BACKGROUND Campylobacter jejuni is a motile, gram-negative rod known for causing self-limiting enterocolitis while rarely causing extraintestinal infections. We report the first case of a patient with Campylobacter jejuni osteomyelitis in both femora. CASE PRESENTATION A 54-year-old female presented with progressive pain in both upper extremities. Her past medical history mentioned a lymphoplasmacytic lymphoma (LPL) for which she had received dexamethasone, cyclophosphamide and fludarabine and was still receiving maintenance therapy with intravenous rituximab. Two months prior to presentation, she received oral fluoroquinolone for a recurrent enterocolitis with stool cultures positive for Campylobacter jejuni. Palpation of the left quadriceps femoris muscle was remarkably painful during physical examination. Laboratory testing showed elevated C-reactive protein and recent low gamma globulin levels. The presumptive diagnosis at this point was a transformation of LPL to a large B cell lymphoma. In order to determine the preferred site for biopsy, a fluorine-18 fluoro-2-deoxy-D-glucose positron emission tomography combined with computed tomography was done. However, blood cultures taken on admission showed growth of Campylobacter jejuni in both aerobic bottles, with a strain resistant to fluoroquinolones. Diagnosis of Campylobacter jejuni osteomyelitis was confirmed with 16S ribosomal RNA gene polymerase chain reaction performed on femoral bone obtained through biopsy. Treatment with intravenous imipenem/cilastatin followed by intravenous and oral doxycycline proved insufficient. Subsequently, the patient was treated successfully with intravenous meropenem for six weeks and concurrent intravenous immunoglobulin. CONCLUSION We report the first case of Campylobacter jejuni osteomyelitis in both femora in a patient with acquired hypogammaglobulinemia. Diagnosis was confirmed by blood cultures and positive 16S ribosomal RNA gene polymerase chain reaction for Campylobacter spp. on bone biopsy. Treatment was successful with intravenous meropenem and immunoglobulin. Our report showcases an unusual manifestation in a patient with immunodeficiency and discusses failure of initial antibiotic therapy.

中文翻译：

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空腹双侧空肠弯曲杆菌骨髓炎患者获得性低血球蛋白血症：一例病例报告。

背景技术空肠弯曲杆菌是一种运动性革兰氏阴性杆菌，以引起自限性小肠结肠炎而很少引起肠外感染而闻名。我们报告第一例两个股骨中空肠弯曲杆菌骨髓炎的患者。病例介绍一名54岁的女性在两个上肢都出现了进行性疼痛。她的既往病史提到了淋巴浆细胞性淋巴瘤（LPL），已接受地塞米松，环磷酰胺和氟达拉滨治疗，并且仍在接受利妥昔单抗静脉维持治疗。在就诊前两个月，她接受了氟喹诺酮口服治疗复发性小肠结肠炎，粪便培养物为空肠弯曲菌阳性。身体检查时，股四头肌左手触诊非常痛苦。实验室测试显示C反应蛋白升高，近期γ球蛋白水平降低。在这一点上的推定诊断是LPL向大B细胞淋巴瘤的转化。为了确定活检的首选部位，做了氟18氟-2-脱氧-D-葡萄糖正电子发射断层扫描与计算机断层扫描相结合。然而，入院时进行的血液培养显示在两个有氧瓶中空肠弯曲杆菌的生长，且菌株对氟喹诺酮类耐药。对通过活检获得的股骨进行的16S核糖体RNA基因聚合酶链反应证实了空肠弯曲菌骨髓炎的诊断。静脉注射亚胺培南/西司他丁，然后静脉注射和口服强力霉素治疗效果不佳。后来，该患者已成功接受美罗培南静脉注射治疗六周，并同时接受静脉注射免疫球蛋白治疗。结论我们报告了第一例空肠弯曲杆菌骨髓炎患者股骨低血球蛋白血症患者的两个股骨。血液培养和弯曲杆菌属的16S核糖体RNA基因聚合酶链反应阳性证实了诊断。进行骨活检。静脉用美罗培南和免疫球蛋白治疗成功。我们的报告显示了免疫缺陷患者的异常表现，并讨论了最初抗生素治疗的失败。血液培养和弯曲杆菌属的16S核糖体RNA基因聚合酶链反应阳性证实了诊断。进行骨活检。静脉用美罗培南和免疫球蛋白治疗成功。我们的报告显示了免疫缺陷患者的异常表现，并讨论了最初抗生素治疗的失败。血液培养和弯曲杆菌属的16S核糖体RNA基因聚合酶链反应阳性证实了诊断。进行骨活检。静脉用美罗培南和免疫球蛋白治疗成功。我们的报告显示了免疫缺陷患者的异常表现，并讨论了最初抗生素治疗的失败。