An Inflammatory Pseudotumor in the Costo-Vertebral Gutter: Case Report with Literature Review

Abstract

Inflammatory pseudotumor (IPT) is a benign tumor-like lesion of unknown cause and is originally described in the lung and orbit, but it has recently been reported throughout the body and only a small number of people [1,3,6]. We presented a rare case of costo-vertebral inflammatory pseudotumor with epidural invasion presenting with progressive cervico-brachial pain. There were no signs of myelopathy. Case PresentationA 50-year-old male had a two month history of left cervicobrachial pain, with progressive weakness in left upper limb. The patient signed an informed written consent form before participation. The neurologic examination showed brachial monoparesis predominantly in distal left without pyramidal signs. Magnetic resonance imaging (MRI) showed hypointense lesion in both T1-and T2-weighed images (Figures 1,2) with intense contrast enhancement at the left costo-vertebral gutter with epidural extension through the lateral foramen, causing a compression on the spinal cord from C7 to D3 (Figure 3). The thoraco-abdominal CT scan did not show other lesions. A CT scan-guided biopsy through posterior-lateral approach was performed. The histological examination revealed that the lesion was made of dense collagen with fibrous background, associating fibroblastic or myofibroblastic cells having a clarified ovoid nuclei and inflammatory cells grouped into small clusters formed mainly of plasma cells and lymphocytes, and the lesion was richly vascularized. No necrosis was observed. The antiactin smooth muscle antibody marked diffuse myofibroblasts that are H-caldesmone negative, the ALK was positive and the anti-cytokeratin AE1/AE3 antibody was negative excluding the possibility of an epithelial tumor. Hence, the diagnosis of an inflammatory pseudotumor was made.Treatment started by steroids (1mg/kg/d) with adjuvant radiotherapy. There was a rapid recovery in his neurological functions, and there was a complete resolution of the lesion on control MRI after three years. There was no evidence of pulmonary or other lesions.