Clinicopathologic study of 24 cases of cherubism

Meng, Xuemei; Yu, Shaohua; Yu, Guohua

doi:10.1016/j.ijom.2004.09.006

Cited by 90 publications

(103 citation statements)

References 30 publications

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“…Furthermore, early tumor removal in the disease process, as advocated by the current report, could prevent any long-term sequelae requiring significantly more complex reconstructive surgery. Additionally, it has also been shown that early operation, with curettage during the growth phase can arrest the tumor growth, and not prompt rapid regrowth, thereby making it a favorable option in preventing further bony deformities [7,13,15,16] . Our report further strengthens this group of patients who have good outcomes with arrested tumor growth and excellent facial contouring following early surgical intervention.…”

Section: Discussionmentioning

confidence: 99%

“…Surgical treatment modalities used in the management of patients with cherubism range from tooth extractions in the lesioned areas [16] , orthodontics, fixed and removable prosthetic implants [17] , osteoplastic surgery [13] to intraosseous curettage of lesions and bone grafting [13] . Taking advantage of the qualities of the Piezo Electric bone cutter, our surgical approach was achieved through minimal soft tissue disruption, but allowed for aggressive tumor removal, as well as mandibular cortical repositioning.…”

Section: Discussionmentioning

confidence: 99%

“…The current recommended approach to the management of most cases of cherubism is to "wait and watch" into late adolescence, with the hope of spontaneous disease remission and therefore avoidance of early surgery. Early surgical intervention is usually advocated only for severe cases [7,[11][12][13][14] .…”

Section: Introductionmentioning

confidence: 99%

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Cherubism in a 4-year-old boy managed with tumor curettage, mandibular osteotomies and repositioning

Garlick¹,

Willis²,

Donato³

et al. 2018

PAR

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Cherubism is a rare disease characterized by significant loss of medullary bone which is replaced by excessive amounts of fibrous tissue growth within the mandible and maxilla. We present a case of a 4-year-old boy with a rapidly enlarging mandible and maxilla, causing significant change in the facial contour, malocclusion and phonation difficulties. He was treated with aggressive tumor curettage, lateral mandibular cortex osteotomies with medial repositioning. This allowed obliteration of the enlarged medullary space and restoration of the normal mandibular anatomy. At 12 months postoperatively, the patient had significant improvement in facial contour, normal outward appearance, and stable dentition.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

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Cherubism in a 4-year-old boy managed with tumor curettage, mandibular osteotomies and repositioning

Garlick¹,

Willis²,

Donato³

et al. 2018

PAR

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“…10,11 The children affected with cherubism do not usually show mental or physical deformities, but when cherubism is associated with other syndromes like Noonan-like syndrome, 13,14 Ramon syndrome, 15 fragile X syndrome, mental and physical deformities may be seen. 16 Grading of cherubism based on the location of the jaw lesions have been described by Seward and Hankey 17 as follows:…”

Section: Discussionmentioning

confidence: 99%

Ramon's Syndrome: A Rare Entity

Lnu¹,

Peter²,

Nampoothiri³

et al. 2010

World Journal of Dentistry

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Introduction Ramon et al in 1967 described a condition, which included mental retardation, fibrous dysplasia of the maxilla and stunted growth. De Pino et al described a Brazilian family of four who had the same features as that of Ramon's syndrome in association with juvenile arthritis. Cherubism was first described in 1933 by Jones as ‘familial multilocular cystic lesion of the jaws’, a rare benign fibroosseous disease of the jaws, which is transmitted as an autosomal dominant trait. Affected children usually present before five years of age with painless progressive swelling of the cheeks, frequently associated with dental malformations. It progresses until puberty, and shows partial or complete spontaneous involution in adulthood; therefore, management is mostly conservative. The condition was initially characterized as familial, particularly as a form of craniofacial fibrous dysplasia. The children affected with cherubism do not usually show mental or physical deformities, but when cherubism is associated with other syndromes like Noonan-like syndrome, Ramon syndrome, and Fragile X syndrome, mental and physical deformities may be seen. Case Report This is a case report of a 12-year-old boy who reported with a massive painless bilateral swelling of the face, which has been increasing since the age of 2 years. The patient had multiple unerupted teeth, gingival hyperplasia, hearing loss and mental retardation. A detailed case report, including the histopathology, radiographic features (extraoral, CT and MDCT), and management of the case will be discussed in detail.

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“…No unusual biochemical findings have been reported in patients with cherubism. The most useful biochemical investigations [6] are calcium, parathyroid hormone (PTH) and alkaline phosphatase level which were within normal range and serve to differentiate it from hyperparathyroidism in which all of them are elevated. However, elevated alkaline phosphatase level was observed in this case which indicates bone formation.…”

Section: Case Reportmentioning

confidence: 99%

Cherubism: A Case Report

Degala

Mahesh

Monalisha³

2013

J. Maxillofac. Oral Surg.

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Cherubism is a benign, self-limiting fibro-osseous lesion characterized by bilateral symmetric painless expansion of jaw which is more prominent in mandible than in maxilla. Males are commonly affected (2:1) and with greater severity. It becomes noticeable in early childhood and gradually regresses after puberty. Although cherubism is considered as a familial/inherited disease but many sporadic cases have been reported in literature with no familial history. Osteoblastic and osteoclastic remodeling replaces normal bone by excessive amount of fibrovascular tissue containing multinucleated giant cells. Here, we present a case report of cherubism in a 10 year old boy describing the clinical, histological, biochemical and radiographic features.

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Clinicopathologic study of 24 cases of cherubism

Cited by 90 publications

References 30 publications

Cherubism in a 4-year-old boy managed with tumor curettage, mandibular osteotomies and repositioning

Cherubism in a 4-year-old boy managed with tumor curettage, mandibular osteotomies and repositioning

Ramon's Syndrome: A Rare Entity

Cherubism: A Case Report

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