RHEUMATOID NODULES OF THE SPINE: CASE REPORT AND REVIEW OF THE LITERATURE

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709 BRIEF REPORT RHEUMATOID NODULES OF THE SPINE: CASE REPORT AND REVIEW OF THE LITERATURE MARK E. PEARSON, MARY KOSCO, WILLIAM HUFFER, WILLIAM WINTER, JAMES A. ENGELBRECHT, and JAMES C.

1 709 BRIEF REPORT RHEUMATOID NODULES OF THE SPINE: CASE REPORT AND REVIEW OF THE LITERATURE MARK E. PEARSON, MARY KOSCO, WILLIAM HUFFER, WILLIAM WINTER, JAMES A. ENGELBRECHT, and JAMES C. STEIGERWALD We present the case of a patient who had rheumatoid nodules of the vertebrae, which had resulted in bony destruction of the spine at 3 levels. Although there have been only 3 previous reports of such findings with confirmation by histologic analysis, we believe the condition is more common than has been thought. From a review of the literature, we found that similar clinical and radiographic features, as well as descriptions of rheumatoid granulation tissue invading the disc spaces, have been described in several subjects, Rheumatoid nodules are found in approximately 20% of patients with rheumatoid arthritis (RA). These nodules occur most commonly on the extensor surface of the forearm, on the Achilles tendon, and in the olecranon bursa (1). Less commonly, they have been found in the lungs, heart, spinal cord, meninges, and on the vocal cords. Standard rheumatology texts, however, make no mention of rheumatoid nodule involvement on body structures (2,3). Autopsy studies have demonstrated that rheumatoid nodules were the cause of vertebral body destruction in 3 subjects with RA (4-6). We report another case of vertebral destruc- From the Department of Medicine, Division of Rheumatology, the Department of Pathology, and the Department of Orthopedic Surgery, University of Colorado Health Sciences Center, Denver. Mark E. Pearson, MD: Fellow in Rheumatology; Mary Kosco, MD: Resident in Internal Medicine; William Huffer, MD: Associate Professor of Pathology; William Winter, MD: Associate Professor of Orthopedic Surgery; James A. Engelbrecht, MD; James C. Steigenvald, MD: Associate Professor of Medicine. Address reprint requests to James C. Steigenvald, MD, Division of Rheumatology, Box B115, University Hospital, 4200 East Ninth Avenue, Denver, CO Submitted for publication March 7, 1986; accepted in revised form October 22, tion at multiple levels of the spine. This case is unique in that, during spinal surgery, rheumatoid nodules were found to have caused the vertebral destruction. Case report. The patient, a 65-year-old American Indian woman, had a 17-year history of seropositive nodular RA and a 9-year history of insulindependent diabetes mellitus. The arthritis had been treated with prednisone for most of the 17 years, and D-penicillamine had recently been added to the regimen. Her arthritis was well controlled with these medications until November 1984, when she noted the onset of dull, nonradiating pain in her lower back. The pain progressed, and in April 1985, roentgenograms were made of the patient s spine; these showed destruction of the L3-L4 disc space. A bane scan was also performed, and it showed diffuse uptake in this same area. Needle aspiration of the disc space was performed. Cytologic results and acid-fast bacilli (AFB) staining were negative, as were bacterial and tuberculosis (TB) cultures. It was believed, however, that the radiographic results were most compatible with a diagnosis of TB of the spine, and she was started on a regimen of isoniazid and rifampin. The pain worsened, and by June 1985, the patient became unable to walk. Roentgenograms of her spine (July 1985) showed progressive destruction of the L3-L4 disc space, with right sacroiliac (SI) joint destruction. A computed tomography scan demonstrated marked destruction of the L4 vertebral body. Needle aspiration of the right SI joint gave negative results on cytologic study, AFB staining, and bacterial and TB cultures. At that time, the patient was referred to the University of Colorado Health Sciences Center for further evaluation. At the time of admission, she was taking the Arthritis and Rheumatism, Vol. 30, No. 6 (June 1987)

Roentgenograms of the lumbosacral spine revealed destruction of the L4 vertebral body, particularly of the inferior endplate, as well as the superior endplate of the L5 vertebral body (Figure 1).

2 710 BRIEF REPORTS serum glucose 239 mg/dl. Results of all other blood chemistries analyzed were within normal limits. The chest roentgenogram performed at admission demonstrated a diffuse increase in interstitial markings bilaterally. Roentgenograms of the lumbosacral spine revealed destruction of the L4 vertebral body, particularly of the inferior endplate, as well as the superior endplate of the L5 vertebral body (Figure 1). There was also widening and sclerosis of the right SI joint. Roentgenograms of the thoracic spine revealed destruction and fusion of the T3 and T4 vertebral bodies, with obscuration of the T3-T4 disc space. A similar process involving the T8-T9 vertebral bodies and disc space was also observed. Roentgenograms of the cervical spine demonstrated destruction and fusion of the C4 and C5 vertebral bodies, with obscuration of the C&C5 disc space (Figure 2). Figure 1. Lateral view radiograph of the lumbosacral spine, revealing destruction of the L4 and LS vertebral bodies. following medications: prednisone (5 mg/day), D- penicillamine (250 mglday), neutral protamine Hagedorn (NPH) insulin (20 unitdday, subcutaneously, in the morning), digoxin (0.25 mglday), amitriptyline HC1 (25 mg/day, at bedtime), theophylline (200 mg every 8 hours), isoniazid (300 mg/day), and rifampin (600 mg/dzty). Pertinent findings of the physical examination included multiple rheumatoid deformities, but no active synovitis. Rheumatoid nodules were present over her ears, knuckles, and the extensor surface of her forearms. There was marked tenderness to palpation over the L3 through L5 vertebral bodies and the right SI joint. No focal neurologic abnormalities were noted. Auscultation of the lungs revealed dry crackles at both bases. No other extraarticular manifestations of RA were observed. Laboratory investigations demonstrated the following results: positive rheumatoid factor at a titer of t : 5,120, peripheral blood leukocyte count 7,900 cells/r~im~ with a normal differential cell count, hemoglobin 14 gm/dl, platelet count 403,000/mm3, Westergren sedimentation rate 78 mm/hour* alkaline phosphatase 327 units/liter (normal ), and Figure 2. Lateral view radiograph of the cervical spine, revealing destruction of the C4 and C5 vertebral bodies.

BRIEF REPORTS 711 An open biopsy of the lumbar spine was performed. There was a minimum of inflammation, but marked destruction of the L4 and L5 vertebral bodies was noted.

3 BRIEF REPORTS 711 An open biopsy of the lumbar spine was performed. There was a minimum of inflammation, but marked destruction of the L4 and L5 vertebral bodies was noted. Tissue specimens were negative for AFB, and all cultures, including bacterial, fungal, and TB, were negative. Sections of the vertebral bodies were stained with hematoxylin and eosin, and multiple granulomas were found in all sections. On close inspection, these granulomas were found to contain an area of central necrosis surrounded by a middle layer of palisading epithelioid cells (Figure 3). An outer third layer, which was extremely dense in some areas, contained lymphocytes and plasma cells. The pathologic findings were most consistent with a diagnosis of rheumatoid nodules of the vertebral bodies, causing bony destruction. Fusion of the lumbar spine was performed during a later operation. A similar procedure for the cervical spine was planned, but because of cardiovascular instability during the lumbar spine fusion, it was not performed. A cervical collar was applied, and the patient was transferred to a rehabilitation hospital for further care. Discussion. Rheumatoid nodules of the vertebral bodies have been described in 3 previous reports (4-6). Baggenstoss et a1 (4), in 1952, were the first to report this finding. Their patient was a 56-year-old man with a 3-year history of nodular RA who developed severe, nonradiating pain of the lower back. Roentgenograms revealed destruction and anterior wedging of the T12 vertebral body. The patient died of cardiac causes. Autopsy revealed rheumatoid nodules involving the TI2 and L3 vertebral bodies, the myocardium, pericardium, synovial membrane, and subchondral bone. Lorber et a1 (5) described a 47-year-old man who presented with a I-year history of kyphosis and pain of the lower thoracic spine. Roentgenograms demonstrated anterior wedge compression of the T9 vertebral body and collapse of the vertebral plates of L1 through L3. The patient subsequently developed synovitis of his right knee, and there were biopsy- Figure 3. Histologic appearance of a section from the vertebral body, obtained at open lumbar biopsy. Rheumatoid nodules were found in all sections. The nodules were characterized by central necrosis, a surrounding middle layer of palisading epithelioid cells, and an outer layer of lymphocytes and plasma cells (hematoxylin and eosin stained, original magnification X 125).

4 712 BRIEF REPORTS proven rheumatoid nodules of both olecranon bursae. Months later, he died of nephritis, pneumonia, and sepsis. At autopsy, grey-white nodules were seen infiltrating the vertebral marrow. Histologically, the nodules consisted of granulation tissue and foci of eosinophilic fibrinoid changes in collagen fibers, surrounded by palisading histiocytes and fibroblasts. Because of the limited central necrosis, these were believed to represent atypical rheumatoid nodules. Glay and Rona (6) described a 79-year-old woman with a 6-year history of deforming RA and biopsy-proven rheumatoid nodules over her buttocks. The patient presented with a 3-month history of severe low back pain, and roentgenograms revealed collapse of the T12, L2, and L3 vertebral bodies. The patient had a debilitating course, and she died. At autopsy, classic rheumatoid nodules were found in the collapsed vertebral bodies. Two other patients with RA were described by Glay and Rona. Both patients had radiographic changes similar to those of the first patient, but histologic confirmation was not obtained. Two other published reports present cases suggestive of a similar process. Shichikawa et a1 (7) described a 55-year-old woman with a 9-year history of seropositive, nodular RA who developed low back pain. Roentgenograms revealed destruction of the L4 and L5 vertebral bodies. The patient died of cardiac causes, and at autopsy, a necrotic area surrounded by granulation tissue was seen in the vertebral bodies of L4 and L5. Although not classic for a rheumatoid nodule, the point of its similarity to a rheumatoid nodule was raised. Seaman and Wells (8) described radiographic findings in 2 patients with RA; both exhibited destruction of intervertebral disc spaces, as well as the neighboring vertebral endplates. These abnormalities were observed at the C6 and C7 level in 1 patient, and at the T8 and T9 level in the other. Followup roentgenograms demonstrated a healing stage, with fusion of the neighboring vertebral bodies and obscuration of the disc space. These findings are remarkably similar to those in our patient; however, the findings in those 2 patients were not confirmed histologically. Some researchers have shown that rheumatoid granulation tissue involves the intervertebral disc spaces. Ball (9,lO) and Bland (11) examined the cervical spines of autopsy subjects with RA and found granulation tissue, which arose from the synovial-lined neurocentral joints nearby, invading the disc spaces. The changes were present despite radiographic evidence of minimal or no abnormalities of these areas. Bywaters (12) demonstrated that granulation tissue had spread from the costovertebral and zygapophyseal joints and had invaded the thoracic and lumbar disc spaces, respectively, in a patient with RA. Shichikawa et a1 (7) demonstrated that granulation tissue had invaded the cervical and thoracic disc spaces of 2 patients with RA. A rheumatoid nodule was also observed in the peridiscal area of the cervical spine in 1 of the patients. In contrast to the thinking of other investigators, it was those authors impression that this inflammatory tissue arises from the surrounding ligaments and areolar tissue and then extends to the adjacent vertebral body and disc space, as occurs in ankylosing spondylitis. Others have refuted these findings, questioning whether all inflammatory tissue in a patient with RA should be deemed secondary to the RA (12). Martel (13) and Resnick (14) have argued that discovertebral lesions in RA are a result of trauma at the discovertebral junction. It is their opinion that the lesions might result from spinal instability caused by zygapophyseal synovitis and ligament laxity. Martel s evidence to support this concept includes the following: (a) Schmorl node-like erosion and disc space narrowing often precede vertebral endplate destruction; (b) Zygapophyseal joint destruction precedes, and occurs at the same level as, disc-cartilage destruction; (c) Histologic examination of an involved disc and the adjacent vertebrae in 1 patient did not reveal inflammation; and (d) This destruction is absent in children, in whom ankylosis of the cervical zygapophyseal joints is a frequent occurrence. In summary, we have described a patient with seropositive nodular RA who was found to have rheumatoid nodules of the vertebrae, which resulted in bony destruction at 3 levels of the spine. The diagnosis was confirmed by histologic methods. Similar clinical and radiographic findings, in addition to the invasion of disc spaces by rheumatoid granulation tissue, have been reported in other patients. Histologic confirmation of such findings has been reported in only 3 of those patients. We believe that vertebral involvement in RA may be more common than has previously been realized. REFERENCES 1. Kaye BR, Kaye RL, Bobrove A: Rheumatoid nodules: review of the spectrum of associated conditions and

5 BRIEF REPORTS 713 proposal of a new classification, with a report of four seronegative cases. Am J Med 76: , Hams ED Jr: Rheumatoid arthritis: the clinical spectrum, Textbook of Rheumatology. Second edition. Edited by WN Kelley, ED Harris Jr, S Ruddy, CB Sledge. Philadelphia, WB Saunders, 1985, pp Decker JL, Plotz PH: Extra-articular rheumatoid disease, Arthritis and Allied Conditions. Tenth edition. Edited by DJ McCarty. Philadelphia, Lea & Febiger, 1985, pp Baggenstoss AH, Bickel WH, Ward LE: Rheumatoid granulomatous nodules as destructive lesions of vertebrae. J Bone Joint Surg 34A: , Lorber A, Pearson CM, Rene RM: Osteolytic vertebral lesions as a manifestation of rheumatoid arthritis and related disorders. Arthritis Rheum , Glay A, Rona G: Nodular rheumatoid vertebral lesions versus ankylosing spondylitis. AJR 94: , Shichikawa K, Matsui K, Oze K, Ota H: Rheumatoid spondylitis. Int Orthop , Seaman WB, Wells J: Destructive lesions of the vertebral bodies in rheumatoid disease. AJR 86: , Ball J: Pathology of the rheumatoid cervical spine (letter). Lancet I:86, Ball J: Enthesopathy of rheumatoid and ankylosing spondylitis. Ann Rheuni Dis 30: , Bland JH: Rheumatoid arthritis of the cervical spine. J Rheumatol 1:31%342, Bywaters GL: Rheumatoid discitis in the thoracic region due to spread from costovertebral joints. Ann Rheum Dis 33:408409, Martel W: Pathogenesis of cervical discovertebral destruction in rheumatoid arthritis. Arthritis Rheum 20: , Resnick D: Thoracolumbar spine abnormalities in rheumatoid arthritis. Ann Rheum Dis 37:38%392, 1978

Case reports CASE 1. A 67-year-old white man had back pain since the age. our clinic several years later with progressive symptoms.

Case reports CASE 1. A 67-year-old white man had back pain since the age. our clinic several years later with progressive symptoms. Annals of the Rheumatic Diseases, 1982, 41, 574-578 Late-onset peripheral joint disease in ankylosing spondylitis MARC D. COHEN AND WILLIAM W. GINSBURG From the Division ofrheumatology and Internal Medicine,