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\begin{document}

\title{Primary synovial chondromatosis in children: a case report and
literature review}



\author[1]{Bettaieb Hiba}%
\author[2]{Hanene Ferjani}%
\affil[1]{Universite de Tunis El Manar Faculte de Medecine de Tunis}%
\affil[2]{Mohammed Kassab National Institute of Orthopaedics}%


\vspace{-1em}



  
  \date{May 25, 2024}


\begingroup
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\maketitle
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\selectlanguage{english}
\begin{abstract}
Primary synovial chondromatosis is an uncommon and benign metaplastic
condition that usually affects large joints. Though shoulder involvement
is scarce, and there are only a few cases in the pediatric population.%
\end{abstract}%



\sloppy


\textbf{INTRODUCTION:}

Primary synovial Chondromatosis (PSC), also called Reichel syndrome, is
a benign tumor with cartilaginous nodules in the synovium joints. It
usually affects diarthrodial joints and occurs between the third and
fifth decade of life {[}5,7,17{]}. The glenohumeral joint is an unusual
location in PSC, particularly in pediatric patients {[}4,7,16{]}. To our
knowledge, there are only 5 cases of children reported in the
literature.~Herein, we report a rare case of PSC in a 14-year-old boy
with an uncommon localization in the shoulder revealed by the chronic
pain and the limited motion in the right arm. We also discuss clinical
presentation, imaging findings and treatment of this rare condition in
children based on current literature.

\textbf{CASE PRESENTATION:}

A 14-year-old, right-handed boy presented to the Pediatric Orthopedics
department with right shoulder pain. He complained of 14 months history
of pain and discomfort in his right shoulder.~He had no trauma or family
history of osteochondroma. He reported no symptoms of weight loss,
fatigue, systemic signs, or any other arthralgia.

On physical examination, there was no obvious deformity or atrophy
involving the affected shoulder. We noted a decreased range of motion,
in comparison to the uninvolved side, with respectively: flexion to
160\selectlanguage{ngerman}°, extension to 40°, abduction to 140°, adduction to 40°, internal
rotation to L4, and external rotation to 50°. Subacromial impingement
signs, as well as~rotator cuff tear tests, were negative.~The joint was
stable. A neurovascular exam revealed no deficits in the upper limb.

The laboratory results were slightly normal, particularly the calcium
and the phosphorus rate.~Plain radiographs showed multiple radio-opaque
bodies distributed throughout the glenohumeral joint, without bone
defection or joint narrowing (Figure 1). Subsequent Magnetic resonance
imaging (MRI) revealed a high number of calcified intra-articular loose
bodies around the joint and the biceps tendon (Figure 2). PSC was
strongly suspected.

We choose a shoulder arthroscopy using a deltopectoral approach to
remove the tumors nodules. More than 50 shiny and solid bodies, with an
average size of 10-15 millimeters, were retrieved (Figure 3). There were
also several bodies within the coracoid process and the conjoint tendon.
The synovial tissue, bursas, and cartilages appeared intact. Upon
removing the particles, we complete a partial synovectomy to avoid the
relapse. Histology of loose bodies and synovium confirmed the diagnosis
of PSC by revealing multiple cartilaginous nodules, composed of
clustered chondrocytes and embedded in synovium (Figure 4). No sign of
malignant transformation was noted.

Postoperative shoulder X-rays did not show any densities. The patient
was discharged after the surgery using the arm sling.~At six months of
follow-up\textbf{,} the patient remains free of symptoms, and shoulder
radiographs showed no recurrence of calcification.

\textbf{DISCUSSION:}

First mentioned by Jaffe et al., PSC is a rare benign tumor affecting
the synovial cavity {[}5{]}. It is a proliferation of multiple
cartilaginous nodules in the synovium of joints, tendon sheaths, and
bursae.

These nodules may break free from the synovial membrane to become
cartilaginous loose bodies in the joint space {[}4,10,15,17-20{]}.

According to the literature, this disorder usually occurs in men between
the ages of 30 and 50 old-years. It has been reported that knees, hip,
elbow, and wrists are the main affected joints in descending order of
frequency {[}10,12,15{]}. The involvement of the shoulder is unusual in
adults, and more exceptional during childhood. To the best of our
knowledge,~only five cases have been reported in the literature
{[}4,7,12,14,16{]}.

Based on the underlying pathogenesis, synovial chondromatosis may be
primary or secondary. The PSC, called idiopathic synovial
osteochondromatosis or Reichel syndrome, usually occurs in a previously
healthy joint.~~In contrast, secondary osteochondromatosis is a sequela
of intra-articular pathology as osteochondral fracture, osteochondritis
dissecans, and osteoarthritis {[}4,9,7,11,15,20{]}.~In our case, the
young-onset, the absence of a history of trauma, and the unremarkable
results of blood tests strengthen the diagnosis of PSC.

It is noteworthy that histopathologic analysis is mandatory to
distinguish between these conditions. {[}4,6,10,15,19{]}. In a series of
136 presumed synovial osteochondromatosis, Villain et al showed that the
histopathological patterns are different. In fact, in primary lesions,
the foci of chondrometaplasia in both synovium cells and loose bodies
are noticeably disorganized. The chondrocytes are frequently binucleate,
and the pattern of calcification is patchy and diffuse. However, in
secondary lesions, calcification is zonal and uniformly distributed, and
fragments of articular cartilage or subchondral bone are usually in the
loose bodies {[}19{]}.~In the present case, histologic evaluation
revealed multiple cartilaginous nodules arranged in clusters and
embedded in synovium.

Clinical presentation is often nonspecific {[}7,15,17{]}. As a result,
patients may experience long symptoms delays before the final diagnosis.
Like the current~case, most children with PSC of the shoulder were
diagnosed between 10 to 14 years old with a diagnosis delay ranging from
1 to 18 months from symptoms onset {[}4,7,11,12,16{]}.

In a recent literature review of cases with osteochondromatosis
occurring in the shoulder, the most common reported symptoms were mainly
shoulder pain, uncomfortable feeling during exercises, and locked joint
movement {[}18{]}.\textbf{~} Our report is following data in the
literature. Interestingly, a palpable bony mass may occur, as described
in two children with PSC of the shoulder {[}3,20{]}.

Radiographic features vary according to the degree of ossification. In
the later stages of the disease, the plain radiographs showed a
characteristic image with multiple intraarticular radio-opacities. These
calcifications are frequently very similar and uniform in size with a
typical nest-like arrangement. Thus, plain radiographs may be normal in
the earlier stages (30\% of cases) {[}3,20{]}. Sometimes, the diagnosis
overlaps between differential diagnostics such as osteosarcoma and
chondrosarcoma.

Hence, MRI plays a pivotal role in confirming the diagnosis by revealing
intra-synovial hypo-intense nodules on T1 and T2-weighted images. MRI
also aids in the management of surgical approaches. {[}2,3,5,17,18{]}.

The optimal therapeutic management of the disease requires surgical
removal of any loose bodies {[}7,8,13,16,17{]}. Partial synovectomy,
optional but often recommended, may decrease the recurrence rate
{[}15,16,20{]}. Histopathological analysis of the loose bodies and
synovial tissue is mandatory as a malign transformation may occur in up
to 5\% {[}1,7,17{]}.~The choice of surgical procedure is still a matter
of debate {[}15{]}. Open surgery remains the mainstay of treatment and
is highly recommended in cases of osteochondromatosis with soft-tissue
involvement and limited anatomic space access {[}7\textbf{{]}.~}
Moreover, this approach was often preferable in pediatric patients with
shoulder involvement {[}7,12,14,16{]}\textbf{.}On the other hand, the
arthroscopic surgery approach may offer less morbidity and earlier
postoperative recovery {[}20{]}. In line with~Hamada et al, we opted for
shoulder arthroscopy using the deltopectoral approach~{[}4{]}.\textbf{~}
To elucidate further the clinical presentation and the therapeutic
management of PSC of the shoulder in children, we analyzed findings
reported in the literature (Table 1).

According to the literature, recurrence is common and ranges between
15\% and 30\% {[}1,2,15,16{]}.\textbf{~} It's noteworthy to mention that
no recurrence of calcification was reported among pediatric patients
with PSC of the shoulder\textbf{~} {[}6,7,16{]}. However, close
follow-up including imaging in the first two years is warranted
{[}7,16{]}. In our case, the short duration of follow-up was not
sufficient to make a definitive conclusion.

In sum, the present case illustrates a rare entity of PSC that combined
the intra and extraarticular involvement of the shoulder. MRI is a
powerful key for early diagnosis. The management of this affection, like
in adult patients, is based on the chondromyxoid bodies remove through
open or arthroscope-assisted surgery. Histological analysis is mandatory
since that a malign transformation might occur. Prognosis is excellent,
associated with a low-moderate risk of recurrence.

\textbf{REFERENCES:}

1. Davis RI, Hamilton A, Biggart JD. Primary synovial chondromatosis: a
clinicopathologic review and assessment of malignant potential. Hum
Pathol. 1998;29(7):683\selectlanguage{english}-8.

2. Duymus TM, Yucel B, Mutlu S, Tuna S, Mutlu H, Komur B. Arthroscopic
treatment of synovial chondromatosis of the shoulder: A case report.
Annals of Medicine and Surgery. 2015;4(2):179\selectlanguage{english}-82. doi:
10.1016/j.amsu.2015.05.001.

3. Fischer M, Modder G. Radionuclide therapy of inflammatory joint
diseases. Nucl Med Commun. 2002;23(9):829\selectlanguage{english}-31. doi:
10.1097/00006231-200209000-00003.

4. Hamada J, Tamai K, Koguchi Y, Ono W, Saotome K. Case report: A rare
condition of secondary synovial osteochondromatosis of the shoulder
joint in a young female patient. J Shoulder Elbow Surg.
2005;14(6):653\selectlanguage{english}-6. doi: 10.1016/j.jse.2004.12.004

5. Jaffe HL. Tumors and tumorous conditions of the bones and joints.
Philadelphia: Lea and Febiger, Kimpton, London, UK, 1958.

6. Jun'ichiro H , Kazuya T, Koichi S. Secondary osteochondromatosis in
the subacromial bursa: a report of two cases and review of the
literature. J Orthop Sci. 2004;9(3):317-22. doi:
10.1007/s00776-004-0778-3.

7. Kirchhoff C, Buhmann S, Braunstein V, Weiler V, Mutschler W,
Biberthaler P. Synovial chondromatosis of the long biceps tendon sheath
in a child: a case report and review of the literature. J Shoulder Elbow
Surg. 2008;17(3):e6\selectlanguage{english}-10. doi: 10.1016/j.jse.2007.06.012.

8. Kramer J, Recht M, Deely DM, Schweitzer M, Pathria MN, Gentili A, et
al. MR appearance of idiopathic synovial osteochondromatosis. J Comput
Assist Tomogr. 1993;17(5):772\selectlanguage{english}-6.

9. McClure L, Lessman D. A Rare Cause of Shoulder Pain in Pediatrics
{[}Abstract{]}. Pediatrics. 2018;142(1):408.
doi:10.1542/peds.142.1MA5.408.

10. Milgram JW. Synovial osteochondromatosis: a histopathological study
of thirty cases. J Bone Joint Surg Am. 1977;59(6):792\selectlanguage{english}-801.

11. Milgram JW. The classification of loose bodies in human joints. Clin
Orthop Relat Res. 1977;(124):282\selectlanguage{english}-91.

12. Miranda JJ, Hooker S, Baechler MF, Burkhalter W. Synovial
chondromatosis of the shoulder and biceps tendon sheath in a 10-year-old
child. Orthopedics. 2004;27(3):321\selectlanguage{english}-3. doi:
10.3928/0147-7447-20040301-17.

13. Murphey MD, Vidal JA, Fanburg-Smith JC, Gajewski DA. Imaging of
Synovial Chondromatosis with Radiologic-Pathologic Correlation.
RadioGraphics. 2007;27(5):1465\selectlanguage{english}-88. doi: 10.1148/rg.275075116.

14. Nashi M, Manjunath B, Banerjee B, Muddu BN. Synovial chondromatosis
in a child: an unusual cause of shoulder pain case report. J Shoulder
Elbow Surg. 1998;7(6):642\selectlanguage{english}-3.

15. Poyser E, Morris R, Mehta H. Primary synovial osteochondromatosis of
the shoulder: a rare cause of shoulder pain. BMJ Case Rep . 2018
;11(1):e227281. doi: 10.1136/bcr-2018-227281.

16. Sinikumpu J-J, Sinikumpu S-P, Sirnio K, Napankangas J, Blanco
Sequeiros R. Pediatric primary synovial chondromatosis of the shoulder,
biceps tendon sheath and subcoracoid bursa. J Clin Orthop Trauma.
2020;11(2):317\selectlanguage{english}-20. doi: 10.1016/j.jcot.2019.12.005.

17. Tokis AV, Andrikoula SI, Chouliaras VT, Vasiliadis HS, Georgoulis
AD. Diagnosis and Arthroscopic Treatment of Primary Synovial
Chondromatosis of the Shoulder. Arthroscopy. 2007 23(9):1023.e1-5. doi:
10.1016/j.arthro.2006.07.009.

18. Utashima D, Matsumura N, Suzuki T, Iwamoto T, Ogawa K. Clinical
Results of Surgical Resection and Histopathological Evaluation of
Synovial Chondromatosis in the Shoulder: A Retrospective Study and
Literature Review. Clin Orthop Surg. 2020;12(1):68\selectlanguage{english}-75. ~doi:
10.4055/cios.2020.12.1.68.

19.Villacin AB, Brigham LN, Bullough PG. Primary and secondary synovial
chondrometaplasia: histopathologic and clinicoradiologic differences.
Hum Pathol. 1979;10(4):439\selectlanguage{english}-51.

20. Wahab H, Hasan O, Habib A, Baloch N. Arthroscopic removal of loose
bodies in synovial chondromatosis of shoulder joint, unusual location of
rare disease: A case report and literature review. Ann Med Surg (Lond).
2018;37:25\selectlanguage{english}-9. doi: 10.1016/j.amsu.2018.11.01

\textbf{Table 1. Clinical Presentation, Treatment, and Outcome in
patients with Synovial Chondromatosis in the literature and the present
case}\selectlanguage{english}
\begin{longtable}[]{@{}lllllllll@{}}
\toprule
\begin{minipage}[b]{0.11\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[b]{0.11\columnwidth}\raggedright\strut
\textbf{Age}\strut
\end{minipage} & \begin{minipage}[b]{0.11\columnwidth}\raggedright\strut
\textbf{Sex}\strut
\end{minipage} & \begin{minipage}[b]{0.11\columnwidth}\raggedright\strut
\textbf{Duration of symptom (months)}\strut
\end{minipage} & \begin{minipage}[b]{0.11\columnwidth}\raggedright\strut
\textbf{Trigger factor}\strut
\end{minipage} & \begin{minipage}[b]{0.11\columnwidth}\raggedright\strut
\textbf{Clinical presentation}\strut
\end{minipage} & \begin{minipage}[b]{0.11\columnwidth}\raggedright\strut
\textbf{Surgical option}\strut
\end{minipage} & \begin{minipage}[b]{0.11\columnwidth}\raggedright\strut
\textbf{Follow-up} \textbf{Period (months)}\strut
\end{minipage} & \begin{minipage}[b]{0.11\columnwidth}\raggedright\strut
\textbf{Recurrence}\strut
\end{minipage}\tabularnewline
\midrule
\endhead
\begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
\textbf{Nashi et al.} \textbf{1998} {[}14{]}.\textbf{}\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
14\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
male\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
6\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
Sporting activities\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
-Shoulder pain\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
- (under observation)\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
24\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
-\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
\textbf{Miranda et al.} \textbf{2004} {[}12{]}.\textbf{}\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
10\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
female\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
1\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
Sporting activities\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
-Shoulder pain -Discomfort\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
Arthrotomy and synovectomy\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
12\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
No\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
\textbf{Hamada et al.} \textbf{2005} {[}4{]}.\textbf{}\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
14\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
female\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
18\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
Sporting activities\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
-Shoulder pain -Discomfort\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
Arthroscopy\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
36\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
No\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
\textbf{Kirchoff et al.} \textbf{2008} {[}7{]}.\textbf{}\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
14\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
male\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
12\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
no\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
-Shoulder pain -Palpable mass\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
Arthrotomy and synovectomy\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
9\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
No\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
\textbf{Sinikumpu et al.} \textbf{2020} {[}16{]}.\textbf{}\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
14\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
male\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
12\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
Sporting activities\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
-Shoulder pain -Stiffness -Palpable mass\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
Arthrotomy and synovectomy\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
12\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
No\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
\textbf{The present case} \textbf{2021}\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
14\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
male\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
14\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
no\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
-Shoulder pain -Discomfort\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
Arthroscopy and synovectomy\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
5\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
No\strut
\end{minipage}\tabularnewline
\bottomrule
\end{longtable}

\textbf{FIGURE LEGENDS}

\textbf{Figure 1:}

Radiograph of the right shoulder demonstrates the presence of numerous
radiopaque loose bodies in the glenohumeral joint.

\textbf{Figure 2:}

STIR magnetic resonance image shows excess of synovial joint fluid and
multiple centers of nodular calcification around the joint and within
the biceps tendon sheath.

\textbf{Figure 3:}

Numerous synovial Chondromatosis nodules removed from the shoulder joint
(size between 10 and 15 mm).

\textbf{Figure 4:}

Histological image of the lesion showing multiple cartilaginous nodules
composed of clustered chondrocytes, embedded in synovium
(hematoxylin-eosin, original magnification \selectlanguage{ngerman}× 400).\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/IMAGE-1/IMAGE-1}

\end{center}
\end{figure}\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/IMAGE-2/IMAGE-2}

\end{center}
\end{figure}\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/IMAGE-3/IMAGE-3}

\end{center}
\end{figure}\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/IMAGE-4/IMAGE-4}

\end{center}
\end{figure}

\selectlanguage{english}
\FloatBarrier
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