A 67-year-old woman, who had a history of an open surgery for mandible and right knee joint fractures in a traffic accident 28 years ago, visited our hospital due to the swelling of lower jaw. Under the diagnosis of titanium mesh and plate infection, the mesh and the plate removal with debridement was performed. After operation, she realized mild erythemas on her trunk and limbs skin. On the fourth day after operation, she had a fever, increased neutrophils and the elevation of C-reactive protein (CRP). Initially, post-operative infection was suspected and an antibiotic was administered, while it was ineffective. Accordingly, the patient was referred to the Department of Internal Medicine, and subsequently to dermatology specialist for the further diagnosis of skin erythema and blood test abnormalities. Since the dermatology specialist suggested a possible clinical diagnosis as Sweet’s syndrome, skin and mucosal biopsy was performed, which confirmed the diagnosis. As far as we could search, this is the first report of Sweet’s syndrome followed by the surgery in oral and maxillofacial region. Since this case developed after surgery, it was difficult to distinguish it from postoperative infection or drug allergy.
“A case of Sweet’s syndrome secondary to removal of infected mandibular titanium mesh and plate” by Hiroko Takada, Katsumitsu Shimada, Michiko Yoshizawa and Hideaki Kagami.
Sweet’s syndrome related to oral and maxillofacial region is extremely rare and this case would be a reference for the diagnosis of similar cases.
Sweet’s syndrome (acute febrile neutrophilic dermatosis) was first reported by Sweet in 1964 and is characterized by 4 cardinal features: 1) fever, 2) a relative increase of neutrophils in the peripheral blood, 3) skin lesions such as tender erythematous plaques, nodules, vesicles, and pustules on the face and extremities, and 4) a dense dermal infiltrate with mature neutrophils seen histologically [ , ]. Since fever and neutrophilia are not consistently present, the diagnosis is based on the two constant features, i.e. typical eruption and characteristic histologic features [ ].
Although the pathogenesis of Sweet’s syndrome is not well understood, dysregulation of immune function or an immune-mediated hypersensitivity to an eliciting bacterial, viral, or tumor antigen are considered as possible triggers [ ]. Although the occurrence of Sweet’s syndrome is not very uncommon, the cases relating to oral maxillofacial diseases are extremely rare and, as far as we could search, there is no previously reported case associating to the titanium plate or mesh infection or their removal in maxillofacial region [ ]. (166 words).
Report of case
A 67 years-old woman visited Matsumoto Dental University Hospital with a chief complaint of the swelling of lower jaw. She had a history of the fracture of mandible and the right knee joint due to a traffic accident 28 years ago and had an open surgery to reconstruct the mandible with a titanium mesh, plate and wires at an emergency hospital. At the first visit to our hospital, her body temperature was 36.8 °C, and her lower jaw showed swelling with redness and a percutaneous fistula was observed, while bone resorption was not evident from panoramic radiograph ( Fig. 1 ). A diagnosis of titanium mesh and plate infection was made and an operation of mesh and plate removal was planned. An incision was made at the skin under chin including the fistula. After elevation of skin-periosteal flap, the titanium mesh and plate were removed. The wires were also removed except for the parts varied inside the mandible. Subsequently, the granulation tissue around the titanium mesh was removed and the incision was closed.
From the evening on the day of surgery, mild erythema was noted at her right hand and the flexion side of right elbow. However, the erythema showed improvement next day, so that no specific treatment was applied. On the third day after the surgery, she complained joint pain on her back and waist, so that a poultice was prescribed. On the fourth day after the surgery, she developed fever (39 °C) and sore throat. Hematologic examination showed a slightly high white blood cell (WBC) count (8500/mm 3 ) with 87.6% neutrophils, and elevated CRP (6.54 mg/dl). Although the inflammation of the operated region was not evident, postoperative infection could not be excluded and the antibiotics were changed from CEPN-PI (300mg/day) to IPM (1g/day, div). However, after two days, the medication was still ineffective and the fever and sore throat were continued. At this time point, significant erythema recurred accompanying with oppressive pain on her upper and lower limbs on both sides, right side of back and right upper eyelid ( Fig. 2 a and b). No culture was performed before surgery. On the fifth day after surgery, the bacteriological examination was carried out for the exudate from the chin, but neither aerobic/anaerobic bacteria nor fungi was detected.