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Apatient with Severe Palindromic Rheumatismand Frequent CASE REPORT A Patient with Severe Palindromic Rheumatism and Frequent Episodes of Pain Shingo Takata, Seishi Harada, Fumihiro Mitsunobu, Takashi Mifune, Yasuhiro Hosaki, Kozo Asida, Hirofumi Tsugeno, Makoto Okamoto, Mitsuhiro Iwahashi* , Masanori Kawashima* , Masahiro Yamamura*, Hirofumi Makino* and Yoshiro Tanizaki Abstract Case Report A 44-year-old man began to experience episodes of joint A44-year-old man had the chief complaint of recurrent epi- pain with erythema in his knees, elbows, shoulders, and sodes of joint pain with erythema. Although his past medical hands in April 1996. He was diagnosed as having palin- history was unremarkable in his family history, his grandmother dromic rheumatism. Due to the increasing frequency and had rheumatoid arthritis. severity of these episodes, he was admitted to our hospital The patient first noticed severe knee pain with erythema in in May1999. Heat therapy to the affected area produced a April 1996. Prior to that time he had generally been in good rapid improvementin symptoms. In addition, the contin- health. The pain was relieved with oral NSAIDtherapy. Sub- ued use of physical therapy during symptom-free periods sequently, the patient had recurrent episodes of pain involving tended to reduce the frequency and severity of pain attacks. the knees, hands, and elbows. The pain occurred every week, Wepresent this case and discuss treatment options in pa- lasted 3 to 7 days, and was generally worse in the late evening tients with palindromic rheumatism. or early morning. The patient was diagnosed with gout by his (Internal Medicine 40: 140-143, 2001) primary care physician based on an elevated serum uric acid level. However, despite normalization of the serum uric acid Key words: rheumatoid arthritis, HLADR-4, spa therapy, with treatment, the patient continued to have episodes of joint warmcompress, vicious cycle pain. The patient was examined at the Ryuso Orthopedic Clinic in November1998. He was then referred to the Third Depart- ment of Internal Medicine at the OkayamaUniversity Hospi- Introduction tal, where laboratory tests for antinuclear antibody, anti-ENA antibody, and rheumatoid factor were negative. The patient was Palindromic rheumatism is a disorder that was first described diagnosed with palindromic rheumatism, and was hospitalized in 1944 by Hench and Rosenberg at the MayoClinic (1). "Pal- at the Third Department of Internal Medicine at Okayama indromic" refers to a rheumatic disorder characterized by re- University Hospital from 10 April 1999 to 30 April 1999 for current episodes of inflammation interspersed with symptom- rest and more detailed evaluation. Chest and bilateral shoul- free intervals ( 1 ). "Rheumatism" refers to inflammation involv- der, hand, elbow, and knee radiographs showed no evidence of ing the joints and periarticular tissues (1). This case concerns a abnormalities. The patient continued to have episodes of pain patient with "palindromic rheumatism"in whomheat therapy while in the hospital, but the use of warmcompresses helped to the affected areas of joint involvement reduced the frequency to relieve his symptoms.The patient was admitted to our hos- and severity of pain attacks. The treatment prescribed for se- pital on May 7 1999 for balneotherapy (spa therapy), includ- vere palindromic rheumatismis often similar to that used dur- ing the use of warmcompresses. On admission, his tempera- ing the early stages of rheumatoid arthritis. Nevertheless, there ture was 36.5°C, height 171.5 cm, weight 72.6 kg, blood pres- have been no formally established therapeutic guidelines. In sure 140/80, and pulse rate, 56/min and regular. Physical ex- addition to this case, here we discuss other treatment options amination revealed no pallor of the palpebral conjunctivea, no for severe palindromic rheumatism. scleral icterus, and no edemaof the palpebral conjunctivea. The lungs, heart, and abdomenwere normal. There was no From the Division of Medicine, the Misasa Medical Branch, OkayamaUniversity Medical School, Misasa and *the Department of Medicine III, Okayama University Medical School, Okayama Received for publication March 24, 2000; Accepted for publication August 7, 2000 Reprint requests should be addressed to Dr. Shingo Takata, the Division of Medicine, the Misasa Medical Branch, Okayama University Medical School, Misasa, Tottori 682-0192 140 Internal Medicine Vol. 40, No. 2 (February 2001) Palindromic Rheumatism and Episodes of Pain Table 1. Hematologic Values on Admission C B C B l o o d C h e m i c a l F i n d i n g s C a 9 . 6 m g / d l W B C 9 , 6 0 0 / u l T . R 7 . 6 g / d l C R P 0 . 2 m g / d l L y 2 4 .7 % B U N 2 2 . 6 m g / d l A S T 1 0 U / / M o n 7. 1 % U . A . 7 . 4 m g / d l A L T 1 7 U / / G r 6 8 . 2 % C r e a 1 . 2 m g / d l A L P 8 9 W l E O O % A l b 3 . 7 g / d l L D H 1 3 5 U / Z B A O % F B S 8 1 m g / d l C P K ¥ 4 6 U / l R B C 4 5 8 x l O 4/u l C H O L 1 7 3 m g /d l G G T 3 1 W l H b 1 4 . 7 g / d l T R I G 1 3 5 m m o l / / E S R 2 9 m m / h H c t 4 2. 1 % C L 1 0 2 . 1 m m o l / / 2 4 h C c r 7 9 . 8 m l / m i n P L T 1 9 .5 x l O 7 u l N A 1 4 2 . 1 m m o l / / K 3 . 9 9 m m o l / / Table 2. Additional Laboratory Findings on strength. Oral treatment with Keishi-ka-jutsubu-to (a Chinese Admission herbal medicine) was started on July 28, 1999. By the second month of hospitalization, the interval between episodes of joint Rheumatoid factor (-) IgG 1,723 mg/dl pain had increased to the point where attacks only occurred IgA 527 mg/dl about twice per month. Theduration of symptomsalso de- > IgM 1 35 mg/dl creased. Rest and application of warmcompresses usually pro- C3 88.0 mg/dl vided pain relief within 1 to 2 days. Other generalized symp- C4 27.0 mg/dl toms such as fever during pain attacks also disappeared. anti-nuclear antibodies x40 Treatment with oral NSAIDswas not used at the patient's anti -nRNP (-) request. Other therapy such as colchicine, sodium aurothio- anti-Sm (-) malate, and D-penicillamine (reported to be effective in the anti-S S -A (-) anti-S S -B (-) medical literature) also was not used. Monthly tests for rheu- HLA-DR4 (-) matoid factor remained negative. HLAtyping for DR4was also negative. HLA-DR4has been associated with a transition from palindromic rheumatism to rheumatoid arthritis. The pa- tient was discharged from the hospital on October 5, 1999. evidence of joint tenderness or swelling, skin rash, superficial lymphadenopathy, morning stiffness, or Raynaud's phenom- Discussion enon. The laboratory findings on admission are shown in Table 1. The patient's BUNwas 22.6 mg/dl. The serum uric acid was Palindromic rheumatism is a disorder characterized by re- initially elevated at 7.4 mg/dl, but repeat testing during hospi- current episodes of acute inflammation of the joints and peri- talization showednormal levels. The erythrocyte sedimenta- articular tissues. The hands, fingers, knees, and shoulders are tion rate at 1 hour was slightly elevated at 29 mm.However, commonlyaffected, and symptoms typically last from a few this was measured immediately after an episode of joint pain. hours to 2 or 3 days. These attacks are interspersed by com- The CRP was normal. Immunologic studies (Table 2) showed pletely symptom-free periods. Hence, the name "palindromic", an elevated IgA of 527 mg/dl. Antinuclear antibody, rheuma- which is of Greek origin, means "to run back" (1). Palindrom- toid factor, and anti-Sm antibody were negative. ic rheumatismoccurs equally amongmenand womenand gen- The episodes of joint pain (with erythema) in the knees, el- erally affects patients in their 20s to 50s (1). Most laboratory bows, and hands that were present on hospital admission con- test findings, including serum uric acid, are normal. The eryth- tinued to recur at weekly intervals. These symptoms lasted as rocyte sedimentation rate and other acute reactive products may long as 7 days and were accompanied by fever (38°C range). be elevated after a pain attack. Radiographic findings remain After the onset of nocturnal pain attacks, the affected areas normal regardless of the number of joint pain episodes (1). were heated by application of warmcompresses, followed by Gonzalez-Lopez et al reported that approximately 30%of pa- the use of hot packs in the morning. This local heat application tients diagnosed with palindromic rheumatism eventually de- helped to relieve the pain. Cool compresses were not used be- velop rheumatoid arthritis (2). Manyof these patients are al- cause this only further intensified the patient's symptoms. Physi- ready positive for serum rheumatoid factor and HLADR-4(3). cal therapy, consisting primarily of heat therapy, to the affected Other patients maybecomepositive for rheumatoid factor at joints (especially the knees and hands) was continued even the time that rheumatoid arthritis actually develops (4, 5). In during symptom-free periods. The patient also performed addition, some reports have described patients with palin- stretching exercises in a warmwater pool to enhance muscle dromic rheumatism whohave developed other collagen vas- Internal Medicine Vol. 40, No. 2 (February 2001) 141 Takata et al cular disorders such as systemic lupus erythematosus (2, 5).
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