S41 Unusual Case of Tongue Deviation

Abstract

Case: Background: Anti-tumor necrosis factor-α (TNFα) therapy is the mainstay of treatment for inflammatory bowel disease such as Crohn’s Disease (CD) and ulcerative colitis (UC). Despite its widespread use, anti-TNFα leads to significantly altered inflammatory response and increased risk of severe skin and soft tissue infections. Here, we highlight an uncommon presentation of severe soft tissue infection in a patient with ileal CD maintained on anti-TNFα therapy. Case Presentation: A 50-year-old female with a 25-year history of Crohn's ileitis maintained on certolizumab for 3 years presented to the emergency department (ED) with an expanding left, lateral neck mass resulting in decreased cervical range of motion. This was accompanied by a left-sided tongue deviation. She first presented to our ED 8 weeks earlier with severe throbbing headaches without focal neurologic symptoms. A head MRI only showed non-specific T2/FLAIR hyperintensities. Her vital signs and labs were normal, and these headaches were presumed to be complex migraines. Three weeks after her first presentation, she came back to the ED with worsening headaches, neck pain and a new left-sided tongue deviation. Again, her vital signs and laboratory evaluation were normal apart from an ESR of 68 mm/hr (nl: 0-30 mm/hr) and CRP of 10.34 mg/dL (nl<0.5 mg/dL). Her repeat imaging studies did not show any evidence of stroke; however, opacification of the paranasal sinuses and left mastoid air cells were observed. Nonetheless, given her lack of systemic inflammatory symptoms, she was discharged home with close outpatient otolaryngology (ENT) referral. As an outpatient, she was diagnosed with bacterial sinusitis which was treated with a 15-day course of cefdinir and dexamethasone. Her symptoms initially improved; however, she eventually experienced dysphagia with pus drainage from her sinuses. Another antibiotic and steroid course was prescribed. Despite this, approximately 8 weeks after her initial presentation, she developed a rapidly expanding neck mass which prompted her return to the ED. Physical examination revealed a tender neck mass limiting her cervical range of motion and a more pronounced tongue deviation. She was afebrile, non-tachycardiac, and normotensive. Laboratory testing again revealed elevated inflammatory markers (ESR 83 mm/hr, CRP 8.59 mg/dL) but otherwise her WBC was normal (8310 cells/ µL, nl: 10400 cells/µL) with no evidence of sepsis-related end-organ dysfunction (Cr 0.89 mg/dL, AST 20, ALT 55 mg/dL, T-bili 0.3 mg/dL, platelets 348000 platelets/µL). Neck imaging showed multifocal ring-enhancing collections within the left paravertebral soft tissue with left hypoglossal nerve involvement. Intravenous vancomycin, cefepime, and metronidazole were started. Surgical debridement of the left posterior paraspinal abscess was performed. Intraoperative cultures were positive for Streptococcus anginosus. She completed a 6-week course of ceftriaxone but follow up imaging 2 weeks later showed recurrence of her neck abscesses. This again required surgical debridement. Her infections finally cleared with 24 weeks of piperacillin/tazobactam. Her certolizumab remains permanently discontinued. Conclusions: Hallmark symptoms of severe infection may not be present in patients on anti-TNF therapy. A high index of suspicion for an underlying infectious etiology must be maintained when treating this population.