Surgical management of severe cherubism persisting into early adulthood: a case report and literature review.

Q2 Medicine Archives of Craniofacial Surgery Pub Date : 2024-02-01 Epub Date: 2024-02-20 DOI:10.7181/acfs.2023.00577
Youngwoong Choi, Jeong Min Ji, Choong Hyeon Kim, Ki Pyo Sung
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Abstract

Cherubism is a rare fibro-osseous condition characterized by bilateral expansion of the mandible and maxilla. Due to its rarity, treatment guidelines for cherubism have not been clearly established. Observation without surgical intervention is typically recommended, as cherubism often regresses spontaneously after puberty. However, a surgical intervention may be necessary if aggressive lesions lead to severe complications. In this report, we present a case involving surgical management of cherubism that did not spontaneously regress until early adulthood. An 18-year-old man was diagnosed with cherubism, presenting characteristic upward-looking eyes and a swollen face. He strongly desired surgical management. Gross contouring of the mandible was performed using an osteotome. Subsequently, delicate contouring was performed by bone burring and curettage. The remaining multiple locular bony defects were filled with demineralized bone matrix. No major complications, including infection and hematoma, occurred during the 8-month follow-up period. The facial contour remained stable without the aggravation of cherubism. The patient was satisfied with the cosmetic results. Considering that cherubism is a rare disease globally, with few reported cases in Korea, and that treatment guidelines are not clearly established, we anticipate that the results of this case will contribute to the development of future protocols for treating cherubism.

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严重樱桃小嘴症持续到成年早期的手术治疗:病例报告和文献综述。
樱桃小嘴症是一种罕见的纤维骨病,其特点是下颌骨和上颌骨双侧膨大。由于其罕见性,小天使症的治疗指南尚未明确制定。由于小天使症通常会在青春期后自然消退,因此通常建议观察而不进行手术干预。然而,如果侵袭性病变导致严重并发症,则有必要进行手术治疗。在本报告中,我们介绍了一例小天使症手术治疗病例,该病例直到成年早期才自发消退。一名 18 岁的男子被诊断患有樱桃小嘴症,表现为特征性的眼睛上翘和面部浮肿。他强烈希望接受手术治疗。他使用截骨器对下颌骨进行了粗轮廓修整。随后,通过骨钻孔和刮除术进行了精细的轮廓修整。余下的多处局部骨缺损由脱矿骨基质填充。在 8 个月的随访期间,没有发生感染和血肿等重大并发症。面部轮廓保持稳定,樱桃小嘴没有加重。患者对美容效果感到满意。考虑到樱桃核是一种全球罕见的疾病,在韩国也鲜有报道,而且治疗指南尚未明确制定,我们预计本病例的结果将有助于未来樱桃核治疗方案的制定。
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来源期刊
Archives of Craniofacial Surgery
Archives of Craniofacial Surgery Medicine-Otorhinolaryngology
CiteScore
2.90
自引率
0.00%
发文量
44
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