Acta Ortopédica Mexicana Volumen Suplemento Julio-Diciembre Volume 18 Supplement 1 July-December 2004 Artículo: Villonodular synovitis of the shoulder joint. A case report Derechos reservados, Copyright © 2004: Sociedad Mexicana de Ortopedia, AC Otras secciones de Others sections in este sitio: this web site: ☞ Índice de este número ☞ Contents of this number ☞ Más revistas ☞ More journals ☞ Búsqueda ☞ Search edigraphic.com Acta Ortopédica Mexicana 2004; 18(Suppl. 1): Jul.-Dec: S55-S58 Case report Villonodular synovitis of the shoulder joint. A case report Oscar Martínez-Molina,* José Vázquez-García** Central South Pemex Hospital SUMMARY. Introduction. Pigmented villonod- RESUMEN. Introducción. La sinovitis vellonodu- ular synovitis is a proliferative disorder affecting lar pigmentada es un desorden proliferativo que the joints, tendon sheaths and bursas. The most af- afecta articulaciones, vainas tendinosas y bursas. Las fected joints are the knees, hips and fingers. Ac- articulaciones más afectadas son: rodilla, cadera y cording to literature reviews, the shoulders are dedos. De acuerdo a revisiones en la literatura, el less affected. Material and methods. In this paper, hombro es el menos afectado. Material y métodos. En we are reporting a case found on the glenohumer- el presente artículo hacemos el reporte de un caso lo- al joint, in a 77 year old female patient. She was calizado en la articulación glenohumeral, en una pa- also suffering from rheumatoid arthritis. Her clin- ciente de 77 años de edad, portadora además de ar- ical chart was characterized by profuse joint effu- tritis reumatoide. Con un cuadro clínico caracteriza- do por derrame articular profuso se le sometió a sion. She underwent shoulder arthroscopy by bi- artroscopía del hombro, mediante biopsias y se co- opsies and the histopathological diagnosis of rroboró el diagnóstico histopatológico de sinovitis diffuse pigmented villonodular synovitis was con- vellonodular pigmentada difusa. Discusión. Dada la firmed. Discussion. Given the rareness of occur- rareza de la presentación en el hombro resulta difícil rence in the shoulder, it is difficult to define the definir los hallazgos clínicos y de apoyo diagnóstico, clinical findings and diagnostic support. However, sin embargo, nuestro caso al igual que la generali- our case, like nearly all those reported so far, dad de los hasta ahora reportados comparten: evolu- shares a slow clinical course, general and non spe- ción clínica lenta, presencia de dolor generalizado e cific pain in the shoulder, increased volume of the inespecífico en el hombro, aumento de volumen, asi- joint, and region asymmetry. In all cases, anato- metría de la región; en todos los casos, son entonces mopathological findings are those showing long los hallazgos anatomopatológicos que muestran ve- villi, extending to the peripheral osteochondral llosidades largas, extendidas de la periférica unión joint into the joint with a brownish color, some- osteocondral, hacia la articulación, con una colora- times bloody, which microscopically describes a ción parda, a veces sanguinolenta y cuya descripción proliferative synovia, fibroblasts or primitive microscópica muestra una sinovial proliferativa, fi- mesenchymatous cells which, eventually provide a broblastos o células mesenquimatosas primitivas, la particular diagnosis. que finalmente da el diagnóstico específico. Key words: synovitis, villonodular, pigmented, Palabras clave: sinovitis, vellonodular, pigmen- shoulder. tada, hombro. Introduction Pigmented villonodular synovitis is a proliferative dis- *Ortopedic Sugeon. Central South Pemex Hospital, (Spanish acronym order of the synovia affecting the joints, bursas, and ten- of Mexican Petroleous). edigraphic.comdon sheaths. Chassaignac was the first to describe a nodu- ** Head of the Orthopedic Department. lar lesion of the membrana synovialis affecting the tendon sheaths of the fingers. Simon described for the first time Mailing address: the localized shape and Moser, was the first to describe the Dr. Oscar Martínez-Molina. Periférico Sur 4091. Unidad Portes Gil 3,4 edificio N-2 Depto. 7 Col. Fuentes del Pedregal. Tlalpan C.P. diffuse shape in the knee. Down was the first to suspect 14140 México, D.F. its malignant origin. The lesion has been described with S55 Oscar Martínez-Molina et al. Figure 1. AP X-ray of the shoulder. Figure 3. Arthroscopic view. Synovial Villi. Figure 2. Arthroscopic view. Synovial Villi. Figure 4. Arthroscopic view. Synovectomy in process. several terms. Among these, synovial xanthoma, synovial fibroendothelioma, benign fibrous histiocytoma, xanth- omatous giant cell tumor, giant cell tumor of the tendon sheath, fibrohemosideric sarcoma and others were includ- ed. In 1941, Jaffe proposed the terms pigmented villonod- ular synovitis, pigmented villonodular bursitis, and pig- mented villonodular tenosynovitis. With these terms clinically occurring variants were covered. In 1945, Jaffe formally published his studies.6 Granowitz subclassified two clinical forms by adding first the prefix (L) for pedicu- lated or localized lesions and (D) for diffuse lesions.3,4,8 Case report This is a 77 year old female patient having a 6 year Figure 5. Arthroscopic view. Synovectomy in process. history of adult rheumatoid arthritis, medicallyedigraphic.com con- trolled with azathioprine, 50 mg every 24 hours, pred- nisone 10 mg per day, and etodolac, 300 mg every 12 (++++) and associated to pain on the anterior face of the hours. In December 1977, the patient was first seen at shoulder, a feeling of distension and limitation of the the orthopedics service when she noticed an increased arches of motion. Previously, the rheumatology service volume of her left shoulder, secondary to joint effusion performed two arthrocenteses drawing 75 and 150 cc of ACTA ORTOPÉDICA MEXICANA 2004; 18(Suppl. 1): S55-S58 S56 Villonodular synovitis of the shoulder joint fluidsustraídode-m.e.d.i.g.r.a.p.h.i.c respectively. The arthrocentesis cytochemical re- In 1994 Tong reported a case of a :ropshoulder odarobale joint. InFDP re- cihpargidemedodaborport showed dark yellow, lustrous and turbid fluid, total viewing the literature he found 14 cases reported so far.18 proteins 4.88 g/dl, 9,700 cells/LU leukocytes, 6,810 In 1999 Müller presentedVC eda caseAS, cidemihparGof a young patient with a LU, erythrocytes, 2,150 LU, PMN 80%, MN 20%. The shoulder lesion studied by MRI. The lesion appeared to monthly report in September, 6 months later, showed simulate a malignant tumor. Upon reviewarap of the literature, turbid yellow fluid, total proteins 2.50 g/dl, 7,910 cells/ he found 25 cases of shoulder villonodular synovitis.11 In LU, 2,940 leukocytes LU, 3,970 erythrocytes LU; PMN our review,acidémoiB in addition arutaretiL to the above :cihpargideM papers, we have found 90% and MN 10%. Clinically the patient did insidious- sustraídode-m.e.d.i.g.r.a.p.h.i.cone more case reported by Joseph Cheng in 1997, refer- ly with functional limitation of her shoulder: abduction ring to a patient subject to two prior procedures for gleno- 80°, adduction 10°, flexion 70° and pain was exacerbat- humeral instability. The first procedure involved arthros- ed when trying to go beyond these ranges. In addition, a copy and the second, was open surgery using the anterior major increased volume with anatomical deformity ac- capsulolabral reconstruction technique. After these two companied these symptoms. The X-ray film showed an approaches and three asymptomatic years, revision ar- AP projection with osteopenia of the humeral head, throscopy was performed and two fibronodular, vascular- thinned corticals, and the presence of subchondral bone ized lesions of the anterior glenoid were found. The histo- cysts (Figure1). pathological analysis revealed localized villonodular An ultrasound of the joint was taken to confirm the flu- synovitis.2 Konrath reported yet another case, also in id collection. The patient underwent shoulder arthroscopy 1997, although this is an extra-articular lesion found in through a posterior portal from which a total 350 cc of yel- the subacromial bursa. Coincidentally, Saw Miller report- low brownish fluid with lumpy detritus was removed. Ar- ed another localized case in the subacromial and deltoid throscopy also revealed a hypertrophic, abundant and dif- bursa, that very same year. fuse synovia, involving the entire glenohumeral joint With regards to the etiopathology of this disorder, there showing gross digitations or elongated nodes with a pink are major ongoing controversies at this time. Some authors base and brown streaks, and distension of the anterior propose synovial hyperplasia related to idiopathic inflam- joint capsule (Figures 2 and 3). matory components as the cause for etiology. Other au- Later, with the arthroscope the author conducted a thors relate the lesion to benign neoplastic processes and broad synovectomy and cleansing of the joint (Figure 4 a reaction to repetitive trauma. More recent histopatholog- and 5). The material taken was sent to the pathologist who ical analyses and DNA densitometry lead towards a neo- made the following report: hyperplastic synovial cells, plastic cause supported also by abnormal findings of subsynovial nodular proliferation with elongated archi- aneoploidism and cytogenic findings. There are reports tectures, isolated findings of hemosiderin. The final diag- such as those by Bertoni with three documented cases of nosis was: pigmented villonodular synovitis. malignant transformation.1,2,4,5,7,13 After the