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Copyright The Korean Academy Inflammatory Myofibroblastic Tumor on Intercostal Nerve Presentingas Paraneoplastic Pemphigus with Fatal Pulmonary Involvement Inflammatory myofibroblastic tumors (IMTs) are benign neoplasms that can occur at different anatomic sites with nonspecific clinical symptoms. A 48-yr-old woman presented with a 2-month history of a relapsed oral ulcer, progressive dyspnea, and Department of Neurosurgery, School of Medicine, a thoracic pain induced by breathing. A tumorous mass was noticed in the right Kyungpook National University, Daegu, Korea costodiaphragmatic recess on chest computed tomography and magnetic reso-nance imaging, and the patient underwent a right costotransversectomy with exci-sion of the tumor, which originated from the 12th intercostal nerve. Histology andimmunohistochemistry showed that the tumor was an IMT of the intercostal nerve.
The patient’s postoperative course was not favorable; dyspnea persisted after sur- Received : 6 March 2006Accepted : 16 June 2006 gery, and a progressive pulmonary compromise developed. The cause of the res-piratory failure was found to be bronchiolitis obliterans, which in this case provedto be a fatal complication of paraneoplastic pemphigus associated with an IMT.
This case of IMT of the spinal nerve in the paravertebral region is unique in terms of its location and presentation in combination with paraneoplastic pemphigus, which is rare. A brief review of the heterogeneous theories concerning the patho- Department of Neurosurgery, School of Medicine, genesis, clinicopathological features, and differential diagnosis of this disease enti- Kyungpook National University, 50 Samduk-2-ga, Jung-gu, Daegu 700-721, KoreaTel : +82.53-420-5649, Fax : +82.53-423-0504 Key Words : Inflammatory Myofibroblastic Tumor; Intercostal Nerves; Paraneoplastic Pemphigus INTRODUCTION
CASE REPORT
Inflammatory myofibroblastic tumors (IMTs) are benign A 48-yr-old woman presented with a 1-month history of tumor-like lesions of unknown etiology that can occur at dif- progressive dyspnea and pleuritic pain in the right lower ferent anatomic sites and have variable non-specific clinical chest, and also a thoracic back pain of 1-month duration.
symptoms. Moreover, this variability has led to a heteroge- There was no history of trauma. She had a history of a chro- neous nomenclature. Plasma-cell granuloma, inflammatory nic relapsing oral ulcer, and had been admitted to the Depart- pseudotumor, fibrous xanthoma, histiocytoma, and mast-cell ment of Internal Medicine at our institution 3 months earli- granuloma are among the terms most frequently used as er due to a fever and multiple oral ulcers (Fig. 1A), the lat- pathomorphologic descriptions (1). The common sites of ter of which had been diagnosed as pemphigus vulgaris by this lesion are the lung (the originally described location), biopsy. However, unfortunately, several examinations failed respiratory tract, gastrointestinal tract, orbit, liver, spleen, to detect the exact origin of the fever. After discharge, she lymph nodes, heart, and brain (2, 3). Seven cases of intras- had been treated with prednisolone and azathiopurine for 3 pinal IMT have been reported (4, 5), and only five cases of IMT involving peripheral nerves have been previously des- When she was readmitted, physical examination revealed cribed (6, 7). Although imaging techniques have made evi- an oral ulcer, multiple blisters and erythematous papules on dent progress, they cannot be used for a preoperative diag- trunk skin (Fig. 1B). All laboratory tests, including WBC nosis of IMT, and only histological examinations are capable count, RBC count, erythrocyte sedimentation rate, C-reac- of identifying its inflammatory character.
tive protein, and hemoglobin level were normal, as well as This paper describes an IMT of the intercostal nerve, a pre- plain chest and spine radiographs. However, computed tomo- viously unreported location, in combination with paraneo- graphy (CT) with intravenous contrast visualized a clearly plastic pemphigus (PNP), in a 48-yr-old woman, and includes demarcated paraspinal mass with homogeneous enhancement a discussion of its clinicopathological features and diagnosis. in the right costodiaphragmatic recess (Fig. 2A). Magneticresonance imaging showed an extradural and paraspinal ellip- soidal mass at the T12 level extending into the spinal fora- cumscribed tumor of 5×3×4 cm3 originating from the men. Large vessels were present at the center of the mass proximal part of the 12th intercostal nerve, but not infiltrat- (Fig. 2B). In view of her progressive symptoms, she was trans- ing the nerve grossly, was found to be bulging into the right ferred to the Neurosurgery Department for further evalua- thoracic cavity. The mass was removed en-bloc with a portion tion and treatment. Under the differential diagnosis for the of the parietal pleura. No involvement of any adjacent organ mass including schwannoma, osteogenic sarcoma, chondro- (lung, diaphragm, or vertebra) was found after complete exci- sarcoma, or metastasis, we approached via costotransversec- sion. Biopsy specimens showed proliferation of a heteroge- tomy and resected it completely. Intraoperatively, a well cir- neous population of inflammatory cells, including macro- Fig. 1. (A) Erosions of the vermilion border, tongue, and buccal mucosa. (B) Multiple erythematous papules on trunk skin.
Fig. 2. (A) Contrast-enhance abdominal CT revealing a solitary paraspinal soft tissue mass with homogeneous attenuation in the posterioraspect of the right hemithorax. The mass displaced the right lower lobe superiorly. (B) Enhanced T1-weighted magnetic resonance axialsection through the disc at T12-L1 showing the mass and its large vessels. Vertebra and ribs were not eroded, and adjacent organs (liver,diaphragm, and thecal sac) were not involved by the mass. IMT Presenting as Paraneoplastic Pemphigus phages, lymphocytes, and neutrophils with peripheral fibro- nal Medicine, and a repeat biopsy of an oral ulcer and direct sis (Fig. 3A). Immunohistochemically, all were negative for tissue immunofluorescence showed intracellular IgG and S100, leu7, and SMA, but positivity for CD3 and CD20 complement C3 deposition within the epidermis (Fig. 3B).
indicated the presence of both mature B and T lymphocytes.
Finally, she was diagnosed as having PNP combined with The final histological diagnosis was IMT. bronchiolitis obliterans. She was continued on respiratory Pleuritic pain improved progressively after surgery, but therapy and with prednisone, cyclosporine, and intravenous dyspnea persisted and a progressive pulmonary compromise immunoglobulins. During the following months progressive developed. She was retransferred to the Department of Inter- dyspnea slightly improved after intravenous immunoglobu- Fig. 3. Histology of tumor samples resected during operation. (A) Photomicrograph showing an admixture of spindle-shaped ovoid cellsand a conspicuous admixture of lymphocytes and plasma cells (H&E, ×400). (B) Direct immunofluorescence showed IgG deposits inthe basement membrane zone and epithelial layer in this biopsy specimen of ulcerated oral mucosa. Table 1. Inflammatory myofibroblastic tumor of peripheral nerves: a review of the literature Mass involving geniculate Resection of mass 2-yr progression of idiopathic Fusiform 16×2 cm Resection of epineurium- Neurological recovery & based lesion with sparing no evidence of recurrence Mass affecting left greater Resection with segment & hypesthesia in 1st 3 fingers mass infiltrating fascicles infiltrated fascicles Present study 48/F Intercostal 1-mon pleuritic pain in right Resection of epineurium- Pain decreased but expired portion of parietal pleura with paraneoplastic lins therapy, as did the skin and mucosal lesion. However, 3 tion with a complex of four proteins (desmoplakin I, BP Ag, months after the operation she was readmitted to the inten- envoplakin and desmoplakin II, and periplakin), whereas sive care unit for progressive dyspnea. Unfortunately methyl- minor signs include histologic evidence of acantholysis in prednisolone and cyclosporin was ineffective, and she died erythematous lichenoid papules, DIF findings of basement membrane and intercellular epidermal IgG and C3, and IIFstaining of the rat bladder epithelium. Three major signs,or two major and two minor signs are required to diagnose DISCUSSION
Several cases of PNP, including one case of IMT, which The clinicopathological features of IMTs described in the resulted in respiratory failure caused by airway obliteration, literature as originating from the peripheral nerve are sum- have been reported (19, 20). Moreover, after the onset of res- marized in Table 1. The present case, an IMT that originat- piratory failure, autoantibody reaction against plakins detect- ed from the intercostal nerve in the paravertebral region, is ed by serum immunoprecipitation at the onset of PNP, dis- unique in terms of its location. IMTs are typically composed appeared as determined by immunofluorescence of the bron- of variable amounts of stromal and cellular elements, and chial epithelium. Based on the above-mentioned findings, myofibroblasts, which are involved in tissue repair, are now it is postulated that autoantibodies against some of these recognized as the principal cell type (4, 8). The precise eti- antigens play a causative role in acute respiratory epithelial ology of IMT is unknown. In some cases, they are consid- inflammation (20). Moreover, when treating PNP the lethal ered to result from inflammation following minor trauma complication of bronchiolitis obliterans should be kept in or surgery or to be associated with another malignancy (9, mind. Furthermore, the prevention of autoantibody-medi- 10). Some authors believe that this tumor is a low-grade ated injury to the respiratory epithelium should be an impor- fibrosarcoma that contains inflammatory (lymphomatous) tant treatment goal. Early diagnosis and tumor removal before cells. Immunohistochemical studies of T- and B-cell subpop- respiratory involvement are essential because of the typically ulations may be helpful for differentiating IMT and lym- poor response to immunosuppressive therapy shown in PNP.
phoma. An immune-autoimmune mechanism has also beenimplicated for the etiology of IMT. Many of the features ofIMTs can be related to the production of inflammatory me- REFERENCES
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