BACKGROUND Systemic lupus erythematosus (SLE) is an autoimmune disease with diverse manifestations. The involvement of the musculoskeletal system is very common, and infection is one of the manifestations, which can involve any part of the body. We report a case of a middle-aged woman with recurrent episodes of infection of her left temple. CASE REPORT A 51-year old woman was referred to our clinic following failures to eradicate infection on her left temple for 9 months. Examination revealed facial asymmetry, with diffuse non-tender swelling involving her left temple area, which extended to her cheek. Computed tomography and magnetic resonance imaging (MRI) showed a periosteal reaction of the zygomatic bone. Left temporalis muscle thickening and residual osteomyelitis of the zygomatic bone were also shown by MRI. In view of the unresolved infection with incision and drainage and antibiotics, further blood investigations led to the discovery of SLE. The antinuclear antibody and anti-double-stranded DNA were positive. In addition, low nephelometry markers, C3 (26.7 mg/dL) and C4 (8.24 mg/dL), were observed. This patient was treated with 200 mg of oral hydrochloroquine once daily and 5 mg of oral prednisolone once daily. After 6 months of treatment, the infection subsided, and the structures involved showed remarkable healing. The patient is still taking the same dose and frequency of both drugs at the present time. CONCLUSIONS Temporalis pyomyositis and osteomyelitis of the zygomatic bone could be manifestation of SLE disease; however, the involvement of infection cannot be ruled out.