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An Unusual Presentation of Neuropathic Arthropathy

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An Unusual Presentation of Neuropathic Arthropathy Annals ofthe Rheumatic Diseases 1991; 50: 717-721 717 Ann Rheum Dis: first published as 10.1136/ard.50.10.717 on 1 October 1991. Downloaded from Pseudoseptic arthritis: an unusual presentation of neuropathic arthropathy Worawit Louthrenoo, Barbara E Ostrov, Young S Park, Susan Rothfuss, H Ralph Schumacher Jr Abstract left shoulder was moderately swollen, warm, A 49 year old black man with scoliosis with a large effusion and was painful with presented with bilateral shoulder swelling motion. Intrinsic muscles of his right hand were eventually shown to be due to neuropathic atrophied. Muscle strength of both arms arthropathy related to underlying syringo- seemed decreased but was difficult to assess myelia. The synovial fluid was highly inflam- owing to the mechanical limitation of the matory, but cultures from synovial fluid and shoulders. Biceps and triceps reflexes were synovial tissue were all sterile. Profuse fat absent bilaterally. Pinprick, temperature, and droplets were noted and considered as a proprioceptive sensations were decreased in possible cause of the inflammation. This is an both arms, but loss' was more severe on the unusual presentation of neuropathic arthro- right. Movement of his left hip was limited pathy in a patient who was also harbouring an without pain. Results of the remainder of the adenocarcinoma that was undetected until eamination were unremarkable. later. Initial laboratory tests showed a haemoglobin of 66 g/l, packed cell volume 0-2, white blood cell count 8 9x I09/l with a normal differential Neuropathic arthropathy is a destructive count, platelets 1127xlO9/l, and reticulocyte process which is thought to be caused by count 2-4%. Erythrocyte sedimentation rate was impaired articular proprioceptive and pain 150 mm/h. Peripheral blood smear showed sensation leading to overuse and repetitive hypochromic and microcytic red cells without injury and by altered blood flow to the joints.' other significant abnormalities. Urine analysis Synovial fluid is usually 'non-inflammatory' or results were normal. Stool occult blood was haemorrhagic.1 We present a patient with negative. syringomyelia who had neuropathic arthropathy Chest radiograph showed marked right of the shoulder and had an unusual highly thoracic scoliosis. The heart and lungs were inflammatory synovial fluid which mimicked normal. Cervical spine radiographs showed http://ard.bmj.com/ septic arthritis. cervical spondylosis of C3-4 with narrowing of the intervertebral disc spaces. Neuroforamina were normal. Radiographs of his right shoulder Case report showed a large soft tissue density around the A 49 year old black man presented with bilateral shoulder. The joint space was widened. There shoulder swelling. Seven months before admis- were destructive changes with resorption of the humeral head and the sion he developed intermittent tingling and portions of glenoid on September 27, 2021 by guest. Protected copyright. numbness in his right hand. Six months later he (fig 1). Soft tissue calcification, chondro- Department ofMedicine, noted painless swelling of his right shoulder, University of with weakness and increased numbness in his Pennsylvania School of Medicine right arm. Soon after this his left shoulder W Louthrenoo began to swell, with pain on motion. He B E Ostrov denied any history of fever, weight loss, joint H R Schumacher stiffness,. back pain, diarrhoea, homosexual Department of activity, intravenous drug abuse, exposure to Pathology, Medical College of Pennsylvania tuberculosis or syphilis, or trauma to his neck or Y S Park shoulders. Medical history included hyper- Ardritis-Immunolog tension, and scoliosis diagnosed when he was in Center and the Pathology the army. He had a left total hip arthroplasty in Section, Veterans 1982. The cause of his left hip disease was not Administration Medical Center, Phidelphia, ascertained. He had gonococcal urethritis on Pennsylvania, USA several occasions but had been last treated six W Louthrenoo months before admission. He admitted past Y S Park S Rothfuss heavy tobacco and alcohol consumption. He H R Schumacher also had a history of exposure to asbestos. Correspondence to: Examination showed a pale, alert man with Dr H R Schumacher Jr, normal vital signs. His back showed marked Arthritis-Immunology Center, Veterans right thoracic scoliosis with the apex at the T9 Administration Medical vertebral body and a secondary left lumbar Center, Philadelphia, Figure I Radiograph ofthe right shouldr showing a large PA 19104, USA. scoliosis. His right shoulder was markedly soft tissue density around the shouderjoint. Thejoint space is Accepted for publication swollen with a massive warm effusion. Range of widened and there is lysis ofthe humeral head and the glenoid 14 August 1990 motion was decreased but was without pain. His surface. No soft tissue calcif,catwns or osteophytes are seen. 718 Louthrenoo, Ostrov, Park, Rothfuss, Schumacherjr calcinosis, or osteophytes were not identified. cells. There were no monosodium urate or Radiographs of his left shoulder showed calcium pyrophosphate dihydrate crystals. Ann Rheum Dis: first published as 10.1136/ard.50.10.717 on 1 October 1991. Downloaded from minimal soft tissue swelling and widening of the Alizarin red S staining for calcium crystals was glenohumeral joint space. Magnetic resonance also negative. Numerous intracellular and extra- imaging of his cervical spine (fig 2) showed a cellular fat droplets were identified by Sudan long thin slit in the middle of the upper cervical stains. Electron microscopy of the synovial fluid spinal cord, which is characteristic of collapsed showed degranulated neutrophils containing syrngomyelia. homogeneous grey globular bodies and other Repeated arthrocenteses of his right shoulder unusual angulated osmiophilic, presumably obtained 100-500 ml of cloudy serosanguinous lipid related, electron dense structures. Closed synovial fluids. The white blood cell counts needle synovial tissue biopsy was performed in ranged between 37-3 and 61-1x109/1 with his right shoulder (fig 3A). The synovium was 93-98% neutrophils. Arthrocentesis of his left oedematous, had areas with congested vessels, shoulder produced 250 ml of serosanguinous and was infiltrated with neutrophils, lympho- fluid. The white blood cell count was 41 -6x 109/1 cytes, plasma cells, and some necrotic cells, with 97% neutrophils. All fluids contained red which were scattered throughout the tissue. Extravasation of red blood cells was also noted. Tissues stained for micro-organisms were nega- tive. Cultures for bacteria, fungi, and myco- bacteria from synovial fluid and synovial tissue were sterile. As there was still concern about the possibility of infection an open arthrotomy and synovial biopsy of his right shoulder was performed two weeks later. At surgery the humeral head was found to be grossly deformed with a thick fibrin exudate covering the bone. The cartilage was completely destroyed. There was no synovial fluid obtainable at this time. Microscopically, the synovial surface was covered with fibrin. There was proliferation of the synovial lining cells. The subsynovial cell layer showed pro- liferation of small blood vessels, prominent fibrous tissue, and some chronic inflammatory cell infiltration. There was no evidence of Figure 2 Magnetic resonance imaging ofthe cervical spine shows a thtn transverse slit (arrow) in the middle ofthe granulomas or tumour. Fragments of bone and atrophic cervical cord. This is characteristic ofa collapsed cartilage were seen embedded in the synovium syrzngomyelia. (fig 3B). Smears stained for micro-organisms http://ard.bmj.com/ on September 27, 2021 by guest. Protected copyright. Figure 3 Synovial tissue ofthe right shoulder. (A) The ynovium obtained by needle biopsy after one month ofswelling in this area is heavily infiltrated with acute inflammatory cells (haematoxylin and eosin). (B) Two weeks later most areas ofthe synoviun show vessels with thickened walls. Fragments ofbone (arrows) are seen in the synovium (haematoxylin and eosin, usinggreenfilter). Pseudoseptic arthritis 719 were negative as were stains for amyloid. received physical treatment. He was never given Synoyial tissue cultures for bacteria, myco- antibiotics. Both shoulders showed some func- Ann Rheum Dis: first published as 10.1136/ard.50.10.717 on 1 October 1991. Downloaded from bacteria, and fungi were all sterile. Electron tional improvement and were less swollen. After microscopy of the synovial lining cells showed discharge from hospital he was lost to follow up many lipid droplets and osmiophilic mem- until he was admitted again one year later with branous arrays suggestive of phospholipids (fig chronic cough and progressive shortness of 4). No crystals or any suggestions of viral or breath. Examination showed diminished breath bacteria-like particles were seen under electron sounds in his right lung and palpable right microscopy. supraclavicular and axillary lymph nodes. His Blood electrolyte and liver enzyme levels right shoulder showed minimal swelling with- were normal. Serum albumin was 24 g/l (normal out effusion. There were no changes in neuro- 35-55) and globulin 66 g/l (normal 25-29). logical findings. A chest radiograph showed Serum protein and immune electrophoresis diffuse alveolar infiltrates in the lower two showed IgG and IgA polyclonal hypergamma- thirds ofthe right lung, thickening ofthe pleura, globulinaemia. Results of iron studies were and a minimal right pleural effusion. He was consistent with anaemia of chronic
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