Vol.1_No.2_Dec.2012.book

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1 (2012 ) Glenn 30 CRP 2 28 CRP CRP 1 hypertrophic osteoarthropathy, cyanosis, congenital heart disease (hypertrophic osteoarthropathy HOA) 3 HOA 2 30 {S,L,L}, mg/dL 2 178cm 60kg /60mmHg 76 /SpO2 80 ( ) 3/6 (Table 1) CRP X (Figure 1) (Figure 2) (Figure 3) 2.5mg 1 3 5mg/ 3mg/ () take down 21 CRP 55

GTP CK BUN Cre egfr 4630 77.2 9.9 509 16.2 49.2 15.0 6.0 3.3 20 25 183 313 20 64.9 1.74 40.3 I I ml/min/ 1.73m 2 Table 1 Laboratory date UA 7.1 Na 134 K 5.2 Cl 102 Ca 8.6 P 4.

2 ( ) 9.6mg/dL cm 39kg /50mmHg 110 / (VVIR , all pacing) SpO2 83 ( ) 3/6 1/6 (Table 2) CRP X (Figure 4) (Figure 5) (Figure 6) 40 5mg/ 5mg/ 2mg/ WBC Neu Lym RBC Hb Ht Plt TP Alb AST ALT LDH GTP CK BUN Cre egfr I I ml/min/ 1.73m 2 Table 1 Laboratory date UA 7.1 Na 134 K 5.2 Cl 102 Ca 8.6 P 4.4 T Bil 3.1 AMY 97 CRP 7.26 Feritin 451 TSH 5.83 ft ft BNP 567 PT INR 2.51 APTT 159 AT 78 FDP 2.3 D dimer 0.91 ng/ml U/ml pg/ml n pg/ml g/ml g/ml MMP3 204 IgG 988 IgA 64 IgM 119 CH C3 102 C <20 RA U/ml NG/ML U/ml Figure 1 Bone X-ray Bone X-ray showed raised, thickend,irregular periosteum ( ) 56

(2012 12 ) Figure 2 Bone scintigraphy Bone scintigraphy showed symmetrically abnormal linear accumulation along tibia and fibula ( ).

3 ( ) Figure 2 Bone scintigraphy Bone scintigraphy showed symmetrically abnormal linear accumulation along tibia and fibula ( ). Figure 3 Clinical course Table 2 Laboratory date WBC Neut Lym RBC Hb Ht Plt TP Alb AST ALT LDH ALP CK BUN Cre egfr UA Na K Cl ml/min/ 1.73m2 Ca P Mg T Bil CRP Feritin ESR 60min 120min TSH ft3 ft4 BNP PT INR APTT Fib AT FDP D dimer IgG IgA IgM ng/ml mm mm U/ml pg/ml n pg/ml g/ml g/ml CH50 C3 C4 RA dsdnag dsdnam CCP U1 RNP Sm MPO ANCA PR3 ANCA EBV VCA IgG IgM EBV EBNA C.Trachoma IgG IgM DNA ( ) <20 <2 <0.6 <7 < U/ml U/ml 57

4 ( ) Figure 4 Bone X-ray Bone X-ray showed raised, thickend,irregular periosteum.( ) Figure 5 Bone scintigraphy Bone scintigraphy showed symmetrically abnormal linear accumulation.( ) 関節所見 Body Temperature ( ) アセトアミノフェンナプロキサン 150mg PSL 内服 5mg/day 5mg/day CRP 6.3 () days( 日 ) days( 日 ) Figure 6 Clinical course 58

5 ( ) 3 1,2) Platelet-Derived Growth Factor (PDGF) Vascular Endothelial Growth Factor (VEGF) 3) 4) 2 5,6) Martinez-Lavin ) platelet derived growth factor (PDGF) transforming growth factor- (TGF- ) 3) HOA HOA 7,8) HOA 10) NSAIDS 1) 1 38(3): , ),,,. 11: 55-60, ) L H Silveria, M Martinez-Lavin, C Pineda, et al: Vascular endothelial growth factor and hypertrophic osteoarthropathy. Clinical and experimental rheumatology 18(1);57-62, ) Rosenthal L,Kirsh J: Observation on radionuclide imaging in hypertrophic pulmonary osteoarthropathy. Radiology 120:359, ) Solomon E Levin, Jeffrey R Harrisberg, Kenny Govendrageloo, et al: Familial primary hypertrophic osteoarthropathy in association with congenital cardiac disease. Cardiology in the young 12(3); , ) Wijesekera VA, Radford DJ. Hypertrophic Eisenmenger Syndrome. Congenit Heart Dis 7.doi: /j ) Yuichi Takiguchi:Osteoarthropathy. Cancer and chemotheraphy,37(6); , ) Rowan E Miller, Rowland O Illing,Jeremy S Whelan: Lung carcinoma with hypertrophic osteoarthropathy in a teenager, Rare tumors 3(1);e8, ) Martinez-Lavin M, Bobadilla M, Casanova J, Attie F, Martinez M. Hypertrophic osteoarthropathy in cyanotic heart disease: its prevalence and relationship to bypass of the lung. Arthritis Rheum. 25(10): ; ) Frand M, Koren G, Rubinstein Z. Reversible hypertrophic osteoarthropathy associated with cyanotic congenital heart disease. Am J Dis Child 136(8): ;

6 ( ) Hypertrophic osteoarthropathy associated with adult cyanotic congenital heart disease Masayuki Abiko, Kei Inai, Gaku Izumi, Kouji Sonoda, Eriko Shimada, Tokuko Shinohara, Hirofumi Tomimatsu, Toshio Nakanishi Department of Pediatric Cardiology, Tokyo Women s Medical University We describe two adult patients with cyanotic congenital heart disease accompanied by hypertrophic osteoarthropathy (HOE). Case 1 is a 30-year-old male who was diagnosed with a single left ventricle during the Glenn operation. While hospitalized with heart failure, he developed a slight fever followed by arthritic pain accompanied by swelling and an increase in C-reactive protein (CRP) levels. Hypertrophic osteoarthropathy was diagnosed based on the bone imaging findings. After confirming reactivity to intravenous dexamethasone, the patient was switched to a prednisolone (small quantity for internal use) and his condition went into remission. Case 2 is a 28-year-old male diagnosed with a single left ventricle during a pulmonary artery banding operation. Fever persisted, CRP levels became elevated and antibiotic treatment was ineffective. Various laboratory cultures were negative and he was also negative for infectious endocarditis, collagen diseases, malignancies tumors and other conditions. While hospitalized, he developed pain in several joints, which was diagnosed as hypertrophic osteoarthropathy based on bone imaging findings. The arthritic pain was promptly improved by prednisolone (small quantity for internal use). Arthritic pain is one long-term complication of cyanotic heart disease, and the incidence will probably increase in the future. 60

BUN, CRP K mg/ cm, 49.6 kg, BMI /72 mmhg, 92/ Hb 6.7 g/dl PT-INR CT 1 MRI 2a, b T1 T2 T1 MRI

BUN, CRP K mg/ cm, 49.6 kg, BMI /72 mmhg, 92/ Hb 6.7 g/dl PT-INR CT 1 MRI 2a, b T1 T2 T1 MRI 20 2 2015 1 2 3 85 K CT Abstract A case of renal subcapsular hematoma resulting from trigger point injection under excessive effect of anticoagulant YAMANE Tateki, UMEDA Akira and SHIMAO Hitoshi An 85-year-old