慢性非细菌性骨髓炎(CNO)是一种慢性、无菌,炎症性疾病。它主要表现为非特异性骨疼痛和肿胀,但最终会导致骨破坏和畸形,如果不及时治疗。在CNO中很少涉及颅骨(下颌骨除外)。在这份报告中,我们介绍了一例罕见的CNO影响面部和颅骨的病例,表现为面神经麻痹,并回顾了有关类似影响的文献。一个10岁的孩子,先前健康的女性最初被评估为左侧面部肿胀,触诊时轻微压痛,但没有发烧.她的全血细胞计数并不显著,她的炎症标志物升高(C反应蛋白7.5mg/dl和红细胞沉降率104mm/h),面部和颅骨的CT和大脑的MRI显示了涉及左上颌骨的破坏性骨过程,颧骨,蝶骨和斜坡.左上颌骨骨活检显示纤维发育不良伴脓肿形成,最符合感染性病因(急性骨髓炎)。她开始口服克林霉素3个月的疗程。开始使用克林霉素后面部肿胀得到改善,但在她治疗的第六周,她出现了右侧贝尔氏麻痹。大脑的MRI显示右第七颅神经过度增强。一个月后,她接受了右手腕和膝盖肿胀的评估,疼痛,和运动的限制。骨骼检查和MRI显示多灶性病变伴有混合性硬化和清醒。她的炎症标志物继续升高。右桡骨的另一个骨活检显示类似的破坏发现,没有恶性肿瘤的证据。她最终被诊断出患有CNO。她开始服用非甾体抗炎药,并进行胃保护和定期随访。经过一年多的随访,患者的炎症标志物保持正常,和关节肿胀/限制一直在缓解。我们在文献中发现了另外5个类似的案例。据我们所知,我们的患者是美国第一例报告的病例,涉及除下颌骨外的颅骨/面部骨骼,并伴有面部麻痹。CNO中面部骨骼(下颌骨除外)的影响非常罕见,但是临床医生对这种表现的认识是达到诊断的一个重要方面。
Chronic nonbacterial osteomyelitis (CNO) is a chronic, sterile, inflammatory disease. It primarily presents with nonspecific bone pain and swelling but ultimately can cause bone destruction and deformities, if left untreated. The involvement of the cranial bones (apart from the mandible) is rare in CNO. In this report, we present a rare case of CNO affecting facial and cranial bones presenting as facial palsy with a
review of the literature about similar affection. A 10-year-old, previously healthy female was initially evaluated for swelling of the left side of her face with slight tenderness on palpation, but no fever. Her complete blood count was unremarkable, her inflammatory markers were elevated (C-reactive protein 7.5 mg/dl and erythrocyte sedimentation rate 104 mm/h), and CT of facial and skull bones and MRI of brain showed a destructive osseous process involving the left maxillary, zygomatic, sphenoid bones and the clivus. Bone biopsy of the left maxilla showed fibrous dysplasia with abscess formation, most consistent with an infectious aetiology (acute osteomyelitis). She was started on oral clindamycin for a 3-month course. The facial swelling improved after starting clindamycin, but on her sixth week of treatment, she developed right-sided Bell\'s palsy. An MRI of the brain showed hyperenhancement of the right seventh cranial nerve. A month later, she was evaluated for right wrist and knee swelling, pain, and limitation of movement. Skeletal survey and MRI showed multifocal lesions with mixed sclerosis and lucency. Her inflammatory markers continued to be elevated. Another bone biopsy of the right radius showed similar findings of destruction with no evidence of malignancy. She was ultimately diagnosed with CNO. She was started on nonsteroidal anti-inflammatory drugs with gastric protection and regular follow-up. Over more than a year of follow-up, the patient\'s inflammatory markers remain normal, and joint swelling/limitation has remained in remission. We found five additional cases in the literature that presented with a similar presentation. To our knowledge, our patient is the first reported case in the USA involving the cranial/facial bones apart from the mandible presenting with facial palsy. The affection of the facial bones (apart from the mandible) in CNO is very rare, but the awareness of such a presentation by the clinician is an important aspect of reaching the diagnosis.