diffuse pigmented villonodular synovitis: an exuberant finding of... · 2019. 11. 8. · santo de...

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Journal of Case Reports and Images in Orthopedics and Rheumatology, Vol. 4, 2019. J Case Rep Images Orthop Rheum 2019;4:100018Z14SJ2019. www.ijcrior.com Sena et al. 1 Diffuse pigmented villonodular synovitis: An exuberant finding Jorge Sena, Gabriel Xavier, Luís Machado, Carla Brazão CASE REPORT A 70-year-old man presented at our institution with functional limitation and bilateral gonalgia. The X-ray showed aspects compatible with bilateral tricompartmental gonarthrosis (grade IV). We proposed the patient to total left knee arthroplasty. DISCUSSION Pigmented villonodular synovitis (PVNS) is a rare (1.8 per million) proliferative of the synovium. The knee is the most commonly affected site (28–70%). Despite being a benign condition, PVNS is often aggressive, resulting in secondary osteoarthritis. Monoarticular involvement occurs in two forms: localized and diffuse. The latter is more common, with a high recurrence rate [1]. Intraoperatively, we verified after incision of the fascia and opening to the articular space, an exuberant brownish/reddish fibroelastic mass that filled the entire articular space (Figure 1). This finding was in favor of diffuse intra-articular pigmented villonodular synovitis. We performed extended synovectomy and sent the piece for pathological analysis. After synovectomy, we continued to perform total knee arthroplasty. The anatomopathological results confirmed the diagnostic hypothesis (Figure 2). Jorge Sena 1 , Gabriel Xavier 2 , Luís Machado 3 , Carla Brazão 4 Affiliations: 1 5th year Resident, Department of Orthopedic Surgery and Traumatology, Hospital Espírito Santo de Évo- ra, Évora, Portugal; 2 Specialist Surgeon, Department of Or- thopedic Surgery and Traumatology, Hospital Espírito Santo de Évora, Évora, Portugal; 3 Specialist Surgeon, Department of Orthopedic Surgery and Traumatology, Hospital Beatriz Ângelo, Lisboa, Portugal; 4 1st year Resident, Department of Orthopedic Surgery and Traumatology, Hospital Espírito Santo de Évora, Évora, Portugal. Corresponding Author: Jorge Sena, Rua Ruy Luís Gomes n293, 2925-689 Azeitão-Setúbal, Portugal; Email: Jorge. [email protected] Received: 11 September 2019 Accepted: 01 October 2019 Published: 07 November 2019 This rare condition usually appears at younger ages [1, 2] and progresses with pain and recurrent hemarthrosis and may produce degenerative changes [1–3]. In these situations, the recommended treatment is synovectomy [1–3] and total knee arthroplasty [3]. Given this finding, the surgeons maintained the surgical proposal of total arthroplasty, previously performing an extensive synovectomy, recommended in this situation. In this case PVNS was just an exuberant finding that did not change treatment (Figure 3). CLINICAL IMAGE PEER REVIEWED | OPEN ACCESS Figure 1: Lush brown fibroelastic mass in the knee joint space. Figure 2: Pathology of lesion biopsy (Hematoxylin and eosin staining): (A) Hyperplastic synovial composed of macrophages containing hemosiderin; (B) Alveolar fissures and spaces lined with synovial cells, multinucleated giant cells and epithelioid cells.

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Page 1: Diffuse pigmented villonodular synovitis: An exuberant finding of... · 2019. 11. 8. · Santo de Évora, Évora, Portugal. Corresponding Author: Jorge Sena, Rua Ruy Luís Gomes n293,

Journal of Case Reports and Images in Orthopedics and Rheumatology, Vol. 4, 2019.

J Case Rep Images Orthop Rheum 2019;4:100018Z14SJ2019. www.ijcrior.com

Sena et al. 1

CASE REPORT OPEN ACCESS

Diffuse pigmented villonodular synovitis: An exuberant finding

Jorge Sena, Gabriel Xavier, Luís Machado, Carla Brazão

CASE REPORT

A 70-year-old man presented at our institution with functional limitation and bilateral gonalgia. The X-ray showed aspects compatible with bilateral tricompartmental gonarthrosis (grade IV). We proposed the patient to total left knee arthroplasty.

DISCUSSION

Pigmented villonodular synovitis (PVNS) is a rare (1.8 per million) proliferative of the synovium. The knee is the most commonly affected site (28–70%). Despite being a benign condition, PVNS is often aggressive, resulting in secondary osteoarthritis. Monoarticular involvement occurs in two forms: localized and diffuse. The latter is more common, with a high recurrence rate [1].

Intraoperatively, we verified after incision of the fascia and opening to the articular space, an exuberant brownish/reddish fibroelastic mass that filled the entire articular space (Figure 1).

This finding was in favor of diffuse intra-articular pigmented villonodular synovitis. We performed extended synovectomy and sent the piece for pathological analysis. After synovectomy, we continued to perform total knee arthroplasty. The anatomopathological results confirmed the diagnostic hypothesis (Figure 2).

Jorge Sena1, Gabriel Xavier2, Luís Machado3, Carla Brazão4

Affiliations: 15th year Resident, Department of Orthopedic Surgery and Traumatology, Hospital Espírito Santo de Évo-ra, Évora, Portugal; 2Specialist Surgeon, Department of Or-thopedic Surgery and Traumatology, Hospital Espírito Santo de Évora, Évora, Portugal; 3Specialist Surgeon, Department of Orthopedic Surgery and Traumatology, Hospital Beatriz Ângelo, Lisboa, Portugal; 41st year Resident, Department of Orthopedic Surgery and Traumatology, Hospital Espírito Santo de Évora, Évora, Portugal.Corresponding Author: Jorge Sena, Rua Ruy Luís Gomes n293, 2925-689 Azeitão-Setúbal, Portugal; Email: [email protected]

Received: 11 September 2019Accepted: 01 October 2019Published: 07 November 2019

This rare condition usually appears at younger ages [1, 2] and progresses with pain and recurrent hemarthrosis and may produce degenerative changes [1–3]. In these situations, the recommended treatment is synovectomy [1–3] and total knee arthroplasty [3]. Given this finding, the surgeons maintained the surgical proposal of total arthroplasty, previously performing an extensive synovectomy, recommended in this situation.

In this case PVNS was just an exuberant finding that did not change treatment (Figure 3).

CLINICAL IMAGE PEER REVIEWED | OPEN ACCESS

Figure 1: Lush brown fibroelastic mass in the knee joint space.

Figure 2: Pathology of lesion biopsy (Hematoxylin and eosin staining): (A) Hyperplastic synovial composed of macrophages containing hemosiderin; (B) Alveolar fissures and spaces lined with synovial cells, multinucleated giant cells and epithelioid cells.

Page 2: Diffuse pigmented villonodular synovitis: An exuberant finding of... · 2019. 11. 8. · Santo de Évora, Évora, Portugal. Corresponding Author: Jorge Sena, Rua Ruy Luís Gomes n293,

Journal of Case Reports and Images in Orthopedics and Rheumatology, Vol. 4, 2019.

Sena et al. 2J Case Rep Images Orthop Rheum 2019;4:100018Z14SJ2019. www.ijcrior.com

villonodular synovitis in knee joint: Diagnosis and treatment. Rev Bras Ortop 2017;52(4):450–7.

2. Casp AJ, Browne JA, Durig NE, Werner BC. Complications after total knee arthroplasty in patients with pigmented villonodular synovitis. J Arthroplasty 2019;34(1):36–9.

3. Mollon B, Lee A, Busse JW, et al. The effect of surgical synovectomy and radiotherapy on the rate of recurrence of pigmented villonodular synovitis of the knee: An individual patient meta-analysis. Bone Joint J 2015;97-B(4):550–7.

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Author ContributionsJorge Sena – Conception of the work, Design of the work, Acquisition of data, Analysis of data, Interpretation of data, Drafting the work, Revising the work critically for important intellectual content, Final approval of the version to be published, Agree to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved

Gabriel Xavier – Conception of the work, Design of the work, Acquisition of data, Analysis of data, Interpretation of data, Drafting the work, Revising the work critically for important intellectual content, Final approval of the version to be published, Agree to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved

Luís Machado – Conception of the work, Design of the work, Acquisition of data, Analysis of data, Interpretation of data, Drafting the work, Revising the work critically for important intellectual content, Final approval of the version to be published, Agree to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved

Carla Brazão – Conception of the work, Design of the work, Acquisition of data, Analysis of data, Interpretation of data, Drafting the work, Revising the work critically for important intellectual content, Final approval of the version to be published, Agree to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved

Guarantor of SubmissionThe corresponding author is the guarantor of submission.

Source of SupportNone.

Consent StatementWritten informed consent was obtained from the patient for publication of this article.

Figure 3: Control X-ray after total knee arthroplasty.

CONCLUSION

Pigmented villonodular synovitis is a diagnostic challenge due to the low specificity of its symptoms and subtle radiological characteristics, constituting a pathology that should be considered in the management of gonalgia, especially in young adults. Surgical excision is the gold standard method in the treatment of PVNS, and the role of arthroscopy in treating the diffuse form of PVNS is debatable. Total arthroplasty should be considered in cases of persistent relapses and advanced osteoarthritis. In this present case, PVNS was an accidental finding during the left knee joint replacement procedure. The surgeons maintained the surgical proposal of total arthroplasty, previously performing an extensive synovectomy, recommended in this situation. After eight months postoperatively, the patient is well, without pain complaints and with full range of motion.

Keywords: Diffuse pigmented villonodular synovitis, Gonalgia, Secondary osteoarthritis, Synovectomy

How to cite this article

Sena J, Xavier G, Machado L, Brazão C. Diffuse pigmented villonodular synovitis: An exuberant finding. J Case Rep Images Orthop Rheum 2019;4:100018Z14SJ2019.

Article ID: 100018Z14SJ2019

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doi:10.5348/100018Z14SJ2019CR

REFERENCES

1. Temponi EF, Barros AAG, Paganini VO, Barbosa VAK, Badet R, Carvalho Júnior LH. Diffuse pigmented

Page 3: Diffuse pigmented villonodular synovitis: An exuberant finding of... · 2019. 11. 8. · Santo de Évora, Évora, Portugal. Corresponding Author: Jorge Sena, Rua Ruy Luís Gomes n293,

Journal of Case Reports and Images in Orthopedics and Rheumatology, Vol. 4, 2019.

Sena et al. 3J Case Rep Images Orthop Rheum 2019;4:100018Z14SJ2019. www.ijcrior.com

Conflict of InterestAuthors declare no conflict of interest.

Data AvailabilityAll relevant data are within the paper and its Supporting Information files.

Copyright© 2019 Jorge Sena et al. This article is distributed under the terms of Creative Commons Attribution License which permits unrestricted use, distribution and reproduction in any medium provided the original author(s) and original publisher are properly credited. Please see the copyright policy on the journal website for more information.

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