Aravind Thara, Savithri Vindhya, Suresh Rakesh, Subash Pramod
Department of Oral Pathology and Microbiology, Amrita School of Dentistry, Amrita Vishwa Vidyapeetham, AIMS Campus, Kochi, Kerala, India.
J Oral Maxillofac Pathol. 2023 Feb;27(Suppl 1):S56-S59. doi: 10.4103/jomfp.jomfp_463_20. Epub 2023 Feb 4.
Nevoid basal cell carcinoma syndrome (NBCCS) is a rare autosomal dominant disorder characterized by a wide range of developmental abnormalities and a predisposition to neoplasms. In majority of the cases, the presence of multiple and recurrent jaw cysts especially during the first two decades of life is one of the first symptoms of this syndrome. We present here a case of 14-year-old female patient who reported with a chief complaint of facial swelling for 3 weeks. The radiographs revealed multiple cysts in maxilla and mandible. Incisional biopsy of the lesions was done and the histopathologic features were suggestive of odontogenic keratocyst. Further investigations revealed the presence of falx cerebri calcifications and multiple nevi on palms and feet. Genetic study was done to confirm the diagnosis of NBCCS, which showed mutations in PTCH gene. This case stresses the importance of genetic study in suspected cases of NBCCS especially in young patients of nonsyndromic parents.
痣样基底细胞癌综合征(NBCCS)是一种罕见的常染色体显性疾病,其特征为广泛的发育异常和肿瘤易感性。在大多数病例中,尤其是在生命的前二十年出现多发且复发的颌骨囊肿是该综合征的首发症状之一。我们在此报告一例14岁女性患者,其主要诉求为面部肿胀3周。影像学检查显示上颌骨和下颌骨有多个囊肿。对病变进行了切开活检,组织病理学特征提示为牙源性角化囊肿。进一步检查发现大脑镰钙化以及手掌和足部有多个痣。进行了基因研究以确诊NBCCS,结果显示PTCH基因存在突变。该病例强调了在疑似NBCCS病例中进行基因研究的重要性,尤其是对于父母无综合征的年轻患者。