Sambyal Shruti S, Dinkar Ajit D, Jayam Cheranjeevi, Singh Bhanu Pratap
Department of Oral Medicine, Himachal Pradesh Government Dental College and Hospital, Shimla, Himachal Pradesh, India.
Department of Oral Medicine, Goa Dental College and Hospital, Bambolim, Goa, India.
BMJ Case Rep. 2016 Sep 21;2016:bcr2016216854. doi: 10.1136/bcr-2016-216854.
A 3-year-old girl child presented with swelling in her right lower jaw that had started 5 days previously. History revealed the child being non-immunised. Initial-evaluation revealed proptosis and bony hard swelling over the right body of the mandible. Radiological evaluation including a CT scan indicated expansile osteolytic lesion involving the body-ramus with onion-peel periosteal reaction suggesting osteomyelitis/malignancy. Blood investigations showed raised erythrocyte sedimentation rate and eosinophilia. Family screening for tuberculosis (TB) revealed that the patient, her father and siblings were PPD positive though chest screening and sputum examinations were negative in all of them. Lesion biopsy showed acute/chronic osteomyelitis with eosinophilia, tilting diagnosis towards eosinophilic granuloma/Hand-Schuller-Christian disease. Further investigations for diabetes insipidus, histiocytosis-X, skull-pelvic-femur radiographs, abdominal ultrasonography, ophthalmic consultation and PCR-TB test were negative. Histology, radiography, purified protein derivative (PPD)-positive result and history led to the diagnosis of primary TB of the mandible. Complete resolution occurred following antitubercular therapy. The case report emphasises that diagnosis of TB can sometimes be based on circumstantial evidence and positive response to antitubercular therapy even in view of a negative PCR result.
一名3岁女童出现右下颌肿胀,该症状始于5天前。病史显示该儿童未接种疫苗。初步评估发现眼球突出以及下颌骨右侧体部有骨质坚硬的肿胀。包括CT扫描在内的影像学评估显示,病变累及下颌体-升支,呈膨胀性溶骨性病变,并伴有葱皮样骨膜反应,提示骨髓炎/恶性肿瘤。血液检查显示红细胞沉降率升高和嗜酸性粒细胞增多。对该家庭进行结核病(TB)筛查发现,患者及其父亲和兄弟姐妹结核菌素试验均为阳性,尽管他们所有人的胸部筛查和痰液检查均为阴性。病变活检显示为伴有嗜酸性粒细胞增多的急/慢性骨髓炎,这使诊断倾向于嗜酸性肉芽肿/汉-许-克病。针对尿崩症、组织细胞增多症-X、颅骨-骨盆-股骨X线片、腹部超声检查、眼科会诊和结核分枝杆菌聚合酶链反应(PCR-TB)检测的进一步检查均为阴性。组织学、影像学、纯化蛋白衍生物(PPD)阳性结果及病史共同得出下颌骨原发性结核的诊断。抗结核治疗后病情完全缓解。该病例报告强调,即使PCR结果为阴性,结核病的诊断有时也可基于间接证据和对抗结核治疗的阳性反应。