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Indian Pediatr 2017;54:946-948 |
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Joint Involvement in
Children with Celiac Disease
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Kapil Garg, Priyanka Agarwal, R K Gupta and S
Sitaraman
From the Department of Pediatrics, SMS Medical
college, Jaipur, India.
Correspondence to: Dr Kapil Garg, 5 JHA 22, Jawahar
nagar, Jaipur 302 004, India.
Email: [email protected]
Received: August 01, 2016;
Initial Review: November 05, 2016;
Accepted: July 17, 2017.
Published online: August 24, 2017.
PII:S097475591600082
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Objective: To determine early joint involvement as detected by
ultrasonography in children with newly diagnosed celiac disease, and in
children with celiac disease on gluten-free diet for more than 6 months.
Methods: Cross-sectional comparative study evaluating joint
abnormalities by ultrasonography. Results: Ultrasonography showed
abnormalities in 19 out of 60 (31.7%) children with newly diagnosed
celiac disease as compared to 2 (3.3%) out of 60 in those on a
gluten-free diet for more than 6 months. Conclusion: Subclinical
synovitis as detected by ultrasound is a frequent finding in newly
diagnosed children with celiac disease.
Keywords: Arthritis, Chronic diarrhea, Gluten-free diet,
Synovitis, Ultrasonography.
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C eliac disease (CD) is an immune mediated systemic
disorder elicited by gluten and related prolamines in genetically
susceptible individuals [1,2]. Together with the classical form, there
are atypical forms presenting with predominating extra-intestinal
clinical features [3,4]. Musculoskeletal manifestations of CD include
arthalgia, myopathy and non-erosive arthritis, which may be clinically
silent in earlier stages [5-7]. Musculoskeletal ultrasound has shown its
superiority over conventional radiology to detect a wide set of early
inflammatory and structural abnormalities in joints [8-11]. There is a
paucity of studies evaluating early joint involvement by ultrasound in
children affected by CD. The present study was undertaken to determine
the proportion of cases with early joint involvement using
ultrasonography in children with newly diagnosed CD, and in children
with CD on gluten-free diet (GFD) for more than 6 months.
Methods
This study included all children (age 2 to 18 years)
diagnosed as CD as per the modified ESPGHAN criteria [2] from outpatient
clinic or impatient services in SMS Medical College, Jaipur from May
2014 to April 2016.
We divided these children into two groups: group 1
constituted children newly diagnosed as CD and were on gluten-containing
diet whereas group 2 constituted those already diagnosed with CD, and on
GFD for more than 6 months. Children who had any connective tissue
disorder, concomitant rheumatic fever or history of injury to joints in
last two months were excluded from the study. Informed consent was
obtained from parents of the enrolled children. The study was approved
by the ethical committee of the institution.
All patients were evaluated by complete medical
history, including history of musculoskeletal symptoms and other
co-morbidities; and detailed physical exami-nation, including
musculoskeletal assessment. Ultrasonographic assessment of bilateral
hip, knee and ankle joints was performed, using a Hitachii Hivision
Preirus machine, equipped with a 9–14 MHz linear array transducer,
operating at 14 MHz frequency (gain 50%). Ultra-sonography evaluation of
joints was performed by a single radiologist experienced in pediatric
musculo-skeletal Ultrasonography who was blinded to patient’s laboratory
and clinical features. The radiologist looked for joint effusion,
synovial hypertrophy or power Doppler signal (evidences of synovitis) in
the above mentioned joints.
Statistical analysis: Chi-square test was used
to compare frequencies between two groups. P<0.05 was considered
statistically significant.
Results
A total of 120 children (mean (SD) age 6.7 (3.9)y; 63
males) were enrolled in the study. Group1 and Group 2 had 60 children
each. No significant difference was observed between the two groups
regarding the demographic profile and clinical presentation of CD (Table
I).
TABLE I Baseline Characteristics of Study Participants
|
Group 1 |
Group 2 |
P value |
|
(n=60) |
(n=60) |
|
Gender (M/F) |
28/32 |
25/35 |
0.27 |
Age (y); mean (SD) |
6.32 ( 3.7) |
7.15 (3.9) |
0.23 |
Clinical form of CD; n (%) |
|
|
|
Typical |
45 (75%) |
52 (86.7%) |
0.16 |
Atypical |
15 (25%) |
8 (13.3%) |
|
Silent |
0 |
0 |
|
Arthralgia, n (%) |
3 (5%) |
0 |
0.242 |
CD: Celiac disease; Group 1: Newly diagnosis patients; Group 2:
Patients on gluten-free diet for >6mo. |
Ultrasonographic assessment showed presence of at
least one abnormality in 19 (31.7%) CD patients in group 1 whereas only
2 (3.3%) of group 2 patients had USG abnormalities (P< 0.001).
The most frequently involved joint was the knee where
11 children of group 1 had USG abnormalities. (Table II).
The finding in knee joint in group1 were: joint effusion in 7, synovial
hypertrophy in 2, and joint effusion along with synovitis in 2 children.
In group 2, both patient had joint effusion. Findings at hip joint
included joint effusion in 3, synovial hypertrophy in one, and combined
hypertrophy and effusion in one child. Two children in group 1 had
evidence of joint effusion in ankle, and only one patient had multiple
joint (hip and knee) involvement. Joint effusion was mild in all the
cases.
TABLE II Joint Ulatrasonography Findings in Children with Celiac Disease
|
No. of patients (%) |
|
P value |
|
Group 1 |
Group 2 |
|
|
(n=60) n |
(n=60) n |
|
Any abnormality |
19 (31.7) |
2 (3.3) |
<0.001 |
Joint effusion |
13 (21.7) |
2 (3.3) |
0.006 |
Synovial hypertrophy |
3 (5) |
0 |
0.24 |
Joint effusion + Synovial hypertrophy |
3 (5) |
0 |
0.24 |
Frequency of Joint Involvements |
|
|
|
Knee |
11 (18.3) |
2 (3.3) |
0.01 |
Hip |
4 (6.7) |
0 |
0.12 |
Ankle |
3 (5) |
0 |
0.24 |
Multiple Joints (Hip+Knee) |
1 (1.7) |
0 |
1.0 |
CD: Celiac disease; Group 1: Newly diagnosis patients; Group 2:
Patients on gluten-free diet for >6mo. |
Majority of the patients with ultrasonographic
evidence of joint abnormalities were asymptomatic suggesting subclinical
synovitis. Clinically only 3 (5%) patients of group 1 had joint pain and
limitation of joint movement at presentation while none of group 2
patients had similar symptoms. The mean (SD) age of cases having
arthralgia at the time of presentation was significantly higher than
asymptomatic cases [11.7 (7.5) vs 6.0 (3.3) years; P=0.01].
Discussion
In this study, ultrasonographic evidence of joint
abnormalities were present in approximately one-third of children newly
diagnosed with CD. This frequency was significantly higher than in
children with CD on GFD for more than six months, suggesting that GFD
may lead to improvement in joint abnormalities associated with CD. In
our study joint effusion was the most common abnormality followed by
synovial hypertrophy. Knee joint was the most frequently involved joint
followed by hip and ankle.
Small sample size, lack of inclusion of healthy
controls and cross-sectional nature of the study were few limitations of
this study.
Adelizzi, et al. [12] first described the
association of arthritis and CD following which there were many similar
case reports [5,6]. Both arthritis and small bowel mucosal changes have
been reported to improve with a gluten-free diet [13,14]. Lubrano, et
al. [15] studied 200 adult patients with CD, and found that
arthritis was present in 26% of patients. Lagnocco, et al. [8] in
2014, for the first time studied children with CD for joint involvement
using ultrasonography. They found that 50% of their newly diagnosed CD
children had abnormalities. The pattern of joint involvement was similar
to our study.
Subclinical joint effusion seems to be a relatively
frequent finding seen in newly-diagnosed CD patients. Ultrasonography
should be considered as a useful imaging tool for identifying CD
patients with joint changes that have not yet manifested clinically.
Early diagnosis and treatment may prevent subclinical synovitis to
manifest clinically in later life. These findings also suggest that GFD
has the potential to improve joint manifestations seen in CD.
Contributors: KG: concept and design of
study, drafted the manuscript; PA: collected and analysed the data; RKG:
analysis of data and literature search; SS: intellectual input in final
drafting and overall supervision.
Funding: None; Competing interest:
None stated.
What This Study Adds?
•
Subclinical synovitis, most commonly
in knee joint, as detected by ultrasonography may be present in
about one-third of newly diagnosed children with celiac disease.
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