Mod Rheumatol (2002) 12:69–71
© The Japan Rheumatism Association and Springer-Verlag Tokyo 2002
CASE REPORT
Kazuhiro Ohtani · Kanji Fukuda · Chiaki Hamanishi
Mod Rheumatol Downloaded from informahealthcare.com by The University of Manchester on 10/29/14 For personal use only.
A case of systemic lupus erythematosus associated with trigger wrist
Received: August 23, 2000 / Accepted: July 6, 2001
Abstract A 54-year-old woman complained of a painful, swollen, and clicking left wrist for 1 year. She had an 8-year history of systemic lupus erythematosus (SLE), treated with oral prednisolone. Flexion and extension of all fingers were difficult to initiate on wrist extension, and movement was accompanied by a palpable click at the wrist. Magnetic resonance imaging (MRI) and tenography revealed the expanded sheath of the flexor tendons and well-defined round free bodies known as rice bodies. Synovectomy and excision of rice bodies resulted in complete disappearance of swelling and triggering of the wrist. Key words Systemic lupus Tenosynovitis · Trigger wrist
erythematosus
(SLE)
·
Case report
difficult to initiate flexion and extension of all fingers on wrist extension, and movement was accompanied by a palpable click at the wrist. There were no symptoms or signs of median nerve compression, and there was no deformity in her fingers or wrist. X-ray examinations revealed slight scapholunate dissociation and atrophic changes of the carpus. Plain and enhanced magnetic resonance imaging (MRI) showed diffuse synovial thickening around the flexor tendons (Fig. 1). Tenography revealed the expanded sheath of the flexor tendons, and well-defined round free bodies were visible in the sheath. These free bodies moved proximally and caused the trigger on finger flexion (Fig. 2). Surgical exploration of the carpal tunnel showed a grossly thickened and congested synovial flexor sheath. There was no edematous alteration in the median nerve. All flexor tendons were surrounded by a hypertrophic synovial cuff to which were attached many nodular rice bodies, i.e., 1 ⫻ 0.5 cm of gross, yellow, soft mass (Fig. 3). Carpal tunnel decompression was completed, the rice bodies were excised, and synovectomy was carried out. The intraarticular lesions were not examined further because they were not thought to be concerned with the triggering phenomenon. Histological examination of the excised synovial tissue showed hyperplastic synovium with lymphocyte infiltration. The rice bodies were noted to consist of dense fibrinous material. One year postoperatively, the patient remains free of pain and swelling and triggering has disappeared, although there is some remaining limitation of wrist movement.
A 54-year-old, right-handed woman presented with a complaint of a painful, swollen, and clicking left wrist present for 1 year. She had an 8-year history of systemic lupus erythematosus (SLE) and fulfilled five criteria of the ACR classification1; i.e., malar rash, discoid rash, oral ulcers, hemolytic anemia, and antinuclear antibody. She was treated with oral administration of corticosteroid and controlled with medication of 5 mg/day prednisolone. She had undergone total hip arthroplasty and total knee arthroplasty for steroid-induced osteonecrosis of her left femoral head and right medial femoral condyle, respectively. She had been advised to walk with a T-cane in her left hand. Swelling and triggering of her left hand had been present for 1 year. On examination, there was a slight synovial thickening over the palmar aspect of the wrist. It was
Discussion
K. Ohtani · K. Fukuda (*) · C. Hamanishi Department of Orthopaedic Surgery, Kinki University School of Medicine, 377-2 Ohnohigashi, Osaka-sayama 589-8511, Japan Tel. ⫹81-723-66-0221 (ext. 3211); Fax ⫹81-723-67-7525 e-mail: [email protected]
Trigger wrist, first reported by Eibel in 1961, is a rare phenomenon.2 Previous reports described that nodules in the flexor digitorum tendon or anomalous muscles in the carpal tunnel cause triggering at the wrist.3–5 In these cases, the deep transverse carpal ligament acted as the triggering point, and triggering was produced by finger flexion and
70
Mod Rheumatol Downloaded from informahealthcare.com by The University of Manchester on 10/29/14 For personal use only.
Fig. 1. Transverse section magnetic resonance imaging (MRI) of the wrist. Plain (A) and enhanced (B) MRI show diffuse synovial thickening around the flexor tendons
Fig. 2. Tenography of the flexor tendons. On wrist and finger extension (A), the expanded sheath of the flexor tendons and well-defined round free bodies (arrow) are visible. These free bodies moved proximally and caused the trigger on wrist and finger flexion (B)
extension, as described in our case. Other authors suggested the definition of a trigger wrist should only apply where triggering is specifically related to wrist movement.6,7 In this context, our patient did not present with true trigger wrist. However, this is the first report of trigger wrist involving SLE. It is true that the overlap with rheumatoid arthritis (RA) cannot be neglected totally, in which flexor tendon synovitis presents with a carpal tunnel syndrome and concomitant trigger finger.8 Our patient had monoarthritis in a wrist and did not fulfill the criteria of RA. Furthermore, histological examination of the excised synovial tissue showed nonspecific inflammation. The hand X-ray exhibited scapholunate dissociation leading to carpal collapse, which is one of the radiological features of deforming arthropathy in the hands of SLE patients.9 van Vugt described 17 patients with deforming arthropathy in 176 SLE patients, who were classified into three types of deforming arthropathy (rhupus hand, Jaccoud’s arthropathy, and mild deforming arthropathy) according to the evidence of erosive change and Jaccoud’s arthropathy
Fig. 3. Surgical exposure of the carpal tunnel. In the median nerve (stars), there were no abnormal findings. Arrow indicates the thickened flexor sheath (A). Flexor tendons were surrounded by synovium and rice bodies (B)
index. In this case, there was no deformation of the finger or wrist (ulnar deviation, swan neck deformities, boutonniere deformity, Z deformities, hallux valgus, or hammer toes), but there was severe tenosynovitis. Jaccoud’s arthropathy is characterized by severe deformities of the hand with little or no evidence of synovitis. According to these criteria, this case may be identified as mild deforming arthropathy. However, this is not the typical form. In this case, one of the possible explanations of mechanism of triggering is partial friction or laceration of the flexor tendon with this arthropathy, as reported previously.10 As a cause of severe flexor tenosynovitis, repeated forced hand movement has been suggested. Carpal tunnel syndrome, which is thought to be an overuse syndrome, is caused by a decrease in the volume of the carpal tunnel (arthritic spur or thickening of the flexor retinaculum) or an increase in the volume of its contents (fluid retention or
Mod Rheumatol Downloaded from informahealthcare.com by The University of Manchester on 10/29/14 For personal use only.
71
synovial proliferation). Flexor tenosynovitis associated with repeated application of force to the hand using a T-cane may be a contributing factor in our case. Rice body formation is most commonly observed in chronic RA accompanied by marked pannus formation and also is less frequently observed in seronegative arthropathy and chronic synovitis secondary to tuberculosis. It was thought that rice bodies arise from infarcted synovial cells. Histological findings of synovial lesion associated with SLE are usually mild and fibrous synovitis with little or no round cell infiltration. In contrast, the histopathological features of RA have been defined as multilayered synovial lining, abundant mononuclear cell infiltration, formation of lymphoid follicles, and fibrinoid degeneration. In this case, the patient had a severe flexor tenosynovitis in her wrist, which did not improve with treatment by splinting and corticosteroid injection around the flexor tendon. Histological features of the synovium in this case revealed nonspecific severe synovitis and rice body formation. These features are not typical in synovial lesions of SLE. Thus, it was thought that her flexor tenosynovitis was accelerated by the use of a T-cane. However, this is the first case of trigger wrist in SLE caused by a severe tenosynovitis around the flexor digitorum tendon. Furthermore, triggering and wrist swelling had disappeared at 2-year follow-up. Enhanced MR scan and a tenography had facilitated diagnosis, leading to a successful resolution of symptoms.
References 1. Tan EM, Cohen AS, Fries JF, Masi AT, McShane DJ, Rothfield NF, et al. The 1982 revised criteria for the classification of systemic lupus erythematosus. Arthritis Rheum 1982;25(11):1271–7. 2. Eibel P. Trigger wrist with intermittent carpal tunnel syndrome. A hitherto undescribed entity with report of a case. Can Med Assoc J 1961;84:602–5. 3. Brown LP, Coulson DB. Triggering at the carpal tunnel with incipient carpal-tunnel syndrome. Report of an unusual case. J Bone Joint Surg Am 1974;56(3):623–4. 4. Aghasi MK, Rzetelny V, Axer A. The flexor digitorum superficialis as a cause of bilateral carpal-tunnel syndrome and trigger wrist. A case report. J Bone Joint Surg Am 1980;62(1):134–5. 5. Kernohan JG, Benjamin A, Simpson D. Trigger wrist due to anomalous flexor digitorum profundus muscle in association with fibroma of tendon sheath. Hand 1982;14(1):59–60. 6. Desai SS, Pearlman HS, Patel MR. Clicking at the wrist due to fibroma in an anomalous lumbrical muscle: a case report and review of literature. J Hand Surg [Am] 1986;11(4):512–4. 7. Smith RD, O’Leary ST, McCullough CJ. Trigger wrist and flexor tenosynovitis. J Hand Surg [Br] 1998;23(6):813–4. 8. Carvell JE, Mowat AG, Fuller DJ. Trigger wrist phenomenon in rheumatoid arthritis. Hand 1983;15(1):77–81. 9. van Vugt RM, Derksen RH, Kater L, Bijlsma JW. Deforming arthropathy or lupus and rhupus hands in systemic lupus erythematosus. Ann Rheum Dis 1998;57(9):540–4. 10. Minami A, Ogino T. Trigger wrist caused by a partial laceration of the flexor superficialis tendon of the ring finger. J Hand Surg [Br] 1986;11(3):457–9.
© The Japan Rheumatism Association and Springer-Verlag Tokyo 2002
CASE REPORT
Kazuhiro Ohtani · Kanji Fukuda · Chiaki Hamanishi
Mod Rheumatol Downloaded from informahealthcare.com by The University of Manchester on 10/29/14 For personal use only.
A case of systemic lupus erythematosus associated with trigger wrist
Received: August 23, 2000 / Accepted: July 6, 2001
Abstract A 54-year-old woman complained of a painful, swollen, and clicking left wrist for 1 year. She had an 8-year history of systemic lupus erythematosus (SLE), treated with oral prednisolone. Flexion and extension of all fingers were difficult to initiate on wrist extension, and movement was accompanied by a palpable click at the wrist. Magnetic resonance imaging (MRI) and tenography revealed the expanded sheath of the flexor tendons and well-defined round free bodies known as rice bodies. Synovectomy and excision of rice bodies resulted in complete disappearance of swelling and triggering of the wrist. Key words Systemic lupus Tenosynovitis · Trigger wrist
erythematosus
(SLE)
·
Case report
difficult to initiate flexion and extension of all fingers on wrist extension, and movement was accompanied by a palpable click at the wrist. There were no symptoms or signs of median nerve compression, and there was no deformity in her fingers or wrist. X-ray examinations revealed slight scapholunate dissociation and atrophic changes of the carpus. Plain and enhanced magnetic resonance imaging (MRI) showed diffuse synovial thickening around the flexor tendons (Fig. 1). Tenography revealed the expanded sheath of the flexor tendons, and well-defined round free bodies were visible in the sheath. These free bodies moved proximally and caused the trigger on finger flexion (Fig. 2). Surgical exploration of the carpal tunnel showed a grossly thickened and congested synovial flexor sheath. There was no edematous alteration in the median nerve. All flexor tendons were surrounded by a hypertrophic synovial cuff to which were attached many nodular rice bodies, i.e., 1 ⫻ 0.5 cm of gross, yellow, soft mass (Fig. 3). Carpal tunnel decompression was completed, the rice bodies were excised, and synovectomy was carried out. The intraarticular lesions were not examined further because they were not thought to be concerned with the triggering phenomenon. Histological examination of the excised synovial tissue showed hyperplastic synovium with lymphocyte infiltration. The rice bodies were noted to consist of dense fibrinous material. One year postoperatively, the patient remains free of pain and swelling and triggering has disappeared, although there is some remaining limitation of wrist movement.
A 54-year-old, right-handed woman presented with a complaint of a painful, swollen, and clicking left wrist present for 1 year. She had an 8-year history of systemic lupus erythematosus (SLE) and fulfilled five criteria of the ACR classification1; i.e., malar rash, discoid rash, oral ulcers, hemolytic anemia, and antinuclear antibody. She was treated with oral administration of corticosteroid and controlled with medication of 5 mg/day prednisolone. She had undergone total hip arthroplasty and total knee arthroplasty for steroid-induced osteonecrosis of her left femoral head and right medial femoral condyle, respectively. She had been advised to walk with a T-cane in her left hand. Swelling and triggering of her left hand had been present for 1 year. On examination, there was a slight synovial thickening over the palmar aspect of the wrist. It was
Discussion
K. Ohtani · K. Fukuda (*) · C. Hamanishi Department of Orthopaedic Surgery, Kinki University School of Medicine, 377-2 Ohnohigashi, Osaka-sayama 589-8511, Japan Tel. ⫹81-723-66-0221 (ext. 3211); Fax ⫹81-723-67-7525 e-mail: [email protected]
Trigger wrist, first reported by Eibel in 1961, is a rare phenomenon.2 Previous reports described that nodules in the flexor digitorum tendon or anomalous muscles in the carpal tunnel cause triggering at the wrist.3–5 In these cases, the deep transverse carpal ligament acted as the triggering point, and triggering was produced by finger flexion and
70
Mod Rheumatol Downloaded from informahealthcare.com by The University of Manchester on 10/29/14 For personal use only.
Fig. 1. Transverse section magnetic resonance imaging (MRI) of the wrist. Plain (A) and enhanced (B) MRI show diffuse synovial thickening around the flexor tendons
Fig. 2. Tenography of the flexor tendons. On wrist and finger extension (A), the expanded sheath of the flexor tendons and well-defined round free bodies (arrow) are visible. These free bodies moved proximally and caused the trigger on wrist and finger flexion (B)
extension, as described in our case. Other authors suggested the definition of a trigger wrist should only apply where triggering is specifically related to wrist movement.6,7 In this context, our patient did not present with true trigger wrist. However, this is the first report of trigger wrist involving SLE. It is true that the overlap with rheumatoid arthritis (RA) cannot be neglected totally, in which flexor tendon synovitis presents with a carpal tunnel syndrome and concomitant trigger finger.8 Our patient had monoarthritis in a wrist and did not fulfill the criteria of RA. Furthermore, histological examination of the excised synovial tissue showed nonspecific inflammation. The hand X-ray exhibited scapholunate dissociation leading to carpal collapse, which is one of the radiological features of deforming arthropathy in the hands of SLE patients.9 van Vugt described 17 patients with deforming arthropathy in 176 SLE patients, who were classified into three types of deforming arthropathy (rhupus hand, Jaccoud’s arthropathy, and mild deforming arthropathy) according to the evidence of erosive change and Jaccoud’s arthropathy
Fig. 3. Surgical exposure of the carpal tunnel. In the median nerve (stars), there were no abnormal findings. Arrow indicates the thickened flexor sheath (A). Flexor tendons were surrounded by synovium and rice bodies (B)
index. In this case, there was no deformation of the finger or wrist (ulnar deviation, swan neck deformities, boutonniere deformity, Z deformities, hallux valgus, or hammer toes), but there was severe tenosynovitis. Jaccoud’s arthropathy is characterized by severe deformities of the hand with little or no evidence of synovitis. According to these criteria, this case may be identified as mild deforming arthropathy. However, this is not the typical form. In this case, one of the possible explanations of mechanism of triggering is partial friction or laceration of the flexor tendon with this arthropathy, as reported previously.10 As a cause of severe flexor tenosynovitis, repeated forced hand movement has been suggested. Carpal tunnel syndrome, which is thought to be an overuse syndrome, is caused by a decrease in the volume of the carpal tunnel (arthritic spur or thickening of the flexor retinaculum) or an increase in the volume of its contents (fluid retention or
Mod Rheumatol Downloaded from informahealthcare.com by The University of Manchester on 10/29/14 For personal use only.
71
synovial proliferation). Flexor tenosynovitis associated with repeated application of force to the hand using a T-cane may be a contributing factor in our case. Rice body formation is most commonly observed in chronic RA accompanied by marked pannus formation and also is less frequently observed in seronegative arthropathy and chronic synovitis secondary to tuberculosis. It was thought that rice bodies arise from infarcted synovial cells. Histological findings of synovial lesion associated with SLE are usually mild and fibrous synovitis with little or no round cell infiltration. In contrast, the histopathological features of RA have been defined as multilayered synovial lining, abundant mononuclear cell infiltration, formation of lymphoid follicles, and fibrinoid degeneration. In this case, the patient had a severe flexor tenosynovitis in her wrist, which did not improve with treatment by splinting and corticosteroid injection around the flexor tendon. Histological features of the synovium in this case revealed nonspecific severe synovitis and rice body formation. These features are not typical in synovial lesions of SLE. Thus, it was thought that her flexor tenosynovitis was accelerated by the use of a T-cane. However, this is the first case of trigger wrist in SLE caused by a severe tenosynovitis around the flexor digitorum tendon. Furthermore, triggering and wrist swelling had disappeared at 2-year follow-up. Enhanced MR scan and a tenography had facilitated diagnosis, leading to a successful resolution of symptoms.
References 1. Tan EM, Cohen AS, Fries JF, Masi AT, McShane DJ, Rothfield NF, et al. The 1982 revised criteria for the classification of systemic lupus erythematosus. Arthritis Rheum 1982;25(11):1271–7. 2. Eibel P. Trigger wrist with intermittent carpal tunnel syndrome. A hitherto undescribed entity with report of a case. Can Med Assoc J 1961;84:602–5. 3. Brown LP, Coulson DB. Triggering at the carpal tunnel with incipient carpal-tunnel syndrome. Report of an unusual case. J Bone Joint Surg Am 1974;56(3):623–4. 4. Aghasi MK, Rzetelny V, Axer A. The flexor digitorum superficialis as a cause of bilateral carpal-tunnel syndrome and trigger wrist. A case report. J Bone Joint Surg Am 1980;62(1):134–5. 5. Kernohan JG, Benjamin A, Simpson D. Trigger wrist due to anomalous flexor digitorum profundus muscle in association with fibroma of tendon sheath. Hand 1982;14(1):59–60. 6. Desai SS, Pearlman HS, Patel MR. Clicking at the wrist due to fibroma in an anomalous lumbrical muscle: a case report and review of literature. J Hand Surg [Am] 1986;11(4):512–4. 7. Smith RD, O’Leary ST, McCullough CJ. Trigger wrist and flexor tenosynovitis. J Hand Surg [Br] 1998;23(6):813–4. 8. Carvell JE, Mowat AG, Fuller DJ. Trigger wrist phenomenon in rheumatoid arthritis. Hand 1983;15(1):77–81. 9. van Vugt RM, Derksen RH, Kater L, Bijlsma JW. Deforming arthropathy or lupus and rhupus hands in systemic lupus erythematosus. Ann Rheum Dis 1998;57(9):540–4. 10. Minami A, Ogino T. Trigger wrist caused by a partial laceration of the flexor superficialis tendon of the ring finger. J Hand Surg [Br] 1986;11(3):457–9.
