Kofoed Mikkel Seremet, Fisker Niels, Christensen Anne Estmann, Kjeldsen Anette Drøhse
Department of Head and Neck Surgery, Odense Universitetshospital, Odense, Denmark.
Department of Paediatrics, The Hans Christian Andersen Children's Hospital, Odense, Denmark.
BMJ Case Rep. 2018 Jan 17;2018:bcr-2017-221449. doi: 10.1136/bcr-2017-221449.
We present two 11-year-old girls with chronic recurrent multifocal osteomyelitis, treated with adalimumab. Both developed severe intracranial complications to sinusitis. Patient 1 had been treated with adalimumab for 15 months when she developed acute sinusitis complicated by an orbital abscess, forehead swelling, a subdural empyema and osteomyelitis of the frontal bone. She was treated with a rhinosurgical and neurosurgical approach with intravenous antibiotics.Patient 2 had been in adalimumab treatment for 10 weeks. Adalimumab was discontinued 8 weeks prior to developing subdural empyema and subcortical abscesses in combination with sinusitis. She was treated with endoscopic sinus surgery and intravenous antibiotics. Both patients had developed psoriasis and episodes of infection during treatment. They were non-septic and had low fever on presentation. None of the patients suffered any long-term neurological sequelae. The immunosuppressive treatment with adalimumab is considered to be the cause of the sinogenic intracranial complications in our cases.
我们报告了两名11岁患有慢性复发性多灶性骨髓炎的女孩,她们接受了阿达木单抗治疗。两人都因鼻窦炎出现了严重的颅内并发症。患者1在接受阿达木单抗治疗15个月时,患上了急性鼻窦炎,并伴有眼眶脓肿、前额肿胀、硬膜下积脓和额骨骨髓炎。她接受了鼻外科和神经外科手术,并静脉注射抗生素治疗。患者2接受阿达木单抗治疗10周。在出现硬膜下积脓和皮质下脓肿合并鼻窦炎前8周停用了阿达木单抗。她接受了内窥镜鼻窦手术和静脉注射抗生素治疗。两名患者在治疗期间均出现了银屑病和感染发作。她们就诊时无败血症,体温低热。两名患者均未出现任何长期神经后遗症。在我们的病例中,阿达木单抗的免疫抑制治疗被认为是鼻窦源性颅内并发症的原因。