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PEDIATRICS Vol. 112 No. 5 November 2003, pp. 1065-1068

Rheumatic Fever in Children Younger Than 5 Years: Is the Presentation Different?

Lloyd Y. Tani, MD, L. George Veasy, MD, L. LuAnn Minich, MD and Robert E. Shaddy, MD

From the Department of Pediatrics, University of Utah, and Primary Children’s Medical Center, Salt Lake City, Utah


   ABSTRACT
TOP
 ABSTRACT
METHODS
 RESULTS
 DISCUSSION
 CONCLUSION
 REFERENCES
 
Objective. To review our experience with children who presented with rheumatic fever (RF) before 5 years of age and to compare their presentation with that of older children.

Methods. The cardiology database was reviewed to identify patients who were younger than 5 years and had a diagnosis RF using the Jones criteria from January 1985 through March 2000. Patient age, sex, date and age at presentation, and the major Jones criteria fulfilled were noted. When carditis was present, its severity was judged to be moderate to severe when there was radiographic cardiomegaly and/or clinical congestive heart failure. The clinical presentation of patients who presented in the first 5 years of life were compared with the presentation of those whose RF was diagnosed after 5 years of age. Clinical findings at follow-up evaluation and echocardiographic findings both at presentation and at follow-up were noted for the children who were younger than 5 years at presentation.

Results. Of 541 cases of RF seen from January 1985 through March 20000, 27 (5%) were in children who were younger than 5 years (median: 4.0 years; range: 1.9–4.9 years). Major Jones criteria at presentation were arthritis in 17, carditis in 14, chorea in 3, and erythema marginatum in 3. The carditis was mild in 4 and moderate to severe in 10 patients. Compared with older children, younger children were more likely to present with moderate to severe carditis, arthritis without carditis or chorea, or the rash of erythema marginatum and were less likely to have chorea. The incidence of carditis was similar in the 2 groups as was the ratio of boys to girls. At follow-up (9.6 ± 5.6 years), 69% of younger children who presented with carditis have clinical rheumatic heart disease. Subclinical, echocardiographically detected valvular abnormalities were detected both at presentation (33% of all children with RF before 5 years of age) and at follow-up (55% of those who initially had carditis).

Conclusions. Approximately 5% of children with RF were younger than 5 years at diagnosis. Compared with older patients, children who presented before 5 years of age were more likely to have moderate to severe carditis and to present with arthritis or the rash of erythema marginatum and were less likely to have chorea. Chronic rheumatic heart disease was common in young children who presented with carditis. Long-term follow-up is necessary to determine the outcome for young children with subclinical echocardiographic evidence of valvular disease.

Key Words: rheumatic fever

Abbreviations: RF, rheumatic fever

Rheumatic fever (RF) is a leading cause of acquired heart disease in children and young adults in many parts of the world.1,2 Although less common in industrialized countries, a resurgence of RF activity occurred in the intermountain west portion of the United States in the mid-1980s3 and has persisted through the 1990s and into the 21st century. Previous series have reported that RF both is uncommon and may present differently in children who are younger than 5 years.4–6 The purposes of this article are to report our experience with RF in children who presented before 5 years of age and to compare the presentation of these children with that of older children.


   METHODS
TOP
 ABSTRACT
 METHODS
RESULTS
 DISCUSSION
 CONCLUSION
 REFERENCES
 
Patients
The Pediatric Cardiology RF database at the University of Utah was reviewed to identify children who were younger than 5 years and received a diagnosis of RF from January 1985 through March 2000 using the Jones criteria.7,8 Patient age at presentation, sex, and clinical presentation were compared with children who were aged 5 to 18 years and received a diagnosis of RF during the same time period. For children who were younger than 5 years at presentation, charts were reviewed for clinical follow-up data and echocardiographic findings at presentation and at follow-up evaluation.

Jones Criteria
The diagnosis of polyarthritis required clinical evidence of inflammation (typically warm, red, swollen) and limitation of motion of >1 joint. Patients with Sydenham chorea had the involuntary, spasmodic, purposeless movements characteristic of this disorder (confirmed by a pediatric neurologist). Carditis was diagnosed only when there was a new murmur of mitral and/or aortic regurgitation. The carditis was considered moderate to severe when there was radiographic cardiomegaly and/or clinical evidence of congestive heart failure. Clinical carditis without cardiomegaly or heart failure was considered mild. The diagnosis of erythema marginatum required historical or observed evidence of the characteristic evanescent macular erythematous rash with serpiginous borders and central clearing.

Echocardiography
Doppler evidence of valvular regurgitation in the absence of clinical findings was not considered carditis as stated in the Jones criteria.7,8 Complete 2-dimensional, M-mode, and Doppler echocardiography was performed on all but a few patients in 1985 and 1986. Since 1987, echocardiography has been performed in all cases to confirm clinical findings, evaluate chamber sizes, assess systolic ventricular function (shortening fraction and/or ejection fraction), and identify pericardial effusions. Color Doppler became a routine part of the echocardiographic evaluation in 1990. Since January 1992, strict criteria have been used to distinguish pathologic from physiologic valvular regurgitation in patients without an audible murmur of mitral or aortic regurgitation.9 For pathologic mitral regurgitation, all of the following criteria were met: 1) a color jet seen from at least 2 imaging planes; 2) a color jet extending >1 cm into the left atrium behind the mitral valve; and 3) pulsed or continuous wave Doppler showing the regurgitant jet to be holosystolic, of high velocity, and turbulent (aliasing).9–12 For aortic regurgitation, a diastolic color jet seen in at least 2 planes and extending >5 mm into the left ventricular outflow tract was considered pathologic. Doppler-detected aortic regurgitation rarely, if ever, occurs in normal children.13,14

Clinical Follow-up
The records of children who were younger than 5 years and presented with carditis were reviewed for the most recent follow-up data, including findings on auscultation, need for surgical intervention, and echocardiographic evidence of rheumatic valvular disease.

Statistical Analysis
Patients who presented in the first 5 years of life were compared with those who presented later in life ({chi}2 or Fisher exact test). For children who presented with carditis, the cardiac status at follow-up evaluation was compared with the severity of cardiac involvement at presentation. Data are presented as mean ± standard deviation. Data were analyzed using SigmaStat software (San Rafael, CA). P < .05 was considered statistically significant.


   RESULTS
TOP
 ABSTRACT
 METHODS
 RESULTS
DISCUSSION
 CONCLUSION
 REFERENCES
 
Of 541 patients that fulfilled the Jones criteria for RF from January 1985 through March 2000, 27 (5.0%) were younger than 5 years (3.96 ± 0.87; median: 3.95; range: 23–59 months); 12 were between 4 and 5 years of age, 10 were between 3 and 4 years of age, 4 were between 2 and 3 years of age, and 1 was younger than 2 years. Of these 27 children, 13 were boys and 14 were girls.

The major Jones criteria at presentation are given in Table 1. Arthritis without chorea or carditis was more common in the younger group (11 of 27 [41%]) than in older patients (104 of 514 [20%]; P = .02). Compared with older patients, the rash of erythema marginatum was more common in the younger patients (3 of 27 [11%] vs 13 of 512 [2.5%]; P = .04). Any presentation with chorea (alone or with arthritis or carditis) was less common in children who were younger than 5 years at presentation (3 of 27 [11%]) than in the older children (167 of 512 [33%]; P = .04).


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  		TABLE 1. Clinical Presentation of RF

 
Compared with older patients, children who were younger than 5 years had a similar incidence of carditis (either alone or with arthritis or chorea). Of the 14 younger children with carditis, 12 had a murmur of mitral regurgitation alone, 1 had audible mitral and aortic regurgitation, and 1 had audible aortic regurgitation alone. When present, the carditis was more often moderate to severe (as opposed to mild) in the younger children (10 of 14 [71%]) than in older patients (115 of 313 [37%]; P = .02).

Echocardiography was performed at presentation in all 27 children who presented before 5 years of age and confirmed clinical findings in all 14 cases with clinical carditis. Of the 12 patients who had a murmur of mitral regurgitation but no diastolic murmur, 3 had Doppler evidence of aortic regurgitation. The 1 child with audible aortic regurgitation but no murmur of mitral regurgitation had Doppler evidence of pathologic mitral regurgitation. Of the 13 children with no evidence of clinical carditis (ie, arthritis or chorea with no murmur), 5 (38%) had subclinical evidence of pathologic mitral and/or aortic regurgitation. Thus, 9 (33%) of 27 children who presented with RF before 5 years of age had subclinical, Doppler-detected pathologic mitral or aortic regurgitation.

Clinical follow-up data were available for 13 of 14 children who presented with clinical carditis before 5 years of age; 1 patient was lost to follow-up. At an average interval of 9.6 ± 5.6 years after presentation with RF, 9 (69%) have clinically evident rheumatic heart disease; the other 4 patients now have normal cardiac examinations. All 9 children with clinical rheumatic heart disease have audible mitral regurgitation. One patient who had audible mitral regurgitation but only Doppler-detected aortic regurgitation at presentation now has murmurs of both aortic and mitral regurgitation. No patient has a mid- to late- diastolic murmur or opening snap, findings of mitral stenosis. Follow-up echocardiography has been performed in 11 patients. Two patients have subclinical aortic regurgitation, 1 has subclinical pathologic mitral regurgitation, and 3 have subclinical mitral stenosis (thickened leaflets, restricted motion, or increased Doppler velocity). All 3 patients with subclinical mitral stenosis had moderate to severe carditis at presentation. One patient, a girl who was 3.8 years of age and had severe mitral regurgitation at presentation, has required surgery (mitral valvuloplasty almost 8 years after presentation). Initial carditis severity was not predictive of clinical chronic rheumatic heart disease at this relatively short follow-up interval. At follow-up, 7 of 9 with initially moderate to severe carditis and 2 of 4 with initially mild carditis have clinically evident chronic rheumatic heart disease.


   DISCUSSION
TOP
 ABSTRACT
 METHODS
 RESULTS
 DISCUSSION
CONCLUSION
 REFERENCES
 
The most important finding of our study is that approximately 5% of children with RF were younger than 5 years at presentation. Therefore, RF should be considered in the differential diagnosis of young children who present with arthritis, chorea, a rash suggestive of erythema marginatum, or a murmur of mitral regurgitation. Although RF is considered to be an entity occurring mainly between the ages of 5 and 15 years,4,6 our numbers are similar to the 4.5% to 6.8% reported by some15,16 but higher than the 1% to 2% reported by others.4,17,18 The different incidences reported may be attributable to differences among populations, but the method of case ascertainment also may have been important. Certainly, some cases of RF in young children may go undiagnosed, adding to the variability in the reported incidences.

The clinical presentation of children whose RF was diagnosed before 5 years of age was different from that of older children. Compared with those older than 5 years, younger children were more likely to present with arthritis without carditis or chorea but less likely to present with chorea. In addition, the rash of erythema marginatum was seen more commonly in the group of children who were younger than 5 years at diagnosis. Our data are similar to previous series that have reported arthritis to be more common in young children.5,19,20 Although reported by some series to be virtually nonexistent in young children,5,20 chorea has been reported in an 8-month-old child, and another series found a 7% incidence of chorea in young children with RF.15 Erythema marginatum is a relatively uncommon finding in patients with RF and may be easily missed because it is evanescent, occurring mainly on the trunk and proximal extremities, and is not associated with symptoms of pain or pruritus.21,22 Although the reasons for the higher incidence of this rash in younger children are unknown, we speculate that this may in part be because parents are more likely to participate in the bathing and dressing of younger children and thus more likely to notice a fleeting, mainly truncal rash.

When carditis was present, it was more often moderate to severe in younger than in older patients. The severity of involvement at presentation is important because the 2 main risk factors reported to influence the development of chronic rheumatic heart disease are the severity of involvement at initial presentation and RF recurrences.23–25 Furthermore, younger age at presentation has been reported to be associated with an increased risk of RF recurrence.26 Other series have also reported greater severity of cardiac involvement in younger children.5,20 In contrast to some previous series reporting carditis to be more common in younger children,5,18,19 we found no difference in the incidence of carditis in children who were younger than 5 years of age compared with older children. The 69% of children who presented with carditis before 5 years of age and developed chronic rheumatic heart disease is similar to the overall incidence of 66% reported by Feinstein et al,25 who followed 441 patients with RF of all ages. Because the natural history of rheumatic heart disease in developed countries such as the United States is one of slow progression with most adults presenting in the fourth or fifth decade,27,28 the follow-up interval of our study is not of sufficient duration to determine the influence of initial carditis severity on long-term outcome.

We found echocardiographic evidence of subclinical rheumatic valvular disease (mitral stenosis or regurgitation, aortic regurgitation) at the time of follow-up evaluation in several of the children who presented before 5 years of age. Doppler-detected subclinical pathologic valvular regurgitation has been reported in several other series of patients with RF.10,11,29–31 There is indirect evidence supporting the existence and potentially the importance of subclinical rheumatic heart disease.23 Many adults who present with chronic rheumatic heart disease cannot recall having an illness resembling RF.32,33 In addition, as many as 20% to 34% of patients with "pure" chorea have been reported to develop chronic rheumatic heart disease.34–36 In their classic report of 1000 patients with RF followed for 20 years, Bland and Jones23 reported that rheumatic heart disease developed in 44% of patients who initially had no clinical evidence of carditis. Presumably, these patients had either very mild or subclinical rheumatic heart disease that progressed over time. Despite this indirect evidence, how often subclinical, echocardiographically detected rheumatic valvular changes progress to clinically significant rheumatic heart disease is unknown. In addition, Doppler may detect small amounts of valvular regurgitation in "normals." Although criteria have been established to differentiate this physiologic mitral regurgitation from pathologic regurgitation,9 the existence of such valvular pathology has been disputed.37,38 Thus, at present, the role of echocardiography in the evaluation and management of patients with RF who have no clinical cardiac involvement remains controversial.8,39–41

Our study has limitations. The study was retrospective, with a relatively small number of patients who presented before 5 years of age. As is common for a retrospective study, clinical follow-up data were incomplete. For chronic rheumatic heart disease, which usually presents in the fourth or fifth decade, the duration of follow-up in our study was relatively short.


   CONCLUSION
TOP
 ABSTRACT
 METHODS
 RESULTS
 DISCUSSION
 CONCLUSION
REFERENCES
 
Although uncommon, RF does occur in children who are younger than 5 years. When it occurs, presentation with arthritis is more common and chorea is less common than in older patients. In addition, the rash of erythema marginatum is noted more frequently in younger children. When present, cardiac involvement is more often moderate to severe (as opposed to mild) in these younger children. Persistent, chronic rheumatic heart disease is common in these young children who present with carditis. Subclinical, echocardiographically detected cardiac involvement is also common, but additional study will be important to determine the prognostic and clinical value of such findings.


   ACKNOWLEDGMENTS
 
We thank Paul C. Young, MD, for review of this manuscript.


   FOOTNOTES
 
Received for publication Dec 30, 2002; Accepted Apr 14, 2003.

Reprint requests to (L.Y.T.) Primary Children’s Medical Center, 100 North Medical Dr, Salt Lake City, UT 84113. E-mail: pcltani{at}ihc.com


   REFERENCES
TOP
 ABSTRACT
 METHODS
 RESULTS
 DISCUSSION
 CONCLUSION
 REFERENCES
 

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PEDIATRICS (ISSN 1098-4275). ©2003 by the American Academy of Pediatrics

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