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CASE REPORT Table of Contents   
Year : 2010  |  Volume : 44  |  Issue : 1  |  Page : 104-107
Primary synovial osteochondromatosis of a subdeltoid bursa


Department of Orthopedics, Maharaja Agrasen Hospital, Punjabi Bagh, New Delhi, India

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Date of Web Publication31-Dec-2009
 

   Abstract 

Primary synovial osteochondromatosis (SOC) is known to be intra-articular and wherever it is observed outside a synovial joint, it is associated with the involvement of the nearby joint. Primary SOC has not been reported to involve a subdeltoid bursa. We present a case of a 52-year-old woman having a large number of loose bodies in a large tumor in the subdeltoid bursa. The swelling was first noticed by the patient 2 years back. Plain roentgenogram revealed soft tissue swelling only with no areas of calcification. On MRI, multiple nonosseous loose bodies were visualized in the bursa deep to the deltoid muscle. A surgical excision of subdeltoid bursa was done. A biopsy confirmed it to be cartilaginous loose bodies in synovial lining sugestive of metaplastic transformation of the synovial tissue.

Keywords: Subdeltoid bursa, synovial osteochondromatosis, extraarticular synovial osteochondromatosis

How to cite this article:
Kumar A, Aggarwal A, Sahni VK. Primary synovial osteochondromatosis of a subdeltoid bursa. Indian J Orthop 2010;44:104-7

How to cite this URL:
Kumar A, Aggarwal A, Sahni VK. Primary synovial osteochondromatosis of a subdeltoid bursa. Indian J Orthop [serial online] 2010 [cited 2019 Dec 7];44:104-7. Available from: http://www.ijoonline.com/text.asp?2010/44/1/104/58613

   Introduction Top


Synovial osteochondromatosis (SOC) is a benign metaplastic proliferative disorder of the synovium [1],[2],[3],[4],[5] which affects subintimal fibroblasts in synovial joints, [4],[6],[7] tendons, and bursae (involving articular or tendon sheath synovial membranes) in which multiple nodules of cartilage are produced. [3],[5],[8] Many of the nodules subsequently become detached from the synovial membrane and float in the joint. [9] The synovial fluid nourishes the loose bodies which can remain viable and increase in size. [2],[3],[7],[10] It is a rare disease and rarely affects the shoulder joint; it has male predominance affecting knee, [11],[12],[13] elbow, [8],[14] hip, [10],[15],[16],[17],[18],[19] and ankle joints (in order of frequency) in the fourth/fifth decade of life. [4],[7] It presents with a stiffness of the joint and dull ache. The intra-articular loose bodies obstruct the joint mobility and the joint may get locked in a particular position. Theoretically, SOC may affect any synovial joint [3],[6] and may lead to a very big tumor sufficient to obstruct range of motion.

In respect to shoulder joint primary SOC has been reported in the subacromial bursa, subclavicular region and along the brachial plexus, but all are associated with the involvement of the glenohumeral joint. [20],[21],[22],[23] We present this case because it was completely extra-articular (below the bulk of the deltoid muscle) without involving the shoulder joint.


   Case Report Top


A 52-year-old woman presented with a swelling over the outer aspect of her left shoulder and proximal part of her left arm which she had first noticed 2 years ago. The swelling was obstructing the movements of her affected shoulder and was cosmetically unacceptable [Figure 1]a. She had no history of trauma or fever. Her appetite was not affected, and there had been no weight loss. The globular swelling was painless and measured approximately 8 inches in diameter. No tenderness or warmth, but some pseudofluctuation was observed, along with restriction of all movements except adduction. No abnormality was detected in central nervous, cardiovascular, respiratory, genitourinary, or gastrointestinal systems. She was postmenopausal with no gynecological problem. Routine preoperative investigations (including liver function and renal function tests) were within the normal limit. Serological investigation for rheumatoid arthritis was negative. Plain roentgenogram revealed a soft tissue swelling only [Figure 1]b. There were no areas of calcification.

On MRI, multiple nonosseous loose bodies were visualized in the bursa deep to the deltoid muscle. These spread up to the subacromial space as well as the anterior and posterior aspects of the shoulder. These appeared to remain outside the rotator cuff [Figure 1]c and d. Aspiration returned the nonhemorrhagic synovial fluid with no sign of tuberculosis or other chronic infective pathology.

The tumor was exposed through a linear incision over the deltopectoral groove. A large protruding synovial membrane was visible under the deltoid muscle [Figure 2]a. The synovial sheath was adherent to the surrounding structures. Innumerable pea-size cartilaginous loose bodies were extruded on an incidental rupture of the sheath [Figure 2]b. All the loose bodies were taken out and the whole bursal sheath was removed from below the deltoid and from the subacromial space [Figure 2]c. There was no connection of synovial bursa to the joint and all loose bodies remained external to the rotator cuff. A histopathological study revealed the early stages of the development of cartilaginous loose bodies in the synovial lining, suggestive of metaplastic transformation of the synovial tissue [Figure 3]a and b. Areas below the synovial lining contained a large number of mononuclear cells and a fair number of small blood vessels. This was consistent with chronic inflammation. In the cut-section of the loose body, chondrocytes were visible inside the newly formed cartilage [Figure 3]c.

By 2 weeks, the wound was well healed and the patient recovered a full range of motion of the affected shoulder. At 1 year follow-up she does not have any recurrence.


   Discussion Top


Osteochondromatosis can be primary or secondary. In secondary osteochondromatosis, the presence of free osteochondral bodies is due to osteoarthritis, trauma-causing osteochondral fractures, or osteochondritis dissecans. Primary SOC is not associated with frank osteoarthritis or synovitis [2] and the source of the osteochondral loose bodies is the benign synovial neoplasia. [22] Both the synovial membrane and articular cartilage develop from the same mesenchymal tissue, and this tissue arises from embryonic rests. [9] Jones, [11] in 1927, showed microscopically that the formation of these cartilaginous and osteocartilaginous bodies follow the same stages that occur in the embryonic formation of cartilage. The process is peculiar in that the bodies are formed from the synovial membrane instead of the articular surface, as in the case of osteochondritis dissecans and at times in the case of osteoarthritis. The bodies formed in SOC are composed of organized tissues, and are distinct from the unorganized tissues of "corpora oryzoidea," or "rice bodies." [2]

Mukerjiea [20] reported SOC of the shoulder joint in a 12-year-old boy. Buess and Friedrich [21] reported a case of a 22-year-old man with SOC of the shoulder joint. Recently, Antonogiannakis et al. [22] reported a case of osteochondromatosis of the subacromial bursa in a 72-year-old retired military officer.

It is a rare disease [7] and it rarely affects the shoulder joint. [3],[5],[7],[9],[15],[16],[17],[18],[19],[20],[21],[22],[23],[24],[25],[26],[27],[28] Multiple loose bodies are formed which get detached from the synovial membrane and may float in the synovial fluid inside the joint. It has been demonstrated in multiple studies in the shoulder [12],[13],[14] but is never limited to the subdeltoid bursa. In our case, a large number of nonossified cartilaginous loose bodies were found in the synovial membrane of the subdeltoid bursa. These surrounded the shoulder but remained extra-articular.

Only when the bodies are calcified or ossified are they visible on plain roentgenograms, [17] but the number is always greater than one would suspect from the film. Air or double contrast arthrography may be necessary to visualize the nonossified chondromatous bodies. [16] But MRI could visualize almost all such bodies. Mussey and Henderson concluded that negative findings on microscopic examination of the synovial membrane do not necessarily negate a diagnosis of osteochondromatosis, for the process might have completed its cycle and the membrane might have resumed its normal appearance. [3]

Loose bodies must be removed early to halt further damage to articular surfaces. [10],[15],[19] Synovial osteochondromatosis does not resolve spontaneously [7] and complications like degenerative osteoarthritis, [10] joint subluxation, [10] and bursitis [8] are not uncommon. The patient should be forewarned that a certain amount of degenerative arthritis is already present and may cause residual symptoms. [11] It is controversial whether to perform complete synovectomy along with excision of all communicating bursae, [13],[15] because recurrences are common [11],[15],[19] irrespective of the extent of the excision. [27] Therefore, many authors favor removal of all loose bodies with subtotal synovectomy. [4],[17],[20]

Though suspected in some observations, [13] sarcomatous degeneration of the synovial chondromatosis has not been proved.


   Acknowledgments Top


We wish to express our grateful thanks to Dr. Manoj Sareen and Dr. Sunita Kapoor for their kind help and histopathological opinion in the preparation of this manuscript.

 
   References Top

1.Henderson MS, Joxis HT. Loose bodies in joints and bursae due to synovial osteochondromatosis. J Bone Joint Surg 1923;5:400-4  Back to cited text no. 1      
2.Jones HT. Loose body formation in synovial osteochondromatosis with special reference to the etiology and pathology. J Bone Joint Surg 1924;6:407-58  Back to cited text no. 2      
3.Mussey RD, Henderson MS. Osteochondromatosis. J Bone Joint Surg Am 1949;31:619-27.  Back to cited text no. 3      
4.Jeffreys TE. Synovial chondromatosis. J Bone Joint Surg Br 1967;49:530-4.  Back to cited text no. 4  [PUBMED]  [FULLTEXT]  
5.Dorfman HD, Czerniak: Synovial chondromatosis. In synovial lesions. In "Bone Tumors". Mosby; 1998. p. 1041-57.  Back to cited text no. 5      
6.Milgram JW. Synovial osteochondromatosis: A histopathological study of thirty cases. J Bone Joint Surg Am 1977;59:792-801.  Back to cited text no. 6  [PUBMED]  [FULLTEXT]  
7.Murphy FP, Dahlin DC, Sullivan CR. Articular Synovial Chondromatosis. J Bone Joint Surg Am 1962;44:77-86.  Back to cited text no. 7      
8.Matsumoto K, Hukuda S, Fujita M, Kakimoto A, Tachibana S. Cubital bursitis caused by localized synovial chondromatosis of the elbow: A case report. J Bone Joint Surg Am 1996;78:275-7.  Back to cited text no. 8  [PUBMED]  [FULLTEXT]  
9.Turek SL. Diseases of joints. In "Orthopaedics: Principles and their application". 4 th ed. (Indian Reprint, 2004), Lippincott - Raven; 1984. Volume-1: p. 455-7.  Back to cited text no. 9      
10.Hardacker J, Mindell ER. Synovial chondromatosis with secondary subluxation of the hip. A case report. J Bone Joint Surg Am 1991;73:1405-7.  Back to cited text no. 10      
11.Jones HT. The Histogenesis of cartilage as shown in chondromatosis of the knee joint. J Bone Joint Surg 1927;9:310-4.  Back to cited text no. 11      
12.Dunn AW, Whisler JH. Synovial chondromatosis of the knee with associated extracapsular chondromas. J Bone Joint Surg Am 1973;55:1747-8.  Back to cited text no. 12  [PUBMED]    
13.Milgram JW, Addison RG. Synovial osteochondromatosis of the knee. chondromatous recurrence with possible chondrosarcomatous degeneration. J Bone Joint Surg Am 1976;58:264-6.  Back to cited text no. 13      
14.Kamineni S, O'Driscoll SW, Morrey BF. Synovial osteochondromatosis of the elbow. J Bone Joint Surg Br 2002;48:961-6.  Back to cited text no. 14      
15.Lim SJ, Chung HW, Choi YL, Moon YW, Seo JG, Park YS. Operative treatment of primary synovial osteochondromatosis of the hip. J Bone Joint Surg Am 2006;88:2456-64.  Back to cited text no. 15  [PUBMED]  [FULLTEXT]  
16.Bloom R, Pattinson JN. Osteochondromatosis of the hip joint. J Bone Joint Surg Br 1951;33:80-4.  Back to cited text no. 16  [PUBMED]  [FULLTEXT]  
17.McIvor RR, King D. Osteochondromatosis of the hip joint. J Bone Joint Surg Am 1962;44:87-97.  Back to cited text no. 17      
18.Okada Y, Awaya G, Ikeda T, Tada H, Kamisato S, Futami T. Arthroscopic surgery for synovial chondromatosis of the hip. J Bone Joint Surg Br 1989;71:198-9.  Back to cited text no. 18  [PUBMED]  [FULLTEXT]  
19.Lim SJ, Park YS. Operative treatment of primary synovial osteochondromatosis of the hip. J Bone Joint Surg Am 2007;89:232-45.  Back to cited text no. 19  [PUBMED]  [FULLTEXT]  
20.Mukerjiea SK. Synovial chondromatosis in shoulder joint. Proc Roy Soc Med 1968;61:265-7.  Back to cited text no. 20      
21.Buess E, Friedrich B. Synovial chondromatosis of the glenohumeral joint: A rare condition. Arch Orthop Trauma Surg 2001;121:109-11.  Back to cited text no. 21  [PUBMED]  [FULLTEXT]  
22.Antonogiannakis E, Yiannakopoulos CK, Mataragas I. Osteochondromatosis of the subacromial bursa arthroscopic treatment of a case and review of the literature. EEXOT 2008;59:67-70.  Back to cited text no. 22      
23.Sim FH, Dahlin DC, Ivins JC. Extra-articular synovial chondromatosis. J Bone Joint Surg Am 1977;59:492-5.  Back to cited text no. 23  [PUBMED]  [FULLTEXT]  
24.Wakhlu A. Synovial osteochondromatosis. J Indian Rheumatol Assoc 2004;12:77.  Back to cited text no. 24      
25.Wilmoth CL. Osteochondromatosis. J Bone Joint Surg Am 1941;23:367-74.  Back to cited text no. 25      
26.Milgram JW, Pease CN. Synovial osteochondromatosis in a young child: A case report. J Bone Joint Surg Am 1980;62:1021-3.  Back to cited text no. 26  [PUBMED]  [FULLTEXT]  
27.Maurice H, Crone M, Watt I. Synovial chondromatosis. J Bone Joint Surg Br 1988;70:807-11.  Back to cited text no. 27  [PUBMED]  [FULLTEXT]  
28.Shanbhag A, Balakrishnan C, Bhaduri A. Primary synovial osteochondromatosis. J Indian Rheumatol Assoc 2004;12:29-30.  Back to cited text no. 28      

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Correspondence Address:
Anil Kumar
Flat No. 103, Block No.-2, Punjabi Bagh Enclave, DDA MIG Flat, Shiv Mandir Road, Madipur, New Delhi - 110 063
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0019-5413.58613

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    Figures

  [Figure 1], [Figure 2], [Figure 3]

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    Abstract
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