A case of congenital infiltrating lipomatosis of the face with the early eruption of permanent teeth with a review of the English literature

IF 0.4 Q4 DENTISTRY, ORAL SURGERY & MEDICINE Journal of Oral and Maxillofacial Surgery Medicine and Pathology Pub Date : 2024-02-15 DOI:10.1016/j.ajoms.2024.02.004
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Abstract

Congenital infiltrating lipomatosis of the face (CILF) is a benign non-epithelial tumor that was initially reported in 1983. Although lipomas are generally encased in fibrous tissue, CILF lacks a fibrous capsule and is characterized by intramuscular invasion. We herein report a case of CILF diagnosed based on the early eruption of permanent teeth and reviewed the English literature. The patient was a 5-year-old male with diffuse swelling of the left upper canine gingiva. He had undergone surgery at 3 years old to debulk left buccal lipoma. At presentation, facial asymmetry and diffuse swelling of the left cheek were noted. Radiographic examinations revealed the early eruption of and accelerated root formation by the left upper permanent teeth, bony hyperplasia of the left maxilla and zygomatic bone, and adipose hyperplasia of the left cheek region. Hormone examinations revealed no abnormalities. A histopathological specimen obtained from surgery to reduce buccal lipoma was re-evaluated and diagnosed as infiltrative lipoma. Based on histopathological and clinical features, the patient was diagnosed with congenial infiltrative lipoma was established. Conservative therapy was performed in consideration of future surgical interventions after adolescence.

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一例面部先天性浸润性脂肪瘤伴恒牙早萌病例及英文文献综述
面部先天性浸润性脂肪瘤病(CILF)是一种良性非上皮性肿瘤,最初于 1983 年被报道。虽然脂肪瘤一般被纤维组织包裹,但 CILF 没有纤维囊,其特点是肌肉内侵。我们在此报告一例因恒牙早萌而被诊断为 CILF 的病例,并回顾了英文文献。患者是一名 5 岁的男性,左上犬齿龈弥漫性肿胀。他在3岁时曾接受过左侧口腔脂肪瘤剥除手术。发病时,他的面部不对称,左侧脸颊弥漫性肿胀。影像学检查显示,左上恒牙提前萌出,牙根形成加快,左上颌骨和颧骨骨质增生,左脸颊部脂肪增生。激素检查未发现异常。对手术缩小口腔脂肪瘤获得的组织病理学标本进行了重新评估,诊断为浸润性脂肪瘤。根据组织病理学和临床特征,患者被确诊为先天性浸润性脂肪瘤。考虑到青春期后的手术治疗,对患者进行了保守治疗。
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来源期刊
CiteScore
0.80
自引率
0.00%
发文量
129
审稿时长
83 days
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