P3.219: Uncommon Being Uncommon: A Case of Facial Aseptic Abscess Syndrome as the Presenting Symptom of Ulcerative Colitis

Introduction: Inflammatory bowel disease (IBD) is of special relevance to Med-Peds physicians because of its bimodal age distribution and the importance of transitioning pediatric-onset chronic illness. Approximately 25% of patients have extraintestinal manifestations (EIMs) precede their IBD diagnosis, highlighting the need for a high index of suspicion.

Case Description: A previously healthy 17-year-old male presented with 14 days of fever (101-103 F) and progressive warm, erythematous, fluctuant and painful bilateral facial swellings in the setting of 1 month of persistent rhinorrhea, malaise, scant non-bloody non-bilious emesis, and 10 lb weight loss. Symptoms were refractory to 8 days of oral levofloxacin taken as prescribed by his PCP.
Vital signs were notable for tachycardia (100s-120s) and fever to 102.0 F. Labs notable for anemia to 7.0 and CRP 12.3 mg/L (nl < 0.5) and albumin 2.6 g/dL. Contrasted CT of the face showed several small multiloculated fluid collections along subcutaneous soft tissues, the largest a 1.3x1.1x1.8 cm rim-enhancing collection along the left zygoma. Additional history obtained after laboratory results revealed one month of daily bloody diarrhea, abdominal discomfort, and tenesmus. GI pathogen PCR was negative. He was admitted for transfusion, IV antibiotics, and further workup.
Through hospital days 1-3, he was treated with IV ampicillin-sulbactam; fever and abscesses persisted. Left zygomatic and right mandibular abscesses spontaneously ruptured showing moderate polymorphonuclear cells on Gram stain but no bacterial growth on aerobic and anaerobic cultures. Staphylococcus aureus PCR was negative.
On hospital day 4, he had a colonoscopy with biopsies consistent with ulcerative colitis (UC). Inpatient treatment with rectal hydrocortisone and mesalamine was initiated that day followed by IV methylprednisolone on day 5. His CRP, tachycardia, fever, and facial abscesses resolved but had minimal colitic response. See Table 1 for CRP and albumin trends. His therapy was escalated to 3 doses of 10mg/kg infliximab given concern for high clearance with improvement in hematochezia and normalization of CRP. At time of writing, he remains on infliximab monotherapy with minimal symptoms.

Discussion: This patient’s sterile facial abscesses and improvement after steroids makes aseptic abscess syndrome (AAS) the most likely diagnosis of his initial presentation.
AAS is a rare EIM most often seen in young males with UC and occurs in the context of disease flares. It commonly affects the spleen and liver but can involve skin. Treating the underlying flare resolves AAS with moderate-high efficacy.

Conclusion: Higher suspicion for EIMs as a sentinel symptom of IBD is a unique and important perspective of Med-Peds physicians, as they will be faced with atypical presentations of diseases across the lifespan, including the vulnerable time of transition to adult health care. Adolescents specifically require sensitive history taking strategies, as this case exemplifies, as his anemia prompted discussion of his hematochezia and ultimate diagnosis.