Case Report

Pigmented Villonodular Synovitis in a 15-Month-Old Boy Challenged the Physicians for Two Years: A Case Report and Literature Review

Abstract

Background: Pigmented villonodular synovitis (PVNS) is a very rare pathology of the synovial membrane and even less common in the pediatric group. Rarity leads to misdiagnosis in pre-puberty cases, which may waste significant time for the patient before definite treatment. Reviewing such cases is useful for every pediatrician or orthopedic surgeon to avoid misdiagnosing possible cases.

 

Case Report: We report a 15-month-old boy who suffered pain and swelling in his knee for about two years before he was eventually diagnosed with PVNS and underwent surgery. Keeping this diagnosis in mind may have saved him and his family from two years of pain, several admissions, and unnecessary prescriptions.

 

Conclusion: PVNS in pediatrics is rare, but it can occur and be misdiagnosed for diseases such as juvenile rheumatoid arthritis (JRA) and septic arthritis which elongates the pain period. PVNS responds well to subtotal synovectomy, and symptoms are relieved after the surgery if well performed.

1. Karami M, Soleimani M, Shiari R. Pigmented villonodular synovitis in pediatric population: review of literature and a case report. Pediatr Rheumatol Online J. 2018;16(1):6. doi: 10.1186/s12969-018-0222-4. [PubMed: 29343257]. [PubMed Central: PMC5772710].

2. Neubauer P, Weber AK, Miller NH, McCarthy EF. Pigmented villonodular synovitis in children: a report of six cases and review of the literature. Iowa Orthop J. 2007;27:90-4. [PubMed: 17907437]. [PubMed Central: PMC2150657].

3. Turkucar S, Makay B, Tatari H, Unsal E. Pigmented villonodular synovitis: Four pediatric cases and brief review of literature. J

Postgrad Med. 2019;65(4):233-6. doi: 10.4103/jpgm.JPGM_305_19. [PubMed: 31611431]. [PubMed Central: PMC6813690].

4. Sen D, Sen AM, Gupta A, Manibanker NB. Pigmented villonodular synovitis in a child: A missed diagnosis. Med J

Armed Forces India. 2019;75(4):467-71. doi: 10.1016/j.mjafi.2017.11.001. [PubMed: 31719744]. [PubMed Central: PMC6838497].

5. Bahrami S, Baheiraei N, Shahrezaee M. Biomimetic reduced graphene oxide coated collagen scaffold for in situ bone regeneration. Sci Rep. 2021;11(1):16783. doi: 10.1038/s41598-021-96271-1. [PubMed: 34408206]. [PubMed Central: PMC8373942].
6. Zhao L, Zhou K, Hua Y, Li Y, Mu D. Multifocal pigmented villonodular synovitis in a child: A case report. Medicine

(Baltimore). 2016;95(33):e4572. doi: 10.1097/MD.0000000000004572. [PubMed: 27537585]. [PubMed Central: PMC5370811].

7. Sahin Er, Tatari H. Pigmented villonodular synovitis of the knee confused with juvenile rheumatoid arthritis in a 3-year-old child: A case report. J Surg Med. 2020;4(9):838-41. doi: 10.28982/josam.716164.

8. Eckhardt BP, Hernandez RJ. Pigmented villonodular synovitis: MR imaging in pediatric patients. Pediatr Radiol. 2004;34(12):943-7. doi: 10.1007/s00247-004-1261-1. [PubMed: 15448943].

9. Norouzi M, Naderi MN, Komasi MH, Sharifzadeh SR, Shahrezaei M, Eajazi A. Clinical results of using the proximal humeral internal locking system plate for internal fixation of displaced proximal humeral fractures. Am J Orthop (Belle Mead NJ ). 2012;41(5):E64-E68. [PubMed: 22715443].

10. Mahdavi SM, Shariati B, Shalbafan M, Rashedi V, Yarahmadi M, Ghaznavi A, et al. The effectiveness of pregabalin with or without agomelatine in the treatment of chronic low back pain: a double-blind, placebo-controlled, randomized clinical trial. BMC Pharmacol Toxicol. 2022;23(1):70. doi: 10.1186/s40360-

022-00612-3. [PubMed: 36104745]. [PubMed Central: PMC9476640].

11. Poutoglidou F, Metaxiotis D, Mpeletsiotis A. Pigmented Villonodular Synovitis of the Knee Joint in a 10-Year-Old Patient Treated With an All-Arthroscopic Synovectomy: A Case Report. Cureus. 2020;12(12):e11929. doi: 10.7759/cureus.11929. [PubMed: 33425510]. [PubMed Central: PMC7785498].

12. Haghighizadeh E, Shahrezaee M, Sharifzadeh SR, Momeni M. Transforming growth factor-beta3 relation with osteoporosis

and osteoporotic fractures. J Res Med Sci. 2019;24:46. doi: 10.4103/jrms.JRMS_1062_18. [PubMed: 31160913]. [PubMed Central: PMC6540765].

13. Ottaviani S, Ayral X, Dougados M, Gossec L. Pigmented villonodular synovitis: a retrospective single-center study of 122 cases and review of the literature. Semin Arthritis Rheum. 2011;40(6):539-46. doi: 10.1016/j.semarthrit.2010.07.005. [PubMed: 20884045].

14. Baroni E, Russo BD, Masquijo JJ, Bassini O, Miscione H. Pigmented villonodular synovitis of the knee in skeletally immature patients. J Child Orthop. 2010;4(2):123-7. doi: 10.1007/s11832-009-0236-z. [PubMed: 21455469]. [PubMed Central: PMC2839859].

15. Flandry F, Hughston JC. Pigmented villonodular synovitis. Journal of Bone and Joint Surgery. 1987;69(6):942-9.

16. Yeganeh A, Amiri S, Moghtadaei M, Otoukesh B, Solati A, Daneshmand S. Moderately elevated erythrocyte sedimentation rate (ESR) or C-reactive protein (CRP) in patients with no clinical signs or symptoms of infection is not a contraindication for total knee arthroplasty: A retrospective cohort study. Curr Orthop Pract. 2022;33(1):52-5. doi: 10.1097/BCO.0000000000001070.

17. Rezaei H, Shahrezaee M, Jalali MM, Ghorbani F, Zamanian A, Sahebalzamani M. Mussel-inspired polydopamine induced the osteoinductivity to ice-templating PLGA-gelatin matrix for bone tissue engineering application. Biotechnol Appl Biochem. 2021;68(1):185-96. doi: 10.1002/bab.1911. [PubMed: 32248561].

18. Nakhaie Amroodi M, Abdolahi Kordkandi S, Moghtadaei M, Farahini H, Amiri S, Hajializade M. A study of characteristic features and diagnostic roles of X-ray and MRI in calcifying tendinitis of the shoulder. Med J Islam Repub Iran. 2022;36(1): 574-8. [In Persian]. doi: 10.47176/mjiri.36.79.

19. Shariatzadeh H, Amiri S, Joudi S, Bahrabadi M. Multiple Schwannomas in Sciatic Nerve: A Rare Case Report. Arch Bone Jt Surg. 2021;9(5):601-4. doi: 10.22038/abjs.2021.53165.2638. [PubMed: 34692945]. [PubMed Central: PMC8503757].
Files
IssueVol 9 No 4 (2023) QRcode
SectionCase Report
DOI https://doi.org/10.18502/jost.v9i4.13937
Keywords
Case Reports Pigmented Villonodular Synovitis Synovial Membrane

Rights and permissions
Creative Commons License This work is licensed under a Creative Commons Attribution-NonCommercial 4.0 International License.
How to Cite
1.
Taheriaazam A, Mohammadnejad R, Baghbani S, Khanmohammadi B, Dindar Mehrabani A, Farokhzadeh A. Pigmented Villonodular Synovitis in a 15-Month-Old Boy Challenged the Physicians for Two Years: A Case Report and Literature Review. J Orthop Spine Trauma. 2023;9(4):189-91.