A Rare Case of Cherubism In a 23-Year-Old Man

A. Heidary, M. Akhavankarbasi, Yasaman Sabaghzadgan, Ehsan Babaie
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Abstract

Background and Objectives: Cherubism is a rare hereditary autosomal dominant fibro-osseous disease that is characterized by painless, bilateral, symmetric swelling in the jaw with multilocular radiolucent lesions in the maxilla, mandible, or both, and give the patient a typical “cherubic” appearance. It occurs in children, especially boys, aged 2-5 years. The aim of this study was to report a case of cherubism at an uncommon age that did not improve with age. Case Presentation: The patient was a 23-year-old man complaining of bilateral jaw pain along with swelling and multiple bilateral radiolucent lesions with a previous history of childhood cherubism. In the examination, bilateral and symmetric swelling of the face, especially in the maxilla region, with eyes raised to heaven were evident. The pain was more in the vicinity of the condyle. No abnormal manifestations were observed by the intraoral examination. In the radiographic view, multiple bilateral and symmetric lucent lesions were observed on trunk and mandible, continued to the neck of the condyle. Conclusion: Despite the common manifestations and age in patients, cherubism can show different manifestations; lack of attention to the patient’s history can lead to misdiagnosis by the pathologist and unnecessary treatments. Due to being a self-limiting condition, non-invasive treatments are preferred; however, surgical management is sometimes required for cosmetic reasons.
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一名23岁男子罕见的小天使症
背景和目的:小天使病是一种罕见的常染色体显性遗传性纤维骨性疾病,其特征是颌骨无痛、双侧对称肿胀,并在上颌骨、下颌骨或两侧有多室放射性病变,患者表现为典型的“小天使”外观。它发生在2-5岁的儿童中,尤其是男孩。本研究的目的是报告一个不寻常年龄的小天使症,并没有随着年龄的增长而改善。病例介绍:患者23岁,男,主诉双侧下颚疼痛伴肿胀及双侧多发透光病灶,既往有儿童小天使病史。在检查中,两侧对称的面部肿胀,特别是在上颌骨区域,眼睛向上抬起明显。疼痛主要发生在髁突附近。口腔内检查未见异常表现。x线片上,躯干和下颌骨可见多发双侧对称透明病灶,并延续至髁突颈部。结论:小天使病虽有常见的表现和年龄,但表现各异;缺乏对患者病史的关注可能导致病理学家的误诊和不必要的治疗。由于是一种自限性疾病,首选非侵入性治疗;然而,出于美观的原因,有时需要手术治疗。
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