Department of Otorhinolaryngology, Head and Neck Surgery, Kitasato University School of Medicine, 1-15-1 Kitasato, Minami-ku, Sagamihara, Kanagawa, 252-0374, Japan.
BMC Infect Dis. 2020 Jun 5;20(1):399. doi: 10.1186/s12879-020-05123-2.
Disseminated cryptococcosis is a well-characterized complication in immunocompromised patients with cryptococcal pneumonia or meningitis; however, isolated cryptococcal osteomyelitis is a rare entity that occurs in approximately 5% of patients with cryptococcosis. Cryptococcal osteomyelitis in the head and neck region is extremely rare. To the best of our knowledge, no cases of cryptococcal osteomyelitis affecting only the zygomatic bone have been reported to date.
A 78-year-old man without other comorbidities presented with progressive swelling of the right cheek along with pain and trismus. Clinical examination revealed a tender swelling in the right zygomatic region; the maximal mandibular opening was about 2 cm. Laboratory data showed mildly elevated inflammatory indices (C-reactive protein: 0.45 mg/dL; erythrocyte sedimentation rate: 35 mm/h). Computed tomography showed a 30-mm-diameter lesion at the right zygomatic arch. A part of the lesion has extended to the subcutaneous area of the cheeks with signs of bone destruction and surrounding contrast effects. Histopathological examination of fine-needle aspirate and needle biopsy showed cryptococcus. Furthermore, culture of the aspirate showed growth of Cryptococcus neoformans. No evidence of any other site involvement was observed. Therefore, the patient was diagnosed with isolated cryptococcal osteomyelitis and was initiated on fluconazole therapy. The treatment was effective, and all symptoms were resolved in 4 weeks. Fluconazole therapy was stopped after 6 months. There are no signs of recurrence as of 15-month follow-up. The patient has no cosmetic abnormalities or sequelae.
Fine-needle aspiration cytology, needle biopsy, and fungal culture were useful for definitive diagnosis. Immunocompetent patients with isolated osteomyelitis may be cured with oral fluconazole alone.
播散性隐球菌病是免疫功能低下的隐球菌性肺炎或脑膜炎患者的一种特征性并发症;然而,孤立性隐球菌性骨髓炎是一种罕见的实体,约发生在 5%的隐球菌病患者中。头颈部的隐球菌性骨髓炎极为罕见。据我们所知,目前尚无仅累及颧骨的隐球菌性骨髓炎病例报告。
一名 78 岁的男性,无其他合并症,出现右侧脸颊进行性肿胀,伴有疼痛和牙关紧闭。临床检查发现右侧颧骨区域有压痛性肿胀;最大下颌开口约 2cm。实验室数据显示炎症指标轻度升高(C 反应蛋白:0.45mg/dL;红细胞沉降率:35mm/h)。计算机断层扫描显示右侧颧骨弓有一个 30mm 直径的病变。部分病变已延伸至脸颊的皮下区域,伴有骨破坏和周围对比效应的迹象。细针抽吸和针活检的组织病理学检查显示隐球菌。此外,抽吸物的培养显示新生隐球菌生长。未发现其他部位受累的证据。因此,患者被诊断为孤立性隐球菌性骨髓炎,并开始氟康唑治疗。治疗有效,4 周内所有症状均得到缓解。6 个月后停止氟康唑治疗。截至 15 个月随访时,无复发迹象。患者无美容异常或后遗症。
细针抽吸细胞学、针活检和真菌培养对明确诊断很有用。免疫功能正常的孤立性骨髓炎患者仅口服氟康唑即可治愈。