontogenic Tumorous Proliferations

Collision Lesion of Mandible—Coexistence of Keratocystic Odontogenic Tumor with Central Giant Cell Granuloma: A Rare Case Report

Central giant cell granuloma: treatment with calcitonin, triamcinolone acetonide, and a cystic finding 3 years and 6 months after the primary treatment

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