Limits...
A case of chloroquine-induced cardiomyopathy that presented as sick sinus syndrome.

Lee JH, Chung WB, Kang JH, Kim HW, Kim JJ, Kim JH, Hwang HJ, Lee JB, Chung JW, Kim HL, Choi YS, Park CS, Youn HJ, Lee MY - Korean Circ J (2010)

Bottom Line: A 52-year-old woman with rheumatoid arthritis who had been treated with prednisone and hydroxychloroquine for >12 years presented with chest discomfort and a seizure.She was diagnosed with restrictive cardiomyopathy combined with sick sinus syndrome.This is the first case report describing chloroquine-induced cardiomyopathy involving the heart conduction system.

View Article: PubMed Central - PubMed

Affiliation: Division of Cardiology, Department of Internal Medicine, College of Medicine, The Catholic University of Korea, Seoul, Korea.

ABSTRACT
A 52-year-old woman with rheumatoid arthritis who had been treated with prednisone and hydroxychloroquine for >12 years presented with chest discomfort and a seizure. She was diagnosed with restrictive cardiomyopathy combined with sick sinus syndrome. A myocardial muscle biopsy was performed to identify the underlying cardiomyopathy, which showed marked muscle fiber hypertrophy, fiber dropout, slightly increased interstitial fibrous connective tissue, and extensive cytoplasmic vacuolization of the myocytes under light microscopy. Electron microscopy of the myocytes demonstrated dense, myeloid, and curvilinear bodies. The diagnosis of hydroxychloroquine-induced cardiomyopathy was made based on the clinical, hemodynamic, and pathologic findings. This is the first case report describing chloroquine-induced cardiomyopathy involving the heart conduction system.

No MeSH data available.


Related in: MedlinePlus

Two-dimensional echocardiography reveals severe concentric LV hypertrophy, mild LV systolic dysfunction, a small pericardial effusion, and increased RV free wall thickness with mild RV systolic dysfunction. A: parasternal long axis view. B: parasternal short axis view at the mid-ventricular level. C: magnified apical 4-chamber view. D: mitral inflow view (E velocity, 90.28 cm/s; A velocity, 36.77 cm/s; deceleration time, 216.64 ms; E/E': 33.4). LV: left ventricular, RV: right ventricular.
© Copyright Policy - open-access
Related In: Results  -  Collection

License
getmorefigures.php?uid=PMC3008834&req=5

Figure 3: Two-dimensional echocardiography reveals severe concentric LV hypertrophy, mild LV systolic dysfunction, a small pericardial effusion, and increased RV free wall thickness with mild RV systolic dysfunction. A: parasternal long axis view. B: parasternal short axis view at the mid-ventricular level. C: magnified apical 4-chamber view. D: mitral inflow view (E velocity, 90.28 cm/s; A velocity, 36.77 cm/s; deceleration time, 216.64 ms; E/E': 33.4). LV: left ventricular, RV: right ventricular.

Mentions: A 52-year-old woman was hospitalized for evaluation of a 2-day-history of chest discomfort. She had rheumatoid arthritis for 13 years and had been treated with hydroxychloroquine for >12 years (total dose, 1,898 grams). In the emergency department, she had a generalized tonic-clonic seizure caused by sinus arrest (Fig. 1A). The initial vital signs were as follows: blood pressure, 130/80 mmHg; heart rate, 38 beats/minute; body temperature, 36.4℃; and respiratory rate, 20/minute. The physical examination revealed a mild degree of jugular venous distention, a regular, slow cardiac beat without murmurs, clear lung sounds, and no pitting edema in the extremities. The 12-lead ECG showed a junctional rhythm with sinus arrest (Fig. 1B). The laboratory findings included a hemoglobin level of 13.1 g/dL and a normal white blood cell count with a normal differential cell count. The blood urea nitrogen was 27 mmol/L, the serum creatinine was 2.02 mg/dL and the C-reactive protein was 0.94 mg/L. The antinuclear and anti-double stranded deoxyribonucleic acid (DNA) antibodies were negative, while the rheumatoid factor was positive. The creatine kinase was 294 UI/L with a normal myocardial band fraction. The aspartate aminotransferase level was 57 U/L, the alanine aminotransferase level was 48 U/L, and the alkaline phosphatase level was 49 UI/L. Chest radiography demonstrated marked cardiomegaly without pulmonary congestion (Fig. 2). Transthoracic echocardiography (TTE) revealed a markedly thickened left ventricular (LV) septum and posterior wall thickness, measuring 17.7 mm and 18.8 mm, respectively, with mild LV systolic dysfunction (ejection fraction, 44%). A slightly thickened RV free wall with mild RV systolic dysfunction was also noted. Both atria were enlarged and a mild degree of mitral regurgitation was present. The mitral inflow pattern was consistent with restrictive physiology. The early mitral inflow (E) velocity was 90.28 cm/s, the late mitral inflow (A) velocity was 36.77 cm/s, and the deceleration time was 216.64 milliseconds. The E/E' was 33.4, suggesting a high left ventricular end diastolic pressure (Fig. 3). Magnetic resonance imaging (MRI) showed diffuse wall thickening of the LV (septal wall thickness at the end-diastolic phase, 19 mm) and the RV free wall, suggesting hypertrophic cardiomyopathy. There was a small pericardial effusion. On delayed enhancement, multifocal patchy myocardial enhancement was noted in the lateral and septal walls (Fig. 4). The patient underwent myocardial biopsy to determine the underlying cause of the cardiomyopathy. Light microscopy showed vacuolated myocytes (Fig. 5A and B). Electron microscopy revealed abundant intra-myocyte lysosomes with numerous large, dense myelin figures occupying large portions of the myocyte sarcoplasm. Lysosomal inclusions with curvilinear substructures were also noted (Fig. 5C and D). There was no evidence of amyloid deposition, myocarditis, or an acute vasculitic process. Chloroquine toxicity was diagnosed based on the pathologic findings and the hydroxychloroquine was promptly discontinued. A permanent pacemaker was inserted for management of sick sinus syndrome. Four months later, the follow-up TTE demonstrated no significant interval change in LV systolic function or the LV wall thickness compared with the previous examination. However, LV diastolic function improved from restrictive physiology to pseudonormal relaxation. The patient's rheumatoid arthritis was subsequently controlled with low-dose prednisolone and analgesics.


A case of chloroquine-induced cardiomyopathy that presented as sick sinus syndrome.

Lee JH, Chung WB, Kang JH, Kim HW, Kim JJ, Kim JH, Hwang HJ, Lee JB, Chung JW, Kim HL, Choi YS, Park CS, Youn HJ, Lee MY - Korean Circ J (2010)

Two-dimensional echocardiography reveals severe concentric LV hypertrophy, mild LV systolic dysfunction, a small pericardial effusion, and increased RV free wall thickness with mild RV systolic dysfunction. A: parasternal long axis view. B: parasternal short axis view at the mid-ventricular level. C: magnified apical 4-chamber view. D: mitral inflow view (E velocity, 90.28 cm/s; A velocity, 36.77 cm/s; deceleration time, 216.64 ms; E/E': 33.4). LV: left ventricular, RV: right ventricular.
© Copyright Policy - open-access
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC3008834&req=5

Figure 3: Two-dimensional echocardiography reveals severe concentric LV hypertrophy, mild LV systolic dysfunction, a small pericardial effusion, and increased RV free wall thickness with mild RV systolic dysfunction. A: parasternal long axis view. B: parasternal short axis view at the mid-ventricular level. C: magnified apical 4-chamber view. D: mitral inflow view (E velocity, 90.28 cm/s; A velocity, 36.77 cm/s; deceleration time, 216.64 ms; E/E': 33.4). LV: left ventricular, RV: right ventricular.
Mentions: A 52-year-old woman was hospitalized for evaluation of a 2-day-history of chest discomfort. She had rheumatoid arthritis for 13 years and had been treated with hydroxychloroquine for >12 years (total dose, 1,898 grams). In the emergency department, she had a generalized tonic-clonic seizure caused by sinus arrest (Fig. 1A). The initial vital signs were as follows: blood pressure, 130/80 mmHg; heart rate, 38 beats/minute; body temperature, 36.4℃; and respiratory rate, 20/minute. The physical examination revealed a mild degree of jugular venous distention, a regular, slow cardiac beat without murmurs, clear lung sounds, and no pitting edema in the extremities. The 12-lead ECG showed a junctional rhythm with sinus arrest (Fig. 1B). The laboratory findings included a hemoglobin level of 13.1 g/dL and a normal white blood cell count with a normal differential cell count. The blood urea nitrogen was 27 mmol/L, the serum creatinine was 2.02 mg/dL and the C-reactive protein was 0.94 mg/L. The antinuclear and anti-double stranded deoxyribonucleic acid (DNA) antibodies were negative, while the rheumatoid factor was positive. The creatine kinase was 294 UI/L with a normal myocardial band fraction. The aspartate aminotransferase level was 57 U/L, the alanine aminotransferase level was 48 U/L, and the alkaline phosphatase level was 49 UI/L. Chest radiography demonstrated marked cardiomegaly without pulmonary congestion (Fig. 2). Transthoracic echocardiography (TTE) revealed a markedly thickened left ventricular (LV) septum and posterior wall thickness, measuring 17.7 mm and 18.8 mm, respectively, with mild LV systolic dysfunction (ejection fraction, 44%). A slightly thickened RV free wall with mild RV systolic dysfunction was also noted. Both atria were enlarged and a mild degree of mitral regurgitation was present. The mitral inflow pattern was consistent with restrictive physiology. The early mitral inflow (E) velocity was 90.28 cm/s, the late mitral inflow (A) velocity was 36.77 cm/s, and the deceleration time was 216.64 milliseconds. The E/E' was 33.4, suggesting a high left ventricular end diastolic pressure (Fig. 3). Magnetic resonance imaging (MRI) showed diffuse wall thickening of the LV (septal wall thickness at the end-diastolic phase, 19 mm) and the RV free wall, suggesting hypertrophic cardiomyopathy. There was a small pericardial effusion. On delayed enhancement, multifocal patchy myocardial enhancement was noted in the lateral and septal walls (Fig. 4). The patient underwent myocardial biopsy to determine the underlying cause of the cardiomyopathy. Light microscopy showed vacuolated myocytes (Fig. 5A and B). Electron microscopy revealed abundant intra-myocyte lysosomes with numerous large, dense myelin figures occupying large portions of the myocyte sarcoplasm. Lysosomal inclusions with curvilinear substructures were also noted (Fig. 5C and D). There was no evidence of amyloid deposition, myocarditis, or an acute vasculitic process. Chloroquine toxicity was diagnosed based on the pathologic findings and the hydroxychloroquine was promptly discontinued. A permanent pacemaker was inserted for management of sick sinus syndrome. Four months later, the follow-up TTE demonstrated no significant interval change in LV systolic function or the LV wall thickness compared with the previous examination. However, LV diastolic function improved from restrictive physiology to pseudonormal relaxation. The patient's rheumatoid arthritis was subsequently controlled with low-dose prednisolone and analgesics.

Bottom Line: A 52-year-old woman with rheumatoid arthritis who had been treated with prednisone and hydroxychloroquine for >12 years presented with chest discomfort and a seizure.She was diagnosed with restrictive cardiomyopathy combined with sick sinus syndrome.This is the first case report describing chloroquine-induced cardiomyopathy involving the heart conduction system.

View Article: PubMed Central - PubMed

Affiliation: Division of Cardiology, Department of Internal Medicine, College of Medicine, The Catholic University of Korea, Seoul, Korea.

ABSTRACT
A 52-year-old woman with rheumatoid arthritis who had been treated with prednisone and hydroxychloroquine for >12 years presented with chest discomfort and a seizure. She was diagnosed with restrictive cardiomyopathy combined with sick sinus syndrome. A myocardial muscle biopsy was performed to identify the underlying cardiomyopathy, which showed marked muscle fiber hypertrophy, fiber dropout, slightly increased interstitial fibrous connective tissue, and extensive cytoplasmic vacuolization of the myocytes under light microscopy. Electron microscopy of the myocytes demonstrated dense, myeloid, and curvilinear bodies. The diagnosis of hydroxychloroquine-induced cardiomyopathy was made based on the clinical, hemodynamic, and pathologic findings. This is the first case report describing chloroquine-induced cardiomyopathy involving the heart conduction system.

No MeSH data available.


Related in: MedlinePlus