Lee Dong-Hoon, Moon Jun-Ki
Department of Orthopedic Surgery, College of Medicine, Chung-Ang University Gwangmyeong Hospital, Chung-Ang University, Seoul, Republic of Korea.
Department of Orthopedic Surgery, College of Medicine, Chung-Ang University Hospital, Chung-Ang University, Seoul, Republic of Korea.
BMC Musculoskelet Disord. 2024 Oct 1;25(1):765. doi: 10.1186/s12891-024-07882-4.
Xanthogranulomatous osteomyelitis (XO) is a rare disease characterized radiologically by an osteolytic lesion with cortical expansion or disruption. Differentiating this condition from other osteolytic diseases such as primary or metastatic bone neoplasms is imperative. Several case reports have been published on XO, with previous reports predominantly identifying bacteria such as Pseudomonas or Staphylococcus as causative organisms. However, fungal infection-induced XO has not yet been reported.
We present the case of a 23-year-old woman with a tumor-like osteolytic lesion in the pubic bone. The patient had experienced pelvic pain and intermittent febrile episodes for 2 months. Plain radiography revealed an osteolytic lesion in the right pubic tubercle. Magnetic resonance imaging suggested a cystic bone tumor or tubercular infection. Surgical intervention included curettage of the lesion and irrigation with normal saline. Histopathological examination of the specimen revealed abundant foamy histiocytes with inflammatory infiltrates consistent with XO. Culture of the osteolytic lesion confirmed an Aspergillus species infection and antifungal treatment was initiated. At 1-year follow-up, no evidence of local recurrence was observed.
Although rare, XO requires differentiation from similar conditions and is treated with surgical intervention and targeted medical therapy based on the identified organisms. Clinicians should be mindful that XO can also be induced by fungal infections and that combination antifungal treatments may be beneficial in such cases.
黄色肉芽肿性骨髓炎(XO)是一种罕见疾病,其影像学特征为伴有皮质膨胀或破坏的溶骨性病变。将这种疾病与其他溶骨性疾病,如原发性或转移性骨肿瘤区分开来至关重要。关于XO已经发表了几例病例报告,先前的报告主要将铜绿假单胞菌或葡萄球菌等细菌鉴定为致病微生物。然而,真菌感染引起的XO尚未见报道。
我们报告一例23岁女性,耻骨出现肿瘤样溶骨性病变。患者有盆腔疼痛和间歇性发热发作2个月。X线平片显示右耻骨结节有溶骨性病变。磁共振成像提示为囊性骨肿瘤或结核感染。手术干预包括病变刮除术并用生理盐水冲洗。标本的组织病理学检查显示有大量泡沫状组织细胞,伴有与XO一致的炎性浸润。溶骨性病变培养证实为曲霉菌感染,并开始抗真菌治疗。随访1年,未观察到局部复发迹象。
尽管XO罕见,但需要与类似疾病相鉴别,并根据鉴定出的病原体进行手术干预和针对性药物治疗。临床医生应注意,XO也可由真菌感染引起,在这种情况下联合抗真菌治疗可能有益。