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A 77-year-old man showed an asymptomatic meningeal lesion beneath the dura matter in the left fronto-parietal region on MRI during an examination for recurrent hoarsness. The lesion showed no gadolinium enhancement, and extended to neither the sulci nor skull. Neurological examinations revealed hoarseness, cochlear and vestibular dysfunction of the right ear, and mildly decreased Achilles tendon reflexes bilaterally. Laboratory findings showed marked inflammatory responses, but no abnormalities for LDH, IgG4, angiotensin-converting enzyme, or soluble IL-2 receptor. There was no serum monoclonal protein. Autoantibody panels in the serum were unremarkable except for an elevation of anti-type II collagen antibodies to a borderline value. Cerebrospinal fluid analysis disclosed an elevated protein concentration (152 mg/dl) and IgG index (1.41) with normal cell counts, negative results for bacterial/tubercular infection, and a normal cytology. 18 F-fluorodeoxyglucose positron emission tomography showed increased uptake in the left frontal region (Max SUV: 7.54). Swelling of the vocal cord, arytenoid cartilage, false vocal cord, and vocal cord palsy on the right side were seen on laryngoscopy, all of which were ameliorated by dexamethasone administration. A meningeal biopsy contained the dura matter and arachnoid, in which a granulation composed of massive mature plasma cells with many Russel bodies, accompanied by occasional lymphocytes and histiocytes were observed. Three months after the biopsy, he developed bilateral auricular chondritis and conjunctivitis. Based on these findings, we diagnosed him with relapsing polychondritis (RP). Prednisolone administration (40 mg/day) improved the chondritis and meningeal lesion. Central nervous system involvement is rare in patients with RP, and meningeal complications, such as aseptic meningitis or pachymeningitis, are mostly observed after the diagnosis of RP. However, due to its rarity, it still remains to be clarified whether a similar pathogenesis of meningeal complications underlies RP. The present case is indicative in that predominant meningeal granuloma arose during chondritis of several portions that had gradually developed, which suggests that meningeal complications could be derived from RP.

A cervical retro-odontoid pseudo-tumor, which is considered as a reactive fibrocartilaginous mass, is a rare condition in cervical myelopathy. A 63-year-old male, with repeated neck axial movements by a long-term leisure-time cycling, developed subacute myelopathy. Cervical MRI showed a mass lesion at the retro-odontoid region, compressing to the upper spinal cord. After detailed systemic and local examinations that ruled out primary or metastatic malignancy and inflammatory disorders such as rheumatoid arthritis or chronic kidney diseases, a retro-odontoid pseudo-tumor was diagnosed clinically. The patient underwent posterior C1-laminectomy without tumor resection and its pathological confirmation. After the surgery, his neurological signs of cervical myelopathy improved, and a follow-up MRI one year later showed a mild reduction of the tumor size. The neuro-physicians should recognize the relatively benign pseudotumor in cervical myelopathy, because the tumor size usually shows no further enlargement or regression only after decompression surgery without tumor resection.

A 65-year-old female presented to our hospital with a 6-month history of pollakuria, low-grade fever and urgent incontinence. Cystoscopy revealed a nonpapillary bladder tumor that was 50 mm in diameter in the trigon. Computed tomography showed the abscess between the urinary bladder and sigmoid colon. Transurethral resection was performed and the histology consisted of inflammatory lesions with inflammatory cell infiltration, which was diagnosed as an inflammatory pseudotumor due to diverticulitis of the sigmoid colon. Sigmoidectomy was subsequently performed. A fistula between the urinary bladder and the sigmoid colon was not detected. Cystoscopy 2 months after the operation revealed no signs of a bladder tumor.

A 36-year-old woman was referred with urinary incontinence and recurrent episodes of pyelonephritis. Two years prior to her visit, she underwent transurethral resection of a bladder tumor, 5 cm in diameter. Total cystectomy was suggested, as the initial diagnosis was sarcoma. Close re-evaluation of the pathological specimen lead to the final diagnosis of a benign inflammatory pseudotumor of the bladder as the tumor consisted of smooth muscle cells with white blood cell infiltration but without mitotic figures. The tumor disappeared during the follow up period. A cystography revealed bilateral vesicoureteral reflux with marked trabeculated bladder. A cystometry showed loss of bladder sensation and a low compliance bladder without detrusor contraction. Neurological examination and a magnetic resonance imaging of the spinal cord failed to prove the presence of definite neurological abnormalities. She was finally diagnosed with neuropathic bladder of unknown origin. In this case, she had been suffering from recurrent cystitis about 6 years before the resection of bladder tumor and it was suggested that the occurrence of the inflammatory pseudotumor of bladder would be related with chronic urinary tract infection due to neuropathic bladder. Urinary incontinence and urinary tract infection were controlled successfully with clean intermittent self-catheterization and adequate administration of antimicrobial drugs. Vesicoureteral reflux was treated with injection of GAX collagen into the ureteral orifices. No tumor recurrence has been found up to the present time, 5 years after the resection of bladder tumor.

A 61-year-old man was admitted to our hospital with the chief complaint of asymptomatic macrohematuria. Because he was diagnosed with arteriovenous malformation of the right kidney by angiography, embolization was performed. However, 2 years and 3 months later, bleeding from the right kidney was detected. Computed tomography (CT) revealed a low density area (1.8 cm) in the right kidney protruding to the renal pelvic adjacent to the old embolized lesion. Since we could not make a conclusive diagnosis as malignant disease by ureteroscopy and angiography, we have decided to follow the case carefully. Nine months later, macroscopic hematuria worsened, and the low density area by CT in the right kidney expanded. Therefore, we finally decided to perform right total nephroureterectomy. The tumor was histologically diagnosed as renal inflammatory pseudotumor. This is the 14th case reported in Japan. We here report the renal inflammatory pseudotumor with a review of the Japanese literature.

A 71-year-old man had noticed a dry sensation in the mouth with swelling of bilateral parotid glands in 1988. He was given a diagnosis of Sjögren's syndrome (SS) on the basis of characteristic findings of sialography and a minor salivary gland biopsy. He was admitted to our department in Febuary 1995 because of general fatigue of 2 month's duration. Laboratory data showed both positive anti-SSA and anti-SSB antibodies, liver dysfunction, hypoalbuminemia, and thrombocytopenia. Abdominal CT and MRI demonstrated a 2-cm intrahepatic mass (S 6) with enhanced areas at the periphery. The liver biopsy yielded fragments from the intrahepatic mass and hepatic parenchyma. The former was composed of plasma cells, lymphocytes, and histiocytes, compatible with the diagnosis of inflammatory pseudotumor of the liver. The pathological diagnosis of the latter specimen was primary biliary cirrhosis, although antimitochondrial antibody was negative. The intrahepatic mass gradually decreased in size without treatment. Inflammatory pseudotumor is considered to be a benign inflammatory condition simulating a neoplasma and the possibility of an autoimmune reaction is suggested on the basis of etiology. This is the first report of an inflammatory pseudotumor associated with Sjögren's syndrome developing in the liver. The inflammatory pseudotumor should be considered as a possible diagnosis in cases where the tumor is embedded in the liver.

A 60-year-old man with renal cell carcinoma was treated of right nephrectomy and high dose administration of Interferon-alpha (IFN) in 1990. Three and half years after operation, he complained of cough and hemoptysis. Chest X-ray showed a abnormal shadow (5.5 x 3.5 cm) in the upper lung field. On chest CT, a tumor mass with small cavity was located at S2, attended with the speculation and pleural indentation. A rough nodule suspected daughter tumor was pointed out near the tumor mass. Laboratory investigation revealed mild leucocytosis, an elevated ESR and C reactive protein. On lymphocyte subset, CD 4/8 ratio was 0.8 and NK cell activity was 11%. Immunosuppressive acid protein (IAP) in serum was 1,137 ng/ml. No organisms or malignant cells could be demonstrated in the biopsy materials or sputum. Right thoracotomy and upper lobectomy was carried out. Postoperative pathological diagnosis was inflammatory pseudotumor. Namely, the lesion was occupied with variable admixture of lymphocytes, plasma cells, histiocytes, phagocytes included hemosiderin, foam cells and whorled fibrosis. Daughter tumor was scar tissue. Now, he is well without recurrence and his immune response is within normal level. Some discussion of the literature was mentioned.

The tracheal tumor is uncommon, and tracheal inflammatory pseudotumor seems to be rare with only four cases reported in the Japanese literature. We here in report a case of tracheal inflammatory pseudotumor in a 61-year-old Japanese man presenting with dyspnea and wheezing 2 weeks after a sub total gastrectomy Billroth I for gastric cancer in april 1991. At bronchofiberscopic examination a rounded tracheal mass 40 mm distal to the vocal cord was found. High vascular on the surface and obstruction of about 90% of the tracheal lumen was noticed. Surgery was attempted immediately and 25 mm of the trachea was excised. The histopathological findings revealed an inflammatory pseudotumor. We concluded that the tumor was caused by injury during tracheal intubation.