Routray Samapika, Adhya Amit Kumar, John Joseph, Dikhit Punit
Department of Dentistry, All India Institute of Medical Sciences, Bhubaneswar, Odisha, India.
Department of Pathology, All India Institute of Medical Sciences, Bhubaneswar, Odisha, India.
BMJ Case Rep. 2019 Jul 23;12(7):e229483. doi: 10.1136/bcr-2019-229483.
A 6-year-old child with an episodic history of ulcerations over buccal mucosa was found to have severe inflammation on the palatal aspect of permanent first molars with grade 2 mobility bilaterally. Radiographical features were suggestive of bone loss around permanent molars extending to the distal aspect of the deciduous first molars. The clinical and radiographical findings were indicative of periodontal degeneration without any apparent cause visible intraorally. Further biopsy was done from the rashes present on the malar prominences, which showed nodular aggregates of atypical cells in superficial dermis. These large histiocytic cells with vesicular nuclei and nuclear grooves were immunopositive for CD1a and S100, concluding the diagnosis of Langerhans cell histiocytosis. For treatment, patient was referred to Department of Haemato-oncology and chemotherapy was suggested as per protocol.
一名6岁儿童有颊黏膜溃疡发作史,发现双侧恒上第一磨牙腭侧有严重炎症,牙齿松动Ⅱ度。影像学特征提示恒磨牙周围骨质丧失,延伸至乳上第一磨牙远中面。临床和影像学检查结果表明存在牙周组织变性,口腔内未发现明显病因。对颧突处出现的皮疹进行了进一步活检,结果显示真皮浅层有非典型细胞的结节状聚集。这些具有泡状核和核沟的大组织细胞对CD1a和S100免疫呈阳性,确诊为朗格汉斯细胞组织细胞增多症。为进行治疗,该患者被转诊至血液肿瘤科,并根据方案建议进行化疗。