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LETTERS TO THE EDITOR
may be useful. Perhaps in these individuals, the recommended dose of additional folic acid needs to be increased. Lauren Dautzenberg, MSc Department of Geriatric Medicine, Jeroen Bosch Hospital, Hertogenbosch, the Netherlands Naomi Jessurum, PharmD Netherlands Pharmacovigilance Centre Lareb, Hertogenbosch, the Netherlands Paul L. J. Dautzenberg, MD, PhD Carolina J. P. W. Keijsers, MD, PhD Department of Geriatric Medicine, Jeroen Bosch Hospital, Hertogenbosch, the Netherlands
ACKNOWLEDGMENTS Conflict of Interest: Paul J. L. Dautzenberg is on the 2012 advisory board for Eli-Lilly and the 2012 advisory board for Novartis; received sponsorship for a memory congress from 2001 to 2013 from Novartis; and participated in Phase III medication trials for MSD, Novartis, and Janssen-Cilag during the conduct of the study. All other authors declare no support from any organization for the submitted work; no financial relationships with any organizations that might have an interest in the submitted work in the previous 3 years; no other relationships or activities that could appear to have influenced the submitted work. Author Contributions: All authors had full access to all of the data in the study and take responsibility for the integrity of the data and the accuracy of the data analysis. All authors contributed to the drafting of the manuscript. Data collection: Dautzenberg. Study supervision: Keijsers. All authors approved the final version. Sponsor’s Role: No sponsor.
REFERENCES 1. Johnston A, Gudjonsson JE, Sigmundsdottir H et al. The anti-inflammatory action of methotrexate is not mediated by lymphocyte apoptosis, but by the suppression of activation ad adhesion molecules. Clin Immunuol 2005;114:154–163. 2. Chan ES, Cronstein BN. Methotrexate—how does it really work? Nat Rev Rheumatol 2010;6:175–178. 3. Verstappen CC, Heijmans JJ, Hoekman K et al. Neurotoxic complications of chemotherapy in patients with cancer: Clinical signs and optimal management. Drugs 2003;63:1549–1563. 4. Madhyastha S, Somayaji SN, Rao MS et al. Hippocampal brain amines in methotrexate-induced learning and memory deficit. Can J Physiol Pharmacol 2002;80:1076–1084. 5. Wernick R, Smith DL. Central nervous system toxicity associated with weekly low-dose methotrexate treatment. Arthritis Rheum 1989;32: 770–775. 6. Ramos MI, Allen LH, Mungas DM et al. Low folate status is associated with impaired cognitive function and dementia in the Sacramento Area Latino Study on Ageing. Am J Clin Nutr 2005;82:1346–1352. 7. Hinterberger M, Fischer P. Folate and Alzheimer: When time matters. J Neural Transm 2013;120:211–224. 8. Watanabe S, Sato S, Nagase S et al. Effects of methotrexate and cyclophosphamide on polyamine levels in various tissues of rats. J Drug Target 1999;7:197–205. 9. Luo J, Yu CH, Yu H et al. Cellular polyamines promote amyloid-beta (Ab) peptide fibrillation and modulate the aggregation pathways. ACS Chem Neurosci 2013;4:454–462.
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10. Skatchkov SN, Woodbury-Farina MA, Eaton M. The role of glia in stress: Polyamines and brain disorders. Psychiatry Clin North Am 2014;37:653– 678. 11. Belzung C, Willner P, Philppot P. Depression: From psychopathology to pathophysiology. Curr Opin Neurobiol 2014;30C:24–30.
PSEUDOTUMOR AFTER METAL-ON-METAL HIP ARTHROPLASTY To the Editor: An 86-year-old woman with a history of Alzheimer’s disease, chronic obstructive pulmonary disease, depression, osteoporosis, and right and left total hip replacements (metal-on-metal implants, performed 13 and 11 years ago, respectively) presented with subacute right hip pain. She was functionally independent, living at home with her daughter and mobilizing without gait aids. She presented with 3 weeks of worsening right hip pain without any history of trauma or falls. She was afebrile and had minimal tenderness on palpation, with full range of motion over the hip joint. X-ray of the right hip showed no fractures but periprosthetic sclerosis, and ultrasound showed a chronic complex collection. Two separate aspirates of the collection were sterile. After discussion with the patient’s daughter, a conservative approach was adopted, with a trial of empirical oral antibiotic suppression for presumed chronic hip prosthesis infection. Over the next 8 months, her pain worsened and was associated with marked swelling anteriorly and posteriorlaterally around the hip joint despite ongoing antibiotics. She now required a frame to walk. Computed tomography (CT) of the pelvis showed bilateral sacral ala and pubic rami fractures. There were circumscribed soft tissue density masses surrounding the right total hip replacement measuring 5 by 3 cm anteriorly and 6 by 5 cm posteriorly that showed rapid enlargement on serial scans. A core biopsy of the masses was performed and yielded negative cultures, with histopathology showing hematoma with no evidence of organization present. Postcontrast magnetic resonance imaging (MRI) showed a heterogenous hyperintense mass without significant enhancement (Figure 1). A diagnosis of pseudotumor secondary to metallosis was made based on the clinical findings with minimal inflammation over the hip joint, the characteristic MRI findings, the negative microbiology, and lack of response to antibiotics. Testing found high serum cobalt levels (22 nmol/L, normal range 2–7 nmol/L) further supporting the diagnosis, but her serum chromium was normal at 15 nmol/L (normal range 2–40 nmol/L). Metal-on-metal (MoM) hip replacements have been commonly used in Australia (21,400 between 1999 and 2010), with advantages of low wear rates and incidence of dislocation, but there are concerns that the release of metal (chromium and cobalt) ions and particles from these implants may have adverse cardiac, neurological, endocrine, and dermatological effects.1 A local inflammatory reaction to this process has also been described and may form pseudotumors or be mistaken for infection, as in this case. These periprosthetic soft tissue lesions have been
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tend to do poorly are potential reasons for early revision.5 In frail elderly adults, particularly with dementia, there are concerns about operative risks, short future lifespan, and a preference for conservative management. Conservative management seems appropriate for the woman describe herein until operative management for palliative reasons cannot be postponed any longer. David Lim, MBBS Austin Hospital, Melbourne, Victoria, Australia Cameron Jeremiah, MBBS Northern Hospital, Melbourne, Victoria, Australia
Figure 1. Pseudotumor after hip arthroplasty. Axial short tau inversion recovery image showing (A) heterogenous hyperintense mass with (B) a hypointense capsule possibly due to microscopic metal particles that displaces but does not invade the muscles surrounding the thigh.
described as bursae, cysts, inflammatory or granulomatous masses, or adverse reactions to metal debris (metallosis).2 The diagnosis is often confirmed using soft-tissue imaging modalities such as ultrasound or metal artifact reduction sequence MRI.3 Histology and serum or hip aspirate metal ion levels can be helpful. Pseudotumors after hip arthroplasty have been described since 1976. Aggressive granulomatous lesions were found in cemented metal-on-polyethylene total hip arthroplasty,4 but similar pathology related to metal-onmetal bearings has only recently been recognized.4 There is often a latent period of 2 to 15 years after the initial total joint replacement before this foreign-body reaction becomes clinically or radiologically apparent.4 Precise incidence and prevalence is unknown because there are appreciable numbers of asymptomatic or subclinical pseudotumors.5 The pathogenesis is due to osteolysis adjacent to prosthetic material and a cytotoxic and delayed hypersensitivity (Type IV) response to the deposition of cobalt–chrome wear particles in periprosthetic tissues.6 Histologically, a spectrum of changes comprising pure metallosis, aseptic lymphocytic vasculitis associated lesion, and granulomatous inflammation is seen.7 All people with MoM implants have modestly high serum chromium and cobalt concentrations, but symptomatic individuals with serum chromium levels greater than 15 ng/mL and cobalt levels greater than 10 ng/mL are likely to have significant implant deterioration.8 On MRI, the pseudotumor can vary in size (but often >5 cm), can be fluid or solid with minimal or no contrast enhancement, and can extend out from the femoral neck into surrounding tissues.9 Pseudotumors might incidentally be present in individuals with well-functioning hip prostheses, suggesting that the presence of a pseudotumor may not necessarily indicate the need for revision arthroplasty.10 However, concerns about the progression of asymptomatic pseudotumors and the fact that pseudotumor revisions
Altay Altuntas, MBBS Northern Hospital, Melbourne, Victoria, Australia St Vincent’s Hospital, Melbourne, Victoria, Australia University of Melbourne, Melbourne, Victoria, Australia Rabin Sinnappu, MBBS Northern Hospital, Melbourne, Victoria, Australia Richard O’Sullivan, MBBS Epworth Hospital, Melbourne, Victoria, Australia Monash University, Melbourne, Victoria, Australia Wen Kwang Lim, MBBS Northern Hospital, Melbourne, Victoria, Australia University of Melbourne, Melbourne, Victoria, Australia
ACKNOWLEDGMENTS Conflict of Interest: The editor in chief has reviewed the conflict of interest checklist provided by the authors and has determined that the authors have no financial or any other kind of personal conflicts with this paper. Author Contributions: Lim: literature review, preparation of manuscript. Jeremiah: preparation of manuscript. Altuntas, Sinnappu, O’Sullivan, Lim: editing of manuscript. Sponsor’s Role: No sponsor for this case report.
REFERENCES 1. Metal-on-Metal Hip Replacement Implants. Therapeutic Goods Administration, Department of Health, Australia [on-line]. Available at https:// www.tga.gov.au/metal-metal-hip-replacement-implants#implant\ Accessed November 23, 2014. 2. Kwon YM, Glyn-Jones S, Simpson DJ et al. Analysis of wear of retrieved metal-on-metal hip resurfacing implants revised due to pseudotumours. J Bone Joint Surg Br 2010;92:356–361. 3. Kwon YM. Cross-sectional imaging in evaluation of soft tissue reactions secondary to metal debris. J Arthroplasty 2014;29:653–656. 4. Pseudotumors following total hip and knee arthroplasty. Joint Evidence [on-line]. Available at http://www.mcminncentre.co.uk/pdf/pseudotumorstotal-hip-knee-arthroplasty.pdf Accessed November 23, 2014. 5. Kwon YM, Ostlere SJ, McLardy-Smith P et al. “Asymptomatic” pseudotumors after metal-on-metal hip resurfacing arthroplasty: Prevalence and metal ion study. J Arthroplasty 2011;26:511–518. 6. Mahendra G, Pandit H, Kliskey K et al. Necrotic and inflammatory changes in metal-on-metal resurfacing hip arthroplasties. Acta Orthop 2009;80:653–659. 7. Natu S, Sidaginamale RP, Gandhi J et al. Adverse reactions to metal debris: Histopathological features of periprosthetic soft tissue reactions seen in association with failed metal on metal hip arthroplasties. J Clin Pathol 2012;65:409–418.
1276
LETTERS TO THE EDITOR
8. Evaluation of Metal-on-Metal Wear of Orthopedic Implants—The Role of Serum Chromium and Cobalt Analysis. Mayo Clinic [on-line]. Available at http://www.mayomedicallaboratories.com/articles/communique/2012/01. html Accessed November 23, 2014. 9. Hayter CL, Gold SL, Koff MF et al. MRI findings in painful metal on metal hip arthroplasty. AJR Am J Roentgenol 2012;199:884–893. 10. Hart AJ, Satchithananda K, Liddle AD et al. Pseudotumors in association with well-functioning metal-on-metal hip prostheses: A case–control study using three-dimensional computed tomography and magnetic resonance imaging. J Bone Joint Surg Am 2012;94:317–325.
IDIOPATHIC PRESENTATION OF REMITTING SERONEGATIVE SYMMETRICAL SYNOVITIS WITH PITTING EDEMA IN AN OLDER MAN To the Editor: A 67-year-old man presented to the outpatient clinic because of swelling, morning stiffness, and pain in the both hands for 6 months. He had no other constitutional symptoms such as fever, fatigue, sweating, and weight loss. There was a past medical history of hypertension and diabetes mellitus. In his examination, he had tenderness of the metacarpal and proximal interphalangeal joints and edema of the dorsal side of both hands (Figure 1A). Initial laboratory tests showed an erythrocyte sedimentation rate of 112 mm/h (normal range 0–20 mm/h), C-reactive protein of 88 mg/L (normal range 0–10 mg/L), and hemoglobin of 9.8 g/L (normal range14–16 g/L). Serum uric acid and total free prostate-specific antigen levels were normal. Tests for rheumatoid factor (RF), anticyclic citrullinated peptide antibodies (anti-CCP), antinuclear antibody (ANA), and human leukocyte antigen (HLA)-B27 were all negative. A plain radiograph of the wrist showed diffuse soft tissue swelling and distal interphalangeal osteoarthritis of the hands. Ultrasound showed tenosynovitis. He was diagnosed with remitting seronegative symmetrical synovitis with pitting edema (RS3PE) syndrome and treated with prednisolone (10 mg/d), acemetacin (120 mg/d), and hydroxychloroquine (400 mg/d). He was screened and investigated for associated malignancies. Thoracoabdominopelvic computed tomography and abdominal ultrasonography were normal. Joint pain and edema improved within 4 weeks (Figure 1B), and acutephase reactants decreased. At follow-up 4 weeks later, prednisolone was slowly tapered and stopped at 2 months. RS3PE syndrome, a rare inflammatory arthritis, mostly affects older adults. It can present as acute-onset polyarthritis with associated pitting edema of the extremi-
A
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ties.1 It can be associated with solid tumors such as prostatic, rectal, gastric, colic, endometrial, hepatocellular, ovarian, pancreatic, and undifferentiated adenocarcinoma and with hematological malignancies.2 Its etiology is unknown. It can occur as an idiopathic phenomenon but also in association with various types of rheumatic diseases, most frequently late-onset rheumatoid arthritis and polymyalgia rheumatica.3 RS3PE can also be associated with gout, especially in men and elderly people.4 Individuals with RS3PE should be monitored for neoplasia with prudent age- and sex-specific surveillance for a long period of time. Long-term follow-up keeping is critical in management of RS3PE. Mustafa Turgut Yildizgoren, MD Department of Physical Medicine and Rehabilitation, School of Medicine, Mustafa Kemal University, Hatay, Turkey Ali Sahin, MD Division of Rheumatology, Faculty of Medicine, Cumhuriyet University, Sivas, Turkey Kasim Osmanoglu, MD Onur Velioglu, MD Department of Physical Medicine and Rehabilitation, School of Medicine, Mustafa Kemal University, Hatay, Turkey
ACKNOWLEDGMENTS Conflict of Interest: The authors declare that they have no conflict of interest. Author Contributions: All authors contributed to this paper. Sponsor’s Role: None.
REFERENCES 1. Salam A, Henry R, Sheeran T. Acute onset polyarthritis in older people: Is it RS3PE syndrome? Cases J 2008;1:132. 2. Cantini F, Olivieri I, Salvarani C. More on remitting seronegative symmetrical synovitis with pitting oedema as paraneoplastic syndrome. J Rheumatol 1998;25:188–189. 3. Yanai H, Yoshida H, Tada N. Clinical, radiological, and biochemical characteristics in patients with diseases mimicking polymyalgia rheumatica. Clin Interv Aging 2009;4:391–395. 4. Hakozaki M, Fukuda H, Tajino T et al. Remitting seronegative symmetrical synovitis with pitting edema syndrome caused by crystal-induced arthritis of the wrist: A case report. Med Princ Pract 2013;22:307–310.
B
Figure 1. Edema of dorsal side of both hands (A) before and (B) after treatment.
LETTERS TO THE EDITOR
may be useful. Perhaps in these individuals, the recommended dose of additional folic acid needs to be increased. Lauren Dautzenberg, MSc Department of Geriatric Medicine, Jeroen Bosch Hospital, Hertogenbosch, the Netherlands Naomi Jessurum, PharmD Netherlands Pharmacovigilance Centre Lareb, Hertogenbosch, the Netherlands Paul L. J. Dautzenberg, MD, PhD Carolina J. P. W. Keijsers, MD, PhD Department of Geriatric Medicine, Jeroen Bosch Hospital, Hertogenbosch, the Netherlands
ACKNOWLEDGMENTS Conflict of Interest: Paul J. L. Dautzenberg is on the 2012 advisory board for Eli-Lilly and the 2012 advisory board for Novartis; received sponsorship for a memory congress from 2001 to 2013 from Novartis; and participated in Phase III medication trials for MSD, Novartis, and Janssen-Cilag during the conduct of the study. All other authors declare no support from any organization for the submitted work; no financial relationships with any organizations that might have an interest in the submitted work in the previous 3 years; no other relationships or activities that could appear to have influenced the submitted work. Author Contributions: All authors had full access to all of the data in the study and take responsibility for the integrity of the data and the accuracy of the data analysis. All authors contributed to the drafting of the manuscript. Data collection: Dautzenberg. Study supervision: Keijsers. All authors approved the final version. Sponsor’s Role: No sponsor.
REFERENCES 1. Johnston A, Gudjonsson JE, Sigmundsdottir H et al. The anti-inflammatory action of methotrexate is not mediated by lymphocyte apoptosis, but by the suppression of activation ad adhesion molecules. Clin Immunuol 2005;114:154–163. 2. Chan ES, Cronstein BN. Methotrexate—how does it really work? Nat Rev Rheumatol 2010;6:175–178. 3. Verstappen CC, Heijmans JJ, Hoekman K et al. Neurotoxic complications of chemotherapy in patients with cancer: Clinical signs and optimal management. Drugs 2003;63:1549–1563. 4. Madhyastha S, Somayaji SN, Rao MS et al. Hippocampal brain amines in methotrexate-induced learning and memory deficit. Can J Physiol Pharmacol 2002;80:1076–1084. 5. Wernick R, Smith DL. Central nervous system toxicity associated with weekly low-dose methotrexate treatment. Arthritis Rheum 1989;32: 770–775. 6. Ramos MI, Allen LH, Mungas DM et al. Low folate status is associated with impaired cognitive function and dementia in the Sacramento Area Latino Study on Ageing. Am J Clin Nutr 2005;82:1346–1352. 7. Hinterberger M, Fischer P. Folate and Alzheimer: When time matters. J Neural Transm 2013;120:211–224. 8. Watanabe S, Sato S, Nagase S et al. Effects of methotrexate and cyclophosphamide on polyamine levels in various tissues of rats. J Drug Target 1999;7:197–205. 9. Luo J, Yu CH, Yu H et al. Cellular polyamines promote amyloid-beta (Ab) peptide fibrillation and modulate the aggregation pathways. ACS Chem Neurosci 2013;4:454–462.
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10. Skatchkov SN, Woodbury-Farina MA, Eaton M. The role of glia in stress: Polyamines and brain disorders. Psychiatry Clin North Am 2014;37:653– 678. 11. Belzung C, Willner P, Philppot P. Depression: From psychopathology to pathophysiology. Curr Opin Neurobiol 2014;30C:24–30.
PSEUDOTUMOR AFTER METAL-ON-METAL HIP ARTHROPLASTY To the Editor: An 86-year-old woman with a history of Alzheimer’s disease, chronic obstructive pulmonary disease, depression, osteoporosis, and right and left total hip replacements (metal-on-metal implants, performed 13 and 11 years ago, respectively) presented with subacute right hip pain. She was functionally independent, living at home with her daughter and mobilizing without gait aids. She presented with 3 weeks of worsening right hip pain without any history of trauma or falls. She was afebrile and had minimal tenderness on palpation, with full range of motion over the hip joint. X-ray of the right hip showed no fractures but periprosthetic sclerosis, and ultrasound showed a chronic complex collection. Two separate aspirates of the collection were sterile. After discussion with the patient’s daughter, a conservative approach was adopted, with a trial of empirical oral antibiotic suppression for presumed chronic hip prosthesis infection. Over the next 8 months, her pain worsened and was associated with marked swelling anteriorly and posteriorlaterally around the hip joint despite ongoing antibiotics. She now required a frame to walk. Computed tomography (CT) of the pelvis showed bilateral sacral ala and pubic rami fractures. There were circumscribed soft tissue density masses surrounding the right total hip replacement measuring 5 by 3 cm anteriorly and 6 by 5 cm posteriorly that showed rapid enlargement on serial scans. A core biopsy of the masses was performed and yielded negative cultures, with histopathology showing hematoma with no evidence of organization present. Postcontrast magnetic resonance imaging (MRI) showed a heterogenous hyperintense mass without significant enhancement (Figure 1). A diagnosis of pseudotumor secondary to metallosis was made based on the clinical findings with minimal inflammation over the hip joint, the characteristic MRI findings, the negative microbiology, and lack of response to antibiotics. Testing found high serum cobalt levels (22 nmol/L, normal range 2–7 nmol/L) further supporting the diagnosis, but her serum chromium was normal at 15 nmol/L (normal range 2–40 nmol/L). Metal-on-metal (MoM) hip replacements have been commonly used in Australia (21,400 between 1999 and 2010), with advantages of low wear rates and incidence of dislocation, but there are concerns that the release of metal (chromium and cobalt) ions and particles from these implants may have adverse cardiac, neurological, endocrine, and dermatological effects.1 A local inflammatory reaction to this process has also been described and may form pseudotumors or be mistaken for infection, as in this case. These periprosthetic soft tissue lesions have been
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B
A
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tend to do poorly are potential reasons for early revision.5 In frail elderly adults, particularly with dementia, there are concerns about operative risks, short future lifespan, and a preference for conservative management. Conservative management seems appropriate for the woman describe herein until operative management for palliative reasons cannot be postponed any longer. David Lim, MBBS Austin Hospital, Melbourne, Victoria, Australia Cameron Jeremiah, MBBS Northern Hospital, Melbourne, Victoria, Australia
Figure 1. Pseudotumor after hip arthroplasty. Axial short tau inversion recovery image showing (A) heterogenous hyperintense mass with (B) a hypointense capsule possibly due to microscopic metal particles that displaces but does not invade the muscles surrounding the thigh.
described as bursae, cysts, inflammatory or granulomatous masses, or adverse reactions to metal debris (metallosis).2 The diagnosis is often confirmed using soft-tissue imaging modalities such as ultrasound or metal artifact reduction sequence MRI.3 Histology and serum or hip aspirate metal ion levels can be helpful. Pseudotumors after hip arthroplasty have been described since 1976. Aggressive granulomatous lesions were found in cemented metal-on-polyethylene total hip arthroplasty,4 but similar pathology related to metal-onmetal bearings has only recently been recognized.4 There is often a latent period of 2 to 15 years after the initial total joint replacement before this foreign-body reaction becomes clinically or radiologically apparent.4 Precise incidence and prevalence is unknown because there are appreciable numbers of asymptomatic or subclinical pseudotumors.5 The pathogenesis is due to osteolysis adjacent to prosthetic material and a cytotoxic and delayed hypersensitivity (Type IV) response to the deposition of cobalt–chrome wear particles in periprosthetic tissues.6 Histologically, a spectrum of changes comprising pure metallosis, aseptic lymphocytic vasculitis associated lesion, and granulomatous inflammation is seen.7 All people with MoM implants have modestly high serum chromium and cobalt concentrations, but symptomatic individuals with serum chromium levels greater than 15 ng/mL and cobalt levels greater than 10 ng/mL are likely to have significant implant deterioration.8 On MRI, the pseudotumor can vary in size (but often >5 cm), can be fluid or solid with minimal or no contrast enhancement, and can extend out from the femoral neck into surrounding tissues.9 Pseudotumors might incidentally be present in individuals with well-functioning hip prostheses, suggesting that the presence of a pseudotumor may not necessarily indicate the need for revision arthroplasty.10 However, concerns about the progression of asymptomatic pseudotumors and the fact that pseudotumor revisions
Altay Altuntas, MBBS Northern Hospital, Melbourne, Victoria, Australia St Vincent’s Hospital, Melbourne, Victoria, Australia University of Melbourne, Melbourne, Victoria, Australia Rabin Sinnappu, MBBS Northern Hospital, Melbourne, Victoria, Australia Richard O’Sullivan, MBBS Epworth Hospital, Melbourne, Victoria, Australia Monash University, Melbourne, Victoria, Australia Wen Kwang Lim, MBBS Northern Hospital, Melbourne, Victoria, Australia University of Melbourne, Melbourne, Victoria, Australia
ACKNOWLEDGMENTS Conflict of Interest: The editor in chief has reviewed the conflict of interest checklist provided by the authors and has determined that the authors have no financial or any other kind of personal conflicts with this paper. Author Contributions: Lim: literature review, preparation of manuscript. Jeremiah: preparation of manuscript. Altuntas, Sinnappu, O’Sullivan, Lim: editing of manuscript. Sponsor’s Role: No sponsor for this case report.
REFERENCES 1. Metal-on-Metal Hip Replacement Implants. Therapeutic Goods Administration, Department of Health, Australia [on-line]. Available at https:// www.tga.gov.au/metal-metal-hip-replacement-implants#implant\ Accessed November 23, 2014. 2. Kwon YM, Glyn-Jones S, Simpson DJ et al. Analysis of wear of retrieved metal-on-metal hip resurfacing implants revised due to pseudotumours. J Bone Joint Surg Br 2010;92:356–361. 3. Kwon YM. Cross-sectional imaging in evaluation of soft tissue reactions secondary to metal debris. J Arthroplasty 2014;29:653–656. 4. Pseudotumors following total hip and knee arthroplasty. Joint Evidence [on-line]. Available at http://www.mcminncentre.co.uk/pdf/pseudotumorstotal-hip-knee-arthroplasty.pdf Accessed November 23, 2014. 5. Kwon YM, Ostlere SJ, McLardy-Smith P et al. “Asymptomatic” pseudotumors after metal-on-metal hip resurfacing arthroplasty: Prevalence and metal ion study. J Arthroplasty 2011;26:511–518. 6. Mahendra G, Pandit H, Kliskey K et al. Necrotic and inflammatory changes in metal-on-metal resurfacing hip arthroplasties. Acta Orthop 2009;80:653–659. 7. Natu S, Sidaginamale RP, Gandhi J et al. Adverse reactions to metal debris: Histopathological features of periprosthetic soft tissue reactions seen in association with failed metal on metal hip arthroplasties. J Clin Pathol 2012;65:409–418.
1276
LETTERS TO THE EDITOR
8. Evaluation of Metal-on-Metal Wear of Orthopedic Implants—The Role of Serum Chromium and Cobalt Analysis. Mayo Clinic [on-line]. Available at http://www.mayomedicallaboratories.com/articles/communique/2012/01. html Accessed November 23, 2014. 9. Hayter CL, Gold SL, Koff MF et al. MRI findings in painful metal on metal hip arthroplasty. AJR Am J Roentgenol 2012;199:884–893. 10. Hart AJ, Satchithananda K, Liddle AD et al. Pseudotumors in association with well-functioning metal-on-metal hip prostheses: A case–control study using three-dimensional computed tomography and magnetic resonance imaging. J Bone Joint Surg Am 2012;94:317–325.
IDIOPATHIC PRESENTATION OF REMITTING SERONEGATIVE SYMMETRICAL SYNOVITIS WITH PITTING EDEMA IN AN OLDER MAN To the Editor: A 67-year-old man presented to the outpatient clinic because of swelling, morning stiffness, and pain in the both hands for 6 months. He had no other constitutional symptoms such as fever, fatigue, sweating, and weight loss. There was a past medical history of hypertension and diabetes mellitus. In his examination, he had tenderness of the metacarpal and proximal interphalangeal joints and edema of the dorsal side of both hands (Figure 1A). Initial laboratory tests showed an erythrocyte sedimentation rate of 112 mm/h (normal range 0–20 mm/h), C-reactive protein of 88 mg/L (normal range 0–10 mg/L), and hemoglobin of 9.8 g/L (normal range14–16 g/L). Serum uric acid and total free prostate-specific antigen levels were normal. Tests for rheumatoid factor (RF), anticyclic citrullinated peptide antibodies (anti-CCP), antinuclear antibody (ANA), and human leukocyte antigen (HLA)-B27 were all negative. A plain radiograph of the wrist showed diffuse soft tissue swelling and distal interphalangeal osteoarthritis of the hands. Ultrasound showed tenosynovitis. He was diagnosed with remitting seronegative symmetrical synovitis with pitting edema (RS3PE) syndrome and treated with prednisolone (10 mg/d), acemetacin (120 mg/d), and hydroxychloroquine (400 mg/d). He was screened and investigated for associated malignancies. Thoracoabdominopelvic computed tomography and abdominal ultrasonography were normal. Joint pain and edema improved within 4 weeks (Figure 1B), and acutephase reactants decreased. At follow-up 4 weeks later, prednisolone was slowly tapered and stopped at 2 months. RS3PE syndrome, a rare inflammatory arthritis, mostly affects older adults. It can present as acute-onset polyarthritis with associated pitting edema of the extremi-
A
JUNE 2015–VOL. 63, NO. 6
JAGS
ties.1 It can be associated with solid tumors such as prostatic, rectal, gastric, colic, endometrial, hepatocellular, ovarian, pancreatic, and undifferentiated adenocarcinoma and with hematological malignancies.2 Its etiology is unknown. It can occur as an idiopathic phenomenon but also in association with various types of rheumatic diseases, most frequently late-onset rheumatoid arthritis and polymyalgia rheumatica.3 RS3PE can also be associated with gout, especially in men and elderly people.4 Individuals with RS3PE should be monitored for neoplasia with prudent age- and sex-specific surveillance for a long period of time. Long-term follow-up keeping is critical in management of RS3PE. Mustafa Turgut Yildizgoren, MD Department of Physical Medicine and Rehabilitation, School of Medicine, Mustafa Kemal University, Hatay, Turkey Ali Sahin, MD Division of Rheumatology, Faculty of Medicine, Cumhuriyet University, Sivas, Turkey Kasim Osmanoglu, MD Onur Velioglu, MD Department of Physical Medicine and Rehabilitation, School of Medicine, Mustafa Kemal University, Hatay, Turkey
ACKNOWLEDGMENTS Conflict of Interest: The authors declare that they have no conflict of interest. Author Contributions: All authors contributed to this paper. Sponsor’s Role: None.
REFERENCES 1. Salam A, Henry R, Sheeran T. Acute onset polyarthritis in older people: Is it RS3PE syndrome? Cases J 2008;1:132. 2. Cantini F, Olivieri I, Salvarani C. More on remitting seronegative symmetrical synovitis with pitting oedema as paraneoplastic syndrome. J Rheumatol 1998;25:188–189. 3. Yanai H, Yoshida H, Tada N. Clinical, radiological, and biochemical characteristics in patients with diseases mimicking polymyalgia rheumatica. Clin Interv Aging 2009;4:391–395. 4. Hakozaki M, Fukuda H, Tajino T et al. Remitting seronegative symmetrical synovitis with pitting edema syndrome caused by crystal-induced arthritis of the wrist: A case report. Med Princ Pract 2013;22:307–310.
B
Figure 1. Edema of dorsal side of both hands (A) before and (B) after treatment.
