Articular symptoms in hypermobile schoolchildren: A prospective study

Articular symptoms in hypermobile schoolchildren: A prospective study

944 Clinical and laboratory observations penicillin-resistantpneumococci from children over the past 3 years. The Centers for Disease Control (CDC) ...

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944

Clinical and laboratory observations

penicillin-resistantpneumococci from children over the past 3 years. The Centers for Disease Control (CDC) study showed that the prevalence of penicillin-resistantpneumococci peaked in 1982 and subsequently declined. Our findings relate to the years after 1987, the last year of the CDC study. Furthermore, we examined only isolates from children, whereas all age groups were examined as a whole in the CDC surveillance study. This increase in the number of patients with pneumococci resistant to penicillin in a children's hospital is cause for concern and warrants continued surveillance, assessment of clinical relevance, and study of alternative antibiotic therapy. REFERENCES 1. Klugman KP. Pneumococcal resistance to antibiotics. Clin Microbiol Rev 1990;3:171-96. 2. Neumann MA, Sahm DF, Thornsberry C, McGowan JE Jr. Cumitech 6A. In: McGowan JE Jr, ed. New developmentsin antimicrobial agent susceptibility testing: a practical guide. Washington, D.C.: American Society for Microbiology, 1991. 3. Carraway NC, Hawkins E, Hinds D, Mason E. Penicillin susceptibility of Streptococcus pneumoniae in a private pediatric hospital in Houston, Texas. Antimicrob Agents Chemother 1989;33:1111-2.

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4. Spika JS, Facklam RR, Plikaytis BD, et al. Antimicrnbial resistance of Streptococcus pneumoniae in the United States, 1979-1987. J Infect Dis 1991;163:1273-8. 5. National Committee for Clinical Laboratory Standards. Performance standards for antimicrobial disk susceptibilitytests: tentative standard. 4th ed. NCCLS document No. M2-T4. Villanova, Pa.: The Committee, 1988. 6. National Committee for ClinicalLaboratory Standards. Methods for dilution antimicrobial susceptibility tests for bacteria that grow aerobically: tentative standard. 2nd ed. NCCLS document No. M7-T2. Villanova, Pa.: The Committee, 1988. 7. Jackson MA, Shelton S, Nelson JD, McCracken GH Jr. Relatively penicillin-resistantpneumococcalinfections in pediatric patients. Pediatr Infect Dis J 1984;3:129-32. 8. Pallares R, Gudiol F, Linares J, et al. Risk factors and response to antibiotic therapy in adults with bacteremic pneumonia caused by penicillin-resistant pneumococci. N Engl J Med 1987;317:18-22. 9. Michel J, Dickman D, Greenberg A, Bergner-Rabinowitz S. Serotype distribution of penicillin-resistant pneumoeocciand their susceptibilitiesto sevenantimicrobial agents. Antimicrob Agents Chemother 1983;23:397-401. 10. Gray BM, Dillon HC Jr. Serotypes of Streptococcuspneumoniae causing disease. J Infect Dis 1979;40:979-83. 11. Gray BM, Dillon HC Jr. Clinical and epidemiologicstudies of pneumococcal infection in children. Pediatr Infect Dis J 1986;5:201-7.

Articular symptoms in hypermobile schoolchildren: A prospective study Abraham

G e d a l i a , MD, a n d J o s e p h Press, MD

From the Pediatric Rheumatotogy Unit, Division of Pediatrics, Soroka Medical Center, Ben Gurion University of the Negev, Beer Sheva, Israel

The first report of an association between hypermobility of the joints and rheumatologic symptoms was by Sutro, l who described 13 young adults with effusions and pain in hypermobile knees and ankles. Kirk et al. 2 later defined the "hypermobile syndrome" in a group of patients with both hypermobility of the joints and musculoskeletal complaints; in the absence of demonstrable systemic rheumatologic disease, the symptoms were attributed to articular hypermobility. In a previous study, we found a high frequency (66%) of joint hypermobility in a group of patients with what we termed "juvenile episodic arthralgia/arthritis" in whom recurrent episodes of joint pain of unknown origin occurred. 3 In this study, we have prospectively observed norSubmitted for publication April 23, 1991; accepted July 5, 1991. Reprint requests: A. Gedalia, MD, Divisionof Pediatrics, Soroka Medical Center, PO Box 151, Beer Sheva, Israel. 9/22/32204

real schoolchildren and those with hypermobility for 1 year to determine the incidence of articular symptoms. METHODS

All 429 children aged 6 through 14 years in one of the public schools in Beer Sheva, Israel, were examined for joint hypermobility. Although quantitative measurements in the assessment of joint hypermobility have been described, the criteria devised by Carter and Sweetnam, 4 as modified by Bird et al., 5 were used. The classic features of hypermobility in children are the ability to perform passively the following maneuvers: (1) hyperextension of the fingers so that they are parallel to the forearms; (2) apposition of the thumbs to the surface of the forearms; (3) hyperextension of the elbows > 10 degrees; (4) hyperextension of the knees > 10 degrees; and (5) flexion of the trunk with knees straight and the palms of the hands touching the floor. Any subject who was able to perform three or more of these maneuvers was considered to have joint hypermobility.

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Table I. Joint hypermobility in normal Israeli schoolchildren Girls

Boys

Hypermobility A g e (yr)

6-10 11-14 TOTAL

Hypermobility

n

n

%

n

n

%

119 84 203

30* 6 36t

25 7 18

131 95 226

9 8 17

7 8 8

*Higher frequencyof hypermobilityis foundamongyoungergirls than amongchildrenin oldergroup (chi-square= 11.0;p <0.005). ~Differencein hypermobilityin girls (comparedwith boys)is statisticallysignificant(chi-square= 10.2;p <0,005).

Table II. Articular symptoms in children with joint hypermobility and children without joint hypermobility Hypermobility

Nonhypermobility

Symptoms

Girls Boys TOTAL

Symptoms

n

n

%

n

n

%

34 19 53

14 7 21"

41 37 40

32 20 52

6 3 9

18 15 17

*Statisticallyhigher prevalenceof articularsymptomsamongthe schoolchildrenwith hypermobility(chi-square= 6.4;p <0.02).

We prospectively followed two groups of age- and sex-matched children, the "hypermobile" group and a "nonhypermobile" control group that was chosen randomly. Episodes of arthralgia or arthritis in both groups were recorded by the parents for a period of l year. The data were gathered by a questionnaire, which the parents were asked to complete each month. Arthralgia was defined as joint pain at rest, on motion, or in both situations. Arthritis was defined as a joint with either swelling or pain on motion or with tenderness and limitation of motion.6 The following questions were included in the questionnaire: 1. How many episodes of joint pain did your child have during the past year? 2. Besides pain, was there any limitation of motion or swelling? 3. How long was it painful? 4. Which of the following joints were involved? Knee/ ankle/hip/other? 5. When did the painful episodes take place? In the morning/daytime/evening/after exercise? 6. Did you seek medical consultation for your child's joint pain? 7. How many days of school has your child missed this year because of joint pain?

years; 8%). A higher frequency of joint hypermobility was found among girls (18%) than among boys (8%; chisquare = 10.2; p <0.005). Hypermobility in the girls was found predominantly among the younger age group (ehisquare = 11.0; p <0.005), as shown in Table I. We prospectively followed (through questionnaires completed monthly by parents) for 1 year the hypermobile group of 53 schoolchildren and a randomly chosen control group of 52 schoolchildren. The mean age of the hypermobile group was 9 years and that of the nonhypermobilegroup was 9.1 years. No episodes of arthritis were recorded in either group. Recurrent arthralgia (two or more episodes) occurred at a higher frequency, 21 (40%) of 53, in the hypermobile group than in the nonhypermobile group, I 1 (17%) of 52 (chi-square = 6.4; p <0.02; Table II). The knees were involved in all children with symptoms in the hypermobile group and in seven of the children with symptoms in the nonhypermobile group. In more than half of the cases of joint pain in both groups (13 and 5, respectively), the episodes occurred in the evenings. Five of the children with hypermobility and only one of the children without hypermobility had at least one episode of joint pain after exercise. No significant difference in school absences was noted between the groups.

RESULTS

DISCUSSION

Of 429 normal schoolchildren, 53 (12%) had joint hypermobility. There was a higher frequency (15%) of joint hypermobility in the younger age group (ages 6 through 10 years) than in the older age group (aged 11 through 14

The frequency of joint hypermobility among Israeli schoolchildren is similar to that found in a previous study of children in Houston, which was done by the same examiner with the same criteria. 3 This frequency of hypermobil-

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ity is very close to the 7% to 12% previously reported by others who used the same scoring system], 8 This study revealed that joint hypermobility was significantly more prevalent among girls than among boys, as has been noted by others)' 9, ~0 In concurrence with our previous study 3 and studies by others, 9, 11 we found that joint hypermobility seems to decline with age. In this study, such a decline was seen predominantly among girls. Since the observations by Kirk et al., 2 who defined the "hypermobility syndrome," it has been noted that joint hypermobility predisposes persons to arthralgias, joint effusion, recurrent joint dislocation, and ligament rupture, 1, 7, 12 but no controlled studies have been done to suggest a real association between joint hypermobility and articular complaints. Our data demonstrate that children with hypermobility are more likely than normal children to have recurrent episodes of arthralgia. A similar study done by Arroyo et al. 13 indicated that there was a nonsignificant trend toward greater frequency of articular complaints among children with hypermobility than among age- and gendermatched control subjects without hypermobility; the sample sizes were small, however, introducing a strong possibility of type II error. It is not understood why some of the children without hypermobility may have recurrent short episodes of joint complaints. The child with symptomatic hypermobility syndrome is usually seen with recurrent short episodes of pain, swelling, or both in one or two joints. The knee is the joint most commonly involved. The hips, elbows, and ankles, in that order, seem to be next in frequency of involvement. 3, 14 Sterile joint effusions have been reported by Sutro 1 in as many as one fourth of these children with hypermobility. There is frequently a history of pain in selected joints after excessive activity or misuse. Fifty percent of the children with hypermobility syndrome who were followed in a pediatric rheumatology clinic had pain in the late afternoon or evening15; morning stiffness was unusual. Some rheumatologists have noticed that a growth spurt occurred just before the onset of symptoms. 16 Close relatives with a history of joint hypermobility were found in nearly half of the patients in one study. 3 This observation was also made by Beighton and Horan, 17 who, after reviewing the clinical features and family history of subjects with hypermobility, suggested an autosomal dominant mode of inheritance. Arthralgia secondary to joint hypermobility is a diagnosis of exclusion; serious forms of joint disease, such as juvenile rheumatoid arthritis and septic arthritis, should be ruled out before this benign diagnosis is considered. No specific laboratory or diagnostic test is available to confirm the diagnosis of hypermobility syndrome. Complete blood

The Journal of Pediatrics December 1991

cell count, erythrocyte sedimentation rate, results of assays for rheumatoid factor, antinuclear antibodies, and quantitative immunoglobulins, complement component (C3, C4, and CHs0) levels, and findings of slit-lamp examination performed in many children with the hypermobility syndrome were within normal limits. 3 It should be carefully explained to the patients and their parents that the hypermobility syndrome is a benign condition. Such an explanation will provide reassurance, helping to reduce patient and parental anxiety and minimizing overinvestigation and overuse of medications. The longterm prognosis for hypermobility syndrome remains to be determined by further prospective studies.

REFERENCES

1. Sutro CJ. Hypermobility of bones due to overlengthened capsular and ligamentous tissues. Surgery 1947;21:67-76. 2. Kirk JA, Ansell BM, Bywaters EGL. The hypermobility syndrome: musculoskeletal complaints associated with generalized joint hypermobility. Ann Rheum Dis 1967;26:419-25. 3. Gedalia A, Person DA, Brewer EJ, Giannini EH. Hypermobility of the joint in juvenile episodic arthritis/arthralgia. J PEDIATR 1985;107:873-6. 4. Carter C, Sweetnam R. Familial joint laxity and recurrent dislocation of the patella. J Bone Joint Surg [Br] 1958;40:664-7. 5. Bird HA, Brodie DA, Wright V. Quantification of joint laxity. Rheumatol Rehabil 1979;18:161-4. 6. Giannini EH, Brewer EJ. Standard methodology for segment I, II, and III pediatric rheumatology study group studies. J Rheumatol 1982;9:114-22. 7. Carter C, Wilkinson J. Persistent joint laxity and congenital dislocation of the hip. J Bone Joint Surg [Br] 1964;46:40-5. 8. Silverman S, Constine L, Harvey W, et al. Survey of joint mobility and in vivo skin elasticity in London schoolchildren. Ann Rheum Dis 1975;34:177-80~ 9. Beighton P, Solomon L, Soskolne CL. Articular mobility in an African population. Ann Rheum Dis 1973;32:413-8. 10. Jessee EF, Owen DS, Sagar KB. The benign hypermobile joint syndrome. Arthritis Rheum 1980;23:1053-6. 11. Wynne-Davies R. Acetabular dysplasia and familial joint laxity: two etiological factors in congenital dislocation of the hip. J Bone Surg [Br] 1970;52:704-16. 12. Nicholas AJ. Injuries to knee ligaments: relationship to looseness and tightness in football players. JAMA 1970;212:701-16. 13. Arroyo IL, Brewer EJ, Giannini EJ. Arthritis/arthralgia and hypermobility of the joints in schoolchildren. J Rheumatol 1988;15:978-80. 14. Biro F, Gewanter HL, Baum J. The bypermobility syndrome. Pediatrics 1983;72:701-5. 15. Gedalia A. The hypermobility syndrome in childhood. Faro Physician [Israel] 1988;16:164-9. 16. Lewkonia RM. Hypermobility of joints. Arch Dis Child 1987;62:1-2. 17. Beighton PH, Horan FTo Dominant inheritance in familial generalised articular hypermobility. J Bone Joint Surg [Br] 1970;52:145-7.