Background: Facial angioedema is a challenging for the medical profession, particularly in patients received cosmetic dermal fillers. Case presentation: A 43 years old fmale presented with one-year history of recurrent facial swelling after receiving unlabeled dermal fillers given by unlicensed esthetician. Her symptom improved with systemic steroid prescribed at private clinic. Dermatologic examination revealed nodular lesions on both cheeks measured 1-3 cm in diameter. Foreign body reaction to injectable filler was highlighted by plastic surgeon and surgical biopsy was advised. Upon allergy review, she reported associated symptoms of mild throat discomfort and occasional difficulty of swallowing. A diagnosis of idiopathic angioedema was raised. However, patient strongly believed that her facial swellings were related to her dermal fillers and reluctant to start antihistamines. Immunologic work up for idiopathic and congenital angioedema was unremarkable. She was labeled allergic to Chlorpheniramine in the past. This however, was excluded by supervised oral graded challenge performed in the allergy clinic. The patient was shortly admitted for of an episode of angioedema, upper gastrointestinal (GI) endoscopy was performed for other complaints of heart burn and mild difficulty of swallowing. Not surprisingly, laryngeal edema was pictured during the procedure, rapid urease test for (H. pylori) was positive. These finding have further supported the diagnosis of idiopathic angioedema. Patient has completed (H. Pylori) eradication therapy. Afterward, she was commenced on a combination H1 and H2 antihistamines, this has resulted in significant improvement of her facial angioedema and throat discomfort. Conclusion: Facial angioedema in patients received injectable dermal fillers may not necessarily be due to local adverse reaction, in certain clinical setting a diagnosis of idiopathic angioedema should be suggested. Therefore, consulting allergy specialist is essential in evaluating such patients.