Abstract
Objectives:
To test the hypothesis that the prevalence of joint hypermobility (is greater in children with irritable bowel syndrome (IBS) and functional abdominal pain (FAP) than in healthy control children and is related to gastrointestinal symptoms and psychosocial distress (anxiety, depression, somatization).
Study design:
Children (IBS, n=109; FAP, n=31; HC, n=69), 7–12 years of age completed prospective 2-week pain and stooling diaries and child- and parent-report measures of anxiety, depression, and somatization. JH was determined using Beighton criteria (score ≥ 4 or 6). We also examined possible relationships between Beighton score, race, body mass index, gastrointestinal symptoms, and psychosocial distress.
Results:
Beighton scores were similar between groups, as was the proportion with JH. Scores were higher in girls (3.1 ± 2.4) than boys (2.3 ± 1.8; P=0.004) and decreased with age (P < .001, r=−0.25). Race and body mass index did not impact JH prevalence. Beighton scores were not related to abdominal pain or stooling characteristics. Participants with a score ≥4 and ≥ 6 had greater somatization and depression by child report (P=0.017 and P=0.048, respectively). No association was seen for anxiety. There was no significant association between JH and psychosocial distress measures per parent report.
Conclusions:
Contrary to adult literature, the prevalence of JH does not differ among children with IBS, FAP, or HC. The presence or severity of JH does not correlate with abdominal pain or stooling characteristics. Somatization and depression by child report appear to have a relationship with JH.
Keywords: Irritable bowel syndrome, functional abdominal pain, gastrointestinal, functional abdominal pain disorder, joint hypermobility
Recurrent abdominal pain is a common complaint affecting up to 15–20% of school age children and adults world-wide.(1–3) The majority are considered to have a functional abdominal pain disorder: irritable bowel syndrome (IBS), functional abdominal pain (FAP), or functional dyspepsia.(4) FAP is characterized by intermittent abdominal pain; when associated with an altered stooling pattern, it is denoted as IBS. (4)
Joint hypermobility , defined as greater than normal joint laxity across multiple joints as measured by the Beighton score.,(5–7) may exist on its own or be part of the diagnostic criteria for JH syndromes such as Marfan and Ehlers-Danlos syndromes. (8, 9) Studies suggest that children are more hypermobile than adults and females more hypermobile than males.(10, 11) Similarly, data suggests that JH may be related to body mass index, and race and/or ethnicity, with those of Asian and African descent more hypermobile than Caucasians.(5, 11, 12)
Studies in adults suggest the prevalence of JH is greater in patients with IBS and/or in patients with gastrointestinal (GI) symptoms than in healthy controls.(13–15) Conflicting data exist on the prevalence of JH in children with IBS.(16, 17) What is known is that children in general with JH experience greater pain intensity compared with healthy controls and it is thought that JH may contribute to GI symptoms in adult IBS.(14, 15, 18) Interpretation of these studies is limited by factors such as their retrospective nature, the use of questionnaires to identify JH and the type of functional GI disorder, and lack of differentiation between functional GI disorder subtypes.(13–17)
Limited data suggest a potential relationship between JH and psychosocial distress in adults and children.(19–21) Adults and children with functional GI disorders have increased psychosocial distress compared with healthy individuals.(22–24) Whether there is a relationship between JH and psychosocial distress in children with IBS or FAP has not been evaluated to our knowledge.
Our aim was to compare the prevalence of JH in children with IBS and FAP with that in healthy control children (HC) using direct measurement of joint mobility. We also sought to evaluate whether JH was associated with psychosocial distress and/or prospectively collected GI symptoms. We hypothesized that JH prevalence is greater in children with IBS and FAP than in HC children and that JH is related to GI symptoms and psychosocial distress (anxiety, depression, somatization).
METHODS
Children (7–12 years of age) were recruited from the Texas Children’s Hospital healthcare network based in Houston, TX. Informed consent was obtained from parents, assent was obtained from children, and the study approved by the Baylor College of Medicine Institutional Review Board.
Design
Medical charts were reviewed by trained research coordinators using ICD-9 codes for abdominal pain and IBS to identify children with IBS and FAP, and well child visit codes (eg, immunizations, school check-ups) to identify HC. Participants then were screened via telephone and using a modified (shortened) pediatric Rome III questionnaire.(25, 26) Children were excluded if chart review or screening revealed a significant chronic medical condition (e.g., diabetes, cystic fibrosis), chronic vomiting, unexplained weight loss, hematochezia, major GI tract surgery, significant developmental delay, or organic GI disorder.
Participants were scheduled for a home visit or to come to the Children’s Nutrition Research Center (depending on family preference). At that visit they completed psychosocial questionnaires (see below) and were assessed for the presence of JH (see below). Weight and height were obtained.
Families then received detailed instructions on how to complete a validated 2-week pain and stooling diary.(27, 28) Using the 2-week pain and stooling diary, participants were rigorously classified as IBS, FAP, or HC using a previously validated algorithm.(26) IBS was subtyped as previously described.(29) Using the Bristol Stool Form scale, constipation was defined as a rating of 1–2, normal as 3–5, and diarrhea as 6–7.(28)
Psychosocial Distress Measures
The Behavioral Assessment System for Children, Second Edition (BASC-2) measures child emotional and behavior problems and competence using age and respondent-dependent measures.(30) The instrument is well-validated and in widespread use with versions for child self-report and parent-report of child behaviors. Scale and composite scores are expressed in t-scores. For this study we used the Anxiety and Depression subscales for both child self-report and parent (mother)-report of the child. A T-score ≥ 60 is considered at risk and a T-score ≥ 70 is considered clinically significant.
Somatization was measured using the Children’s Somatization Inventory (CSI). The frequency and severity of 24 somatic symptoms taken from DSM-III-R criteria for somatization disorder and the somatization factor of the Hopkins Symptoms Checklist are presented;(31) both the child (CSI) and mother (P-CSI) rate on a 5-point scale (0- “not at all” to 4- “a whole lot”) the extent to which the physical symptoms have “bothered” the child during the last 2 weeks. Total scores are calculated.(32)
Testing for Joint Hypermobility
The research coordinators were trained in the use of the goniometer and determination of joint laxity as defined by Beighton.(7, 33) The Beighton score ranges from 0 (none) to 9 (greatest hypermobility) and has been validated for use in children.(7, 8, 33) Debate continues regarding the appropriate cutoff to define JH.(6, 33) A recent study in children used the traditional cutoff of ≥ 4 and also ≥ 6.(5) Another pediatric study also suggested a less strict cutoff of ≥ 4.(33) A r systematic review of adult and pediatric data suggested a cutoff of ≥ 6 for children.(34) Thus, we utilized both cutoff values.
Sample Size
The necessary sample size was determined based on a previous study in children with functional abdominal pain disorders and an adult study of patients with IBS.(14, 16) The studied sample size was adequate to detect a difference in Beighton score between children with IBS/FAP and HC with P<0.05 and power = 0.95 and for detecting a difference in the proportion of children with/without JH (cutoff of 6) with P<0.05 and power = 0.95.
Statistical Analyses
Data presented in Tables are expressed as mean ± SD. Differences in demographics were tested using Student t-test. Proportions were tested using Chi-square analysis. Beighton scores were not normally distributed, so they were log transformed. The raw data are shown. To evaluate the possible effects of age, sex, race, and ethnicity on the Beighton score, general linear modeling was used. P<0.05 was considered significant.
RESULTS
Participants
Sex, age, body mass index, race, and ethnicity were comparable among the groups (Table I). The majority of children with IBS were categorized as constipation subtype and unsubtyped (IBS-constipation, n=47; IBS-diarrhea, n=10; IBS-mixed, n=6; IBS-unsubtyped, n=46).
Table 1.
Demographics of Subjects
| Group | ||||
|---|---|---|---|---|
| IBS | FAP | Control | ||
| n=109 | n=31 | n=69 | P Value | |
| Sex | ||||
| Male | 41 | 13 | 34 | 0.31 |
| Female | 68 | 18 | 35 | |
| Age (yrs.)* | 9.4 ± 1.4 | 9.4 ± 1.6 | 9.6 ± 1.5 | 0.57 |
| Race | 0.58 | |||
| White | 78 | 20 | 50 | |
| Black | 18 | 6 | 12 | |
| Asian | 8 | 0 | 0 | |
| Native | 0 | 1 | 0 | |
| American/Hawaiian | ||||
| Mixed or unidentified | 5 | 4 | 7 | |
| Ethnicity | 0.90 | |||
| Non-Hispanic | 79 | 22 | 50 | |
| Hispanic | 30 | 8 | 19 | |
| Unidentified | 0 | 1 | 0 |
Mean ± SD
Joint Hypermobility and Sex, Age, Race, and BMI
For the total sample across groups, Beighton scores were higher in girls than in boys (3.1 ± 2.4 vs 2.3 ± 1.8, respectively; P=0.008) (Table 2). The difference between boys and girls in the IBS group was significant (P=0.023). The mean Beighton score was numerically higher for girls in the FAP group but the difference did not reach significance (P=0.12). There was no difference between sexes in scores for the HC group. Older children had lower Beighton scores than younger children (Figure). There were no differences in the prevalence of JH using either a cutoff value of ≥ 4 or ≥ 6 across racial or ethnic groups or related to BMI (data not shown).
Table 2.
Beighton Scores
| Group | Total Sample |
||||||
|---|---|---|---|---|---|---|---|
| IBS | FAP | HC | IBS vs FAP | IBS vs HC | FAP vs HC | ||
| n=109 | n=31 | n=69 | n=209 | ||||
| By Sex | |||||||
| Boys | 2.2 ± 1.7* | 2.5 ± 2.0 | 2.2 ± 1.8 | 2.3 ± 1.8 | |||
| Girls | 3.1 ± 2.4 | 3.6 ± 2.4 | 3.1 ± 2.4 | 3.1 ± 2.4 | |||
| P Value# | 0.023 | 0.12 | 0.57 | 0.008 | |||
| Cohen’s d | 0.5 | 0.6 | 0.15 | 0.4 | |||
| By Group | 2.7 ± 2.2 | 3.1 ± 2.3 | 2.6 ± 2.1 | P=0.7 | P=0.43 | P=0.98 | P=0.45 |
Mean ± SD
Boys versus Girls
Figure.
Relationship between Beighton score and age. Beighton score decreased with increasing age.
Joint Hypermobility in IBS, FAP, HC Groups
Beighton scores were similar among the groups and this lack of difference remained even taking into account age and sex (P=0.6). No significant differences emerged for comparisons of Beighton scores between groups (Table 2).
The proportion of children with JH as defined by Beighton scores ≥ 4 did not differ between the groups: IBS=35%, FAP=36%, HC=36%; P=0.98. Proportions also were similar when using a Beighton score cutoff of ≥ 6: IBS=12%, FAP=13%, HC=9%; P=0.74.
Abdominal Pain and Stooling Characteristics
As expected, the number of abdominal pain episodes and pain severity over the 2-week period of the diary were greater in the IBS and FAP groups compared with those in the HC group. No differences in pain measures between IBS and FAP groups were found (Table 3; available at www.jpeds.com).
Table 3.
Online Abdominal Pain and Stooling Characteristics
| Group | P Values | |||||
|---|---|---|---|---|---|---|
| IBS | FAP | Control | IBS vs FAP | IBS vs HC | FAP vs HC | |
| n=109* | n=40 | n=69 | ||||
| Abdominal Pain | ||||||
| Number of Episodes | 12.8 ± 9.1# | 11.4 ± 11.1 | 0.4 ± 0.6 | 0.52 | <0.001 | <0.001 |
| Severity (0–10) | 3.1 ± 1.3 | 3.0 ± 1.3 | 1.2 ± 2.0 | 0.68 | <0.001 | <0.001 |
| Stooling | ||||||
| Number of Stools | 13.2 ± 4.8 | 10.7 ± 4.8 | 11.8 ± 4.4 | 0.012 | 0.052 | 0.25 |
| % Constipation | 27.4 ± 22.9 | 21.6 ± 25.3 | 23.7 ± 26.3 | 0.26 | 0.35 | 0.70 |
| % Normal | ||||||
| % Diarrhea | 10.4 ± 15.8 | 8.0 ± 20.9 | 2.9 ± 6.6 | 0.56 | <0.001 | 0.19 |
Complete diary data not obtained on all participants
Mean ± SD
The number of stools passed was greater in the IBS vs FAP and HC groups with no differences between FAP and HC. The percent of stools rated as diarrhea was greater in the IBS vs HC group but not vs the FAP group.
Beighton Scores and Abdominal Pain and Stooling Characteristics
For all participants across groups, Beighton scores did not correlate with the number of abdominal pain episodes or pain severity (Table 4; available at www.jpeds.com). This also was the case when evaluating just the IBS and FAP groups individually (data not shown). There was no difference in the number of abdominal pain episodes between those children with a Beighton score ≥ 4 versus < 4 or those with scores of ≥ 6 versus < 6. Similarly, abdominal pain severity did not differ between those children with a Beighton score ≥ 4 versus < 4 or those with scores of ≥ 6 versus < 6.
Table 4.
Online Beighton Scores versus Abdominal Pain and Stooling Characteristics
| Parameter | Abdominal pain | Number of Bowel Movements | Proportion of Stools (%) | |||
|---|---|---|---|---|---|---|
| Number of episodes | Severity (0–10) | Constipation | Normal | Diarrhea | ||
| Beighton score | P=0.39 | P=0.70 | P=0.12 | P=0.12 | P=0.35 | P=0.4 |
| All Participants* | r=0.0 | r=0.0 | r=0.1 | 0.1 | r=0.0 | r=0.0 |
| Beighton score by cutoff | ||||||
| <4 (n=139)# | 8.7 ± 9.7 | 2.4 ± 1.7 | 12.5 ± 5.1 | 23.8 ± 25.3 | 68.4 ± 26.5 | 7.8 ± 16.0 |
| ≥ 4 (n=79) | 8.7 ± 9.8 | 2.5 ± 1.9 | 12.0 ± 4.0 | 27.2 ± 23.0 | 65.8 ± 25.2 | 7.0 ± 12.6 |
| P value | 0.97 | 0.76 | 0.39 | 0.33 | 0.48 | 0.70 |
| Cohen’s d | 0.0 | 0.0 | 0.1 | 0.1 | 0.1 | 0.1 |
| <6 (n=192) | 8.4 ± 9.5 | 2.5 ± 1.8 | 12.4 ± 4.8 | 24.4 ± 24.4 | 67.8 ± 26.2 | 7.7 ± 15.1 |
| ≥ 6 (n=26) | 11.1 ± 11.3 | 2.4 ± 1.9 | 11.7 ± 3.8 | 29.4 ± 24.8 | 64.6 ± 24.8 | 6.0 ± 122.9 |
| P value | 0.18 | 0.72 | 0.47 | 0.33 | 0.55 | 0.59 |
| Cohen’s d | 0.3 | 0.1 | 0.2 | 0.2 | 0.1 | 0.1 |
Correlation
Complete diary data not obtained on all participants
Beighton scores did not correlate with the number of bowel movements nor with the proportion of stools rated as constipated, normal, or diarrheal. Again, this also was the case when evaluating the IBS and FAP groups individually (data not shown). There were no differences in stooling characteristics (i.e., number of bowel movements, stools rated constipated, normal, or diarrheal) between those children with a Beighton score ≥ 4 versus < 4 or those with scores of ≥ 6 versus < 6.
Psychosocial Distress
As expected, anxiety, depression, and anxiety by both child and mother report were greater in the IBS and FAP groups compared with HC (Table 5; available at www.jpeds.com). Only somatization (by child report) differed between the IBS and FAP group, being greater in the former (P=0.025). The mean scores for anxiety and depression were below the mean of a normative sample.(30) The somatization scale has no normative range.(31)
Table 5.
Online Psychosocial Distress Measures
| Group | P Values | |||||
|---|---|---|---|---|---|---|
| IBS | FAP | Control | IBS vs FAP | IBS vs HC | FAP vs HC | |
| Child Report | (n=106) | (n=30) | (n=64) | |||
| Anxiety | 51 ± 11* | 48 ± 11 | 44 ± 8 | 0.31 | <0.001 | 0.02 |
| Depression | 48 ± 9 | 46 ± 7 | 44 ± 5 | 0.31 | <0.001 | 0.07 |
| Somatization | 27 ± 19 | 20 ± 13 | 12 ± 13 | 0.025 | <0.001 | 0.001 |
| Parent Report | (n=109) | (n=30) | (n=68) | |||
| Anxiety | 52 ± 12 | 54 ± 11 | 46 ± 9 | 0.33 | <0.001 | 0.001 |
| Depression | 50 ± 10 | 50 ± 8 | 45 ± 9 | 0.81 | <0.001 | 0.015 |
| Somatization | 15 ± 11 | 17 ± 11 | 3 ± 6 | 0.48 | <0.001 | <0.001 |
Mean ± SD
Beighton Scores and Psychosocial Distress
Beighton scores did not correlate with child- or mother-report of anxiety, depression, or somatization (Table 6). However, those with a Beighton score ≥ 4 had greater scores for child report of somatization than those with a score < 4. Similarly, those with a Beighton score ≥ 6 had greater scores for child report of depression than those with a score < 6. Child report of anxiety did not differ by Beighton score cutoff of ≥ 4 or 6. Mother report of anxiety, depression, or somatization did not differ by Beighton score cutoff of ≥ 4 or 6.
Table 6.
Beighton Scores and Psychosocial Distress
| Parameter | Child Report | Mother Report | ||||
|---|---|---|---|---|---|---|
| Anxiety | Depression | Somatization | Anxiety | Depression | Somatization | |
| Beighton score | ||||||
| All Participants* | ||||||
| P value | 0.17 | 0.15 | 0.15 | 0.94 | 0.24 | 0.24 |
| r value | 0.07 | 0.07 | 0.07 | 0.0 | 0.04 | 0.0 |
| Beighton score by cutoff | ||||||
| < 4 (n=167)# | 48.5 ± 10.2 | 46.4 ± 7.5 | 19.2 ± 15.6 | 50.9 ± 11.8 | 48.0 ± 8.9 | 12.4 ± 12.6 |
| ≥ 4 (n=86) | 50.2 ± 11.0 | 47.1 ± 7.9 | 24.6 ± 19.2 | 50.4 ± 11.6 | 49.2 ± 10.4 | 11.2 ± 10.5 |
| P value | 0.16 | 0.51 | 0.017 | 0.77 | 0.31 | 0.45 |
| Cohen’s d | 0.2 | 0.1 | 0.3 | 0.0 | 0.1 | 0.1 |
| < 6 (n=224) | 48.7 ± 10.4 | 46.3 ± 7.5 | 20.5 ± 16.8 | 50.6 ± 11.8 | 48.2 ± 9.3 | 12.0 ± 12.0 |
| ≥ 6 (n=29) | 52.4 ± 10.8 | 49.2 ± 8.2 | 25.2 ± 18.6 | 51.5 ± 11.5 | 49.9 ± 10.2 | 11.6 ± 11.2 |
| P value | 0.07 | 0.048 | 0.16 | 0.70 | 0.36 | 0.84 |
| Cohen’s d | 0.4 | 0.4 | 0.3 | 0.0 | 0.2 | 0.0 |
Correlation
Complete psychosocial measures not obtained on all participants
DISCUSSION
We found no differences between children with IBS, FAP, and HC in Beighton scores (Table 2) nor in the prevalence of JH using cutoffs of ≥ 4 or ≥ 6. We did find that Beighton scores were related to sex and age, even within the narrow age range that we studied (Table 2 and Figure). Thus, as suggested by this and previous research, age and sex need to be considered when assessing the potential presence of hypermobility.(10, 11) We also found that child report of somatization was greater in those children with a Beighton score of ≥ 4 and child report of depression was greater in those with a Beighton score of ≥ 6 (Table 6).
There has been interest in the possible association between JH and functional abdominal pain disorders given that patients in general with JH are prone to experience greater GI and other pain (e.g., musculoskeletal).(14, 15, 18) Indeed, it is well recognized that patients with JH syndromes (e.g., Ehlers-Danlos syndrome, Marfan syndrome) commonly have GI symptoms (e.g., abdominal pain, acid reflux, dyspepsia).(9, 35) Hence, it has been suggested that patients with functional abdominal pain disorders be screened for JH.(14–16)
Our results converge with those of Saps et al in which a school-based study found no difference in hypermobility frequency among children with a functional abdominal pain disorder (n=136) diagnosed by questionnaire versus HC (n=136).(17) Our results extend their findings into functional abdominal pain disorder subtypes, as we used prospective diaries rather than questionnaires to identify children as to whether they had IBS or FAP or were HC given the greater reliability of diaries versus questionnaires for this purpose.(36, 37) Prospective diaries also allowed us to examine the potential relationships between JH and abdominal pain, stooling characteristics, and psychosocial distress which previously had been understudied.
The prevalence of JH in the HC in our study (36% for scores ≥ 4) is similar to that reported previously in healthy children of similar ages worldwide.(5, 10–12, 20) Similarly, the prevalence of JH in HC using a cutoff score of ≥ 6 (9%) is comparable with previous reports from Australia.(5, 11)
In a retrospective chart review of children and young adults with a functional GI disorder, Kovacic et al reported that of the 45 subjects who had a Beighton score recorded, 56% met criteria for JH.(16) They suggested that the prevalence of JH was greater in a specialty practice than in the general population, though our results do not suggest this difference.(16) Their results may have been biased, however, given that only those with a suspicion of JH may have been tested.(16)
A small number of studies in adults suggest there may be a relationship between JH and functional GI pain disorders. Zarate et al reported that patients referred to a neurogastroenterology clinic completed a questionnaire to assess the possible presence of JH.(13) Those with evidence of JH had a greater prevalence of gastroesophageal reflux and bloating symptoms than those without questionnaire evidence of hypermobility.(13) Fikree at al performed Beighton testing in consecutive new patients attending a gastroenterology clinic.(14) Patients completed GI symptom, psychosocial, and quality of life questionnaires.(14) Adjusting for age and sex, heartburn, water brash, and postprandial fullness were more common in the JH group.(14) In a later study, the same group compared the prevalence of JH based on Beighton score in patients with functional GI disorders as defined by a questionnaire, with that in patients with organic GI disorders (e.g., inflammatory bowel disease, erosive reflux disease).(15) After adjusting for age and sex there was a relationship between the presence of JH and the diagnosis of a functional GI disorder, but only the specific diagnosis of functional gastroduodenal disorders was significantly related.(15) In the organic group a high prevalence of JH was found in those with reflux disease.(15) The discrepancy between our results and the adult data may relate to differences in age and/or in the underlying pathogenesis of functional abdominal pain disorders in children versus adults.
To our knowledge there are no data on the potential relationships between Beighton scores, the presence of JH, and the severity of GI symptoms (i.e., abdominal pain) and stooling characteristics. This is likely due to the fact that previous studies have relied upon retrospective reporting in which symptom severity was not assessed. Our prospective diary data suggest no relationship between Beighton scores and GI symptoms.
There are limited data on the potential relationship between psychosocial distress and JH. In one adult study no difference was seen in the prevalence of anxiety, depression, or somatization in those with/without JH.(14) In contrast, a systematic review and meta-analysis of 14 studies (n=3957) published the same year suggested that individuals with JH had a higher prevalence of anxiety and higher anxiety scores compared with those without JH.(19) Similarly, the prevalence of depression also was greater, but the severity was comparable between those with/without JH.(19) Limited data are available for children. Ezpeleta et al reported on children 9 years of age who were part of a longitudinal study of behavior problems (n=336); those with JH had more severe anxiety symptoms than in those with less symptoms.(20) In another study JH prevalence in 160 children recruited from a mental health clinic was 22%, with anxiety scores greater in those with JH.(21) A subgroup had JH syndrome (e.g., Ehlers-Danlos syndrome), and in this group somatization (based on the Child Behavior Checklist) was greater than in those with JH without a syndrome.(21)
In our study anxiety, depression, or somatization were not significantly correlated with Beighton score. However, children with a Beighton score ≥ 4 had higher scores for child report of somatization and those with a Beighton score ≥ 6 had higher scores for child report of depression (Table 6). We previously have shown the importance of somatization in the expression of abdominal pain in children with functional GI disorders.(38, 39) It has been suggested that somatization may be magnified in individuals with JH syndrome because of disordered autonomic nervous system function (e.g., postural tachycardia syndrome) and a mismatch between cognitions and actual body state.(40) Our results suggest this may be the case in children with JH occurring apart from JH syndrome, though this requires confirmation in a future study as does the finding related to depression.
With respect to study limitations, the results may only be representative of our center and would benefit from replication, though our inclusion of children from both tertiary and primary care should improve generalizability of the results. We evaluated for the presence of JH using the Beighton criteria but we did not inquire about other symptoms (e.g., arthralgia) that would have allowed assessment for JH syndrome (JH associated with musculoskeletal symptoms in more than four joints, including pain over ≥ 12 weeks).(6, 8)
Our study extends the literature through utilization of improved methodology (prospective validated diary, subtyping of the functional pain disorder, and objective measurement of JH); examination of the potential relationship between JH and abdominal pain and stooling characteristics; and appraisal of the potential relationship between JH and psychosocial distress using validated questionnaires. Use of the diaries is more reliable than retrospective questionnaires, allowing us to confidently separate IBS, FAP, and HC groups in contrast to previous studies in which different subgroups of functional GI disorders were not identified or only identified by recall questionnaires.(13–17, 36) Further, use of a goniometer to determine joint laxity as defined by Beighton is a more robust method than questionnaire assessment.(34)
In summary, we found no evidence that children with IBS or FAP have JH greater than that in HC. The presence or severity of JH do not appear related to abdominal pain or stooling characteristics in children with IBS or FAP. There was an association between the severity of JH and somatization and depression by child report. Future studies evaluating the severity of JH in children should include measures of somatization and depression to better understand these possible associations.
Although Beighton scores were similar among children with IBS, FAP, and HC and the proportion of children with JH did not differ among the groups, it should be noted that in an individual patient JH may be present and be accompanied by associated musculoskeletal symptoms that may contribute to the extraintestinal pain symptoms and disability often found in children with IBS and FAP. Similarly, it should be kept in mind that additional studies are needed to determine if older children with IBS and FAP (i.e., outside of the age group in the current study) differ from HC in terms of the presence of JH.
Acknowledgments
supported by the National Institutes of Health (R01 NR013497), the Daffy’s Foundation, the USDA/ARS under Cooperative Agreement (6250-51000-043 and P30 DK56338), which funds the Texas Medical Center Digestive Disease Center. The content is solely the responsibility of the authors and does not necessarily represent the official views of the National Institutes of Health. This work is a publication of the USDA/ARS Children’s Nutrition Research Center, Department of Pediatrics, Baylor College of Medicine and Texas Children’s Hospital. The contents do not necessarily reflect the views or policies of the USDA, nor does mention of trade names, commercial products, or organizations imply endorsement by the US Government. The authors declare no conflicts of interest.
Footnotes
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