Rheumatol Int (2013) 33:1601–1604 DOI 10.1007/s00296-011-1941-8
C A S E RE P O RT
Polyarticular lipoma arborescens—a clinical and aesthetical case Lígia Silva · Georgina Terroso · Luzia Sampaio · Eurico Monteiro · SoWa Pimenta · Fernanda Pinto · José A. Pinto · Francisco S. Ventura
Received: 18 May 2010 / Accepted: 13 April 2011 / Published online: 28 April 2011 © Springer-Verlag 2011
Abstract Lipoma arborescens is a benign tumor, but it may be a reactive process to other disorders, and its clinical, analytical, radiological and ultrasound presentation may be redundant to any synovial tumor. Despite the characteristic feature on magnetic resonance imaging (MRI), the correct diVerential diagnosis in atypical presentation, and the need for timely removal of the lesion to prevent
L. Silva (&) · G. Terroso · L. Sampaio Interna Complementar de Reumatologia, Serviços de Reumatologia do Hospital de São João e da Faculdade de Medicina da Universidade do Porto, Alameda Professor Hernâni Monteiro, 4202-451 Porto, Portugal e-mail:
[email protected] E. Monteiro Interno Complementar de Ortopedia, Serviço de Ortopedia do Hospital de São João, Alameda Professor Hernâni Monteiro, 4202-451 Porto, Portugal S. Pimenta Assistente Hospitalar de Reumatologia, Serviços de Reumatologia do Hospital de São João e da Faculdade de Medicina da Universidade do Porto, Alameda Professor Hernâni Monteiro, 4202-451 Porto, Portugal F. Pinto Interna Complementar de Anatomia Patológica, Serviço de Anatomia Patológica do Hospital de São João, Alameda Professor Hernâni Monteiro, 4202-451 Porto, Portugal J. A. Pinto Assistente Hospitalar Graduado de Reumatologia, Serviço de Reumatologia do Hospital de São João, Alameda Professor Hernâni Monteiro, 4202-451 Porto, Portugal F. S. Ventura Director dos Serviços de Reumatologia do Hospital de São João e da Faculdade de Medicina da Universidade do Porto, Alameda Professor Hernâni Monteiro, 4202-451 Porto, Portugal
joint damage, forces, ultimately, to invasive procedures. The clinical case reported here, fourth described in English language publications on the polyarticular form, also presented other speciWcities related to one of the swellings, in the knee. Because of its atypical location in the popliteal fossa, recurrent episodes of joint eVusion, personal history of knee trauma, pulmonary tuberculosis, and family history of rheumatoid arthritis required particular attention. This process was hampered by the refusal of knee (and ankle) surgery by the patient. He accepted surgical removal of the swellings of the wrists, for aesthetical reasons, with pathologic conWrmation of the diagnosis, and clinical success in that location. MRI of the knee showed the typical image of lipoma arborescens, but also other changes that compromise the prognosis. Keywords Lipoma arborescens · Synovial · Polyarticular
Introduction Lipoma arborescens is a benign encapsulated tumor arising from synovial villous proliferation [1–3]. Its pathogenesis is not known, and most patients have no relevant past medical history. However, it has been reported to be associated with degenerative joint disease, rheumatoid arthritis, psoriatic arthritis, diabetes mellitus, suggesting that this may be a reactive process [3, 4]. Benign tumors of synovial are rare; lipoma arborescens represents among them, only 3–8% of cases [1, 2]. Moreover, it is usually monoarticular; we found only 3 cases of polyarticular presentation described in English language publications [5–7]. Here, we report the fourth case, with its speciWcities.
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Clinical case Here, we report the clinical case of a 45-year-old Caucasian man, employee in a restaurant. He has past medical history of pulmonary tuberculosis in childhood, dyslipidemia and excessive alcohol intake (about 80 g/day), and family history of rheumatoid arthritis (his mother), with no other relevant medical history. In 2002, after a car accident with trauma to his right knee, he had pain that remitted spontaneously in a few months. He remained asymptomatic until 2006, when he again suVered a knee injury with pain of mechanical rhythm, which led him to Orthopedics consultation. Because there was a suspicion of internal meniscus injury, he underwent arthroscopy, which conWrmed the clinical suspicion. The initial clinical outcome was favorable. However, in the following months, he had worsening of knee pain (always of mechanical rhythm), associated with recurrent episodes of hydrarthrosis, without other inXammatory signs. By that time, he also noted the emergence of nonpainful swellings of elastic consistency on the extensor surface of the wrists, right popliteal fossa, and anterior left ankle. Given the progressive increase of swellings and family history of rheumatoid arthritis, he was oriented by his doctor to Rheumatology consult. With the exception of knee pain and the described swellings, he had no other symptoms. On examination, he had hydrarthrosis of the right knee and juxta-articular swellings with dimensions of
Fig. 1 Swelling on the extensor surface of the left wrist (a), right popliteal fossa (b), and left ankle (c)
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about 3 cm at the wrists and ankle and 7 cm in the popliteal fossa (Fig. 1), without inXammatory signs. There were no signs of arthritis, fever, weight loss, or other changes. Analytically, there was an elevation of transaminases (SGOT: 139, ALT: 187 IU/l), gamma-glutamyltransferase (292 IU/ l), and cholesterol (total: 221 and LDL: 140 g/l). The other measured parameters (blood count, sedimentation rate, C-reactive protein, alkaline phosphatase, bilirubin, triglycerides, urea, creatinine, calcium, phosphorus, uric acid, reaction of Wright, immunoglobulin G, A, M, light chains, complement [C3c, C4 and CH50] and antibodies [antinuclear, nuclear-speciWc, citrulline and rheumatoid factor]) were within normal reference values. The microbiological, cytological, and biochemical studies of the Xuid from the right knee arthrocentesis revealed no changes suggestive of an infectious (including bacillary) or inXammatory process (cytology: 3,684 cells, 26.3% neutrophils, 73.7% mononuclear cells, 2,600 red cells/mcL). Abdominal and renal ultrasonography revealed “fatty liver”, and radiographs showed no abnormalities, except for juxta-articular swellings (Fig. 2). MRI of the right knee conWrmed the diagnosis of “…lipoma arborescens, with 71.8 £ 32.9 mm…” and also showed “…slight heterogeneity of the internal meniscus,…intra-articular free chondroid bodies, the largest with 6 mm,… large volume of joint eVusion, Baker’s cyst with 130 £ 40 £ 34 mm, and irregular bone surfaces with marginal osteophytes and prominent tibial spines…” (Fig. 3). The patient was proposed for surgical excision of the
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Fig. 2 Radiography of the left wrist (a), right knee (b), and left foot (c)
Fig. 4 Pathology (right wrist): “lipoma arborescens”
of “lipoma arborescens” (Fig. 4), and 2 years after, there were no signs of recurrence or any local symptoms. Fig. 3 MRI of right knee
Discussion swelling in the knee (and ankle), which he refused, even though it had been explained the possible complications. On the contrary, he showed great interest in surgical removal of the swellings on the extensor surface of the wrists, for aesthetical reasons. Its pathology conWrmed the diagnosis
Lipoma arborescens is a benign tumor of the synovial, of unknown etiology. Despite reports of association with some diseases such as rheumatoid arthritis, usually there is no record of relevant clinical history [3, 4], unlike HoVa’s
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disease, an infrapatellar fat hyperplasia, often secondary to trauma or inXammatory process [2]. It aVects mostly men, aged 40-60 years [3, 4]. The most characteristic form of presentation is the appearance of a monoarticular painless swelling of elastic consistency, usually in the knee (suprapatellar pouch), which insidiously increases, over months to years. However, there may be less typical manifestations, such as the attainment of other joints (shoulder, wrist, hip, ankle) [3], exceptionally in a polyarticular form, and can cause mechanical symptoms such as pain, clicking, locking [2], or even recurrent episodes of hydrarthrosis, possibly due to the interposition and trauma of villi between the joint surfaces [3, 4]. Just as symptoms, also the analytical study (usually normal), radiological, and ultrasound are nonspeciWc, contributing mainly to the exclusion of other processes. Occasionally, the joint Xuid can be moderately inXammatory or xanthochromic (probably by trauma of synovial villi) [3, 4] and plain radiography, usually with no other changes besides the increased density of juxta-articular soft tissues, may, in cases of late diagnosis, already reveal “degenerative” joint damage [1, 3]. In contrast, MRI shows a typical image of the lesion, with hyperintensity on T1-weighted images (which is abolished in the sequences with fat saturation), intermediate signal on T2, and does not capture contrast (Gadolinium-DTPA) [3]. According to many authors, this aspect is pathognomonic, bypassing the need for preoperative biopsy [4]. Pathognomonic or not, associated with its location, is usually suYcient to establish the diVerential diagnosis with the most frequent swellings. They are, in the knee, the pigmented villonodular synovitis (usually in the semimembranous pouch, in the popliteal fossa), the synovial hemangioma, articular lipoma and HoVa’s disease (usually located in the infrapatellar fat pad), and the synovial osteochondromatosis. Arthroscopy also occupies a prominent position, with dual function: diagnostic, by typical appearance and possibility of biopsy, and therapeutic, with resection of the lipoma. However, this technique is recommended for those forms localized to the anterior compartment of the knee; in the remaining cases, surgical synovectomy is preferable [3]. There are rare reported cases of chemical [8] and radiosynovectomy [9], without recurrence after 1 year of follow-up. The prognosis is generally good, with no sequelae, and no recurrence after complete resection [3, 4], but depends on the duration of
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the disease, since there are reports of joint damage in cases of late diagnosis [1]. For all this, it is essential to proper diagnosis, excluding other causes or associated diseases. In this speciWc case, special attention was given to the swelling of the knee, by its atypical location, recurrent episodes of hydrarthrosis, personal history of local trauma, pulmonary tuberculosis, and family history of rheumatoid arthritis. The diYculty was increased by limiting access to material of biopsy, because the patient refused surgery of the knee. Although MRI could establish the diagnosis with some certainty, the local prognosis is already compromised. The patient was informed about the likelihood of progression of joint damage and possible complications (e.g local vasculo-nervous compression), but his motivation remained targeted solely to the (well succeeded) excision of the wrist swellings, for aesthetical reasons. Acknowledgement interest.
The authors declare that they have no conXict of
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