http://informahealthcare.com/mor ISSN 1439-7595 (print), 1439-7609 (online) Mod Rheumatol, 2014; Early Online: 1–4 © 2014 Japan College of Rheumatology DOI: 10.3109/14397595.2014.924193

CASE REPORT

Spontaneous flexor tendon rupture in systemic lupus erythematosus: A case report

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Ryo Oda1, Hiroyoshi Fujiwara1, Daisaku Tokunaga1, Aiko Kishida1, Daigo Taniguchi1, Takahiro Seno2,3, Yutaka Kawahito2,3, and Toshikazu Kubo1,3 1Department of Orthopaedics, Graduate School of Medical Science, Kyoto Prefectural University of Medicine, Kyoto, Japan, 2Inflammation and Immunology, Graduate School of Medical Science, Kyoto Prefectural University of Medicine, Kyoto, Japan, and 3Department of Rheumatic Diseases and Joint Function, Graduate School of Medical Science, Kyoto Prefectural University of Medicine, Kyoto, Japan

Abstract Spontaneous flexor tendon rupture is an unusual complication of systemic lupus erythematosus (SLE) and has not previously been reported. While tendon ruptures in association with SLE have been focused on the previous studies, upper extremity tendon ruptures are infrequently reported in the literature. Here, we present an uncommon case of spontaneous flexor tendon rupture of the ring and little fingers in a patient with SLE and discuss the mechanism of injury and its surgical treatment.

Keywords Flexor tendon rupture, Hook of hamate, Jaccoud’s arthropathy, Steroid, Systemic lupus erythematosus History Received 7 August 2013 Accepted 6 May 2014 Published online 19 June 2014

Case The patient was a 68-year-old homemaker who developed SLE at 24 years of age and who had been treated with 7.5–30 mg of oral prednisolone daily for 15 years. She had noticed impairment in active flexion of her right little finger without any distinct cause. Two years later, she noticed impairment in active flexion of her right ring finger and presented to our orthopedic department. The initial examination showed limited range of motion (ROM) in active flexion of the distal interphalangeal (DIP) joint of the right ring and little fingers when the proximal interphalangeal (PIP) joint and the metacarpophalangeal (MP) joint were in extension. Active flexion of the PIP joint of the right little finger was also limited when the other fingers were in full extension. A physical examination revealed no swelling or redness in her right palm. Total active motion (TAM) on the affected side was 67% of the unaffected side in the ring finger and 57% in the little fingers, respectively. Grip strength was 12.3 kg on the right side and 13.1 kg on the left side, and the Disabilities of the Arm, Shoulder and Hand (DASH) scores were 37 points for the disability/symptom scale which ranges from 0 (no disability) to 100. Laboratory results revealed a white blood cell (WBC) count of 4900/μl, C-reactive protein of 0.04 mg/dl, and the following titers were measured: anti-cyclic citrullinated peptide antibodies, 0.6 U/ml; anti-double-stranded DNA antibodies, 320 x; anti-Smith antigen, 7.0 U/ml; complement 3, 50.4 mg/ dl; and complement 4, 12.1 mg/dl. Ulnar deviation without bone erosion, typically seen in Jaccoud’s arthropathy, was not observed. However, there was a marked osteoarthritic change with bone Correspondence to: Ryo Oda, MD, PhD, Department of Orthopaedics, Graduate School of Medical Science, Kyoto Prefectural University of Medicine, Kajii-cho, 465, Kamigyo-ku, Kyoto City, Kyoto Prefecture, Japan. Tel: ⫹ 81-75-251-5549. Fax: ⫹ 81-75-251-5841. E-mail: roda@ koto.kpu-m.ac.jp

destruction at the CM joint of both thumbs. Osteoarthritic change was also seen at the DIP joint of the both index fingers, and the DIP joint of the left middle, ring, and little fingers in those joints as well as in the IP and MP joints of the bilateral thumbs (Figure 1). The patient exhibited radial deviation of the right thumb at the IP joint and had undergone IP joint arthrodesis 6 years prior to presentation at our department. There was a severe swan-neck deformity in the left thumb. No instability was detected in the wrists and fingers. In ultrasound and magnetic resonance (MR) images, the distal stump of the flexor digitorum profundus (FDP) tendon of the ring finger was observed at the proximal portion of the 4th metacarpal head. Rupture of the FDP tendon and the flexor digitorum superficialis (FDS) tendon of the little finger was observed at the proximal portion of the 5th metacarpal head (Figure 2). Surgical findings showed that the FDS tendon of the ring finger was incompletely ruptured, and the FDP tendon of the ring finger was completely ruptured. Both FDP and FDS tendons of the little finger were completely ruptured. Flexor tendons were degenerated significantly around the rupture areas, and they had adhered to the surrounding tissue; the proximal stumps had retracted. Ruptured stumps of the FDP tendon of the ring finger and both FDP and FDS tendons of the little finger were observed approximately 2 cm proximal to the A1 pulley (Figure 3). Degenerated synovium was evident in the carpal tunnel, and eburnation was visible at the hook of the hamate. During surgery, the proximal stump of the FDS tendon of the ring finger was transferred to the distal stump of the FDP tendon of the same finger. Using interlacing sutures, the resected FDS tendon of the little finger was able to be used as a tendon graft to form a bridge between the distal stump of the FDP tendon of the little finger and the FDS tendon of the ring finger (Figure 4). Pathological findings related to the resected stumps of the ruptured tendons are described below. Villous changes and hyalinization were observed in some areas of the synovial tissue,

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Figure 1. (a) Preoperative radiograph of the bilateral hand. (b) The affected hand with fingers in flexion. (c) The affected hand with fingers in extension.

along with slight hyperplasia of the synovial lining cells, fibrin deposition, stromal fibrosis, and vascular hyperplasia. However, there was no conspicuous infiltration of inflammatory cells and no rheumatoid nodule formation. These findings were consistent with the diagnosis of SLE. Although there was no notable active inflammation, synovitis associated with SLE was evident. The postoperative regimen included a dorsal extension block splint beginning the day after surgery and early mobilization to prevent adhesion of the tendon graft. The extension block splint was applied with the wrist in 45 degrees flexion, the MP joints in 20 degrees flexion, and the PIP and DIP joints in neutral. The patient performed ROM and place/hold exercises, as well as passive flexion and active extension of her ring and little fingers. Five weeks after the surgery, the patient was permitted to begin active motion and blocking exercises, mainly of the ring and little fingers. On the 9th week after surgery, the patient performed activities of daily living (ADL) using kitchen knives and pans. The pulp-topalm distance was improved from 38 mm before surgery to 10 mm 5 months after surgery for the ring finger and from 20 mm to 0 mm, respectively, for the little finger. These outcomes resulted in an improvement in the patient’s ADL (Figure 5). For 2 months after surgery, the patient wore a dorsal extension block splint only at night. Fourteen months after surgery, postop-

erative TAM increased to 73% of the unaffected hand in the ring finger and 85% in the little finger. Grip strength was 16.4 kg, and the DASH scores were 17.2 points for the disability/symptom scale which indicated improvement. Based on these results, the patient was able to satisfactorily perform active flexion of the ring and little fingers, and to perform ADL without any problems.

Discussion Subcutaneous tendon rupture is an unusual complication of SLE. In the previous studies of tendon ruptures in SLE, most cases have focused on Achilles tendon or patellar tendon ruptures [1–4]. Only a few reports have focused on upper extremity tendon ruptures, predominantly of the extensor tendon of the hands [2–6]. We have previously reported a case of spontaneous rupture of the extensor digitorum communis tendon in a patient with SLE and the associated mechanism of injury [7]. Ulnar deviation occurred due to Jaccoud’s arthropathy in association with SLE. Both ulnar subluxation of the extensor tendon and tendon rupture occurred. We considered that the causes of tendon rupture were mechanical stress during tendon gliding, increased vulnerability of the tendon due to SLE, and steroid treatment for SLE. In the case of our present study, the degeneration and rupture of the

Figure 2. The upper panels show T2-weighted MR images of the flexor tendon rupture. (a) Coronal view of the right hand and (b, c, d) sagittal views of the right hand. The lower panels show (e) a longitudinal ultrasound image of the ring finger and (f) a longitudinal ultrasound image of the little finger with their respective schemas.

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DOI 10.3109/14397595.2014.924193

Spontaneous flexor tendon rupture in systemic lupus erythematosus 3

Figure 2. (Continued).

flexor tendon occurred proximal to the hook of the hamate. One possible cause of rupture could be the same mechanical stress that commonly causes subcutaneous rupture of the flexor tendon that is not associated with an underlying disease [8]. Flexor tendons can progressively degenerate and eventually rupture as a result of long-term stress [9]. After trauma or a stress fracture

Figure 3. Intraoperative photograph showing the ruptured FDS/FDP tendons of the ring and little fingers.

of the hook of the hamate, complications such as nonunion can occur; this injury is sometimes observed in baseball players. Nonunion can be another cause of increased mechanical stress on the ring and middle fingers [10,11]. The patient in this study

Figure 4. Tendon transfer and grafting were performed using the FDS tendon of the little finger. The course of the grafted tendon remained in an approximately anatomical position.

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There are fewer reports of SLE-related tendon rupture compared with that of RA-related tendon rupture. This difference can be explained by the low prevalence of SLE compared with that of RA (0.01–0.10% vs. 0.5–1.0% for RA) such that RA is ten times more prevalent than SLE. In addition, there is a difference in the pathology between RA and SLE. RA is characterized by aggressive synovitis with bone destruction, whereas gradual chronic inflammation occurs without bone lesions in patients with SLE. Reconstructive procedures for the flexor tendon rupture in the little finger involves end-to-side or side-to-side suture of the distal stump of the ruptured tendon to the adjacent tendon on the radial side [13]. In this case, the distal stump of the FDP tendon of the little finger was sutured to the FDS tendon of the ring finger using a bridge graft of a freed FDS tendon, and it was functioning well. We have reported an unusual case of spontaneous tendon rupture of the ring and small fingers in a patient with SLE. The mechanism of injury and surgical treatment of the flexor tendon rupture were also discussed. Periarthritis associated with SLE and long-term oral corticosteroid therapy appeared to have caused degeneration and increased vulnerability of flexor tendons, contributing to the rupture. In addition, mechanical stress in the vicinity of the hook of the hamate may also have contributed to the rupture. Tendon transfer and tendon grafting were an effective treatment for flexor tendon rupture.

Conflict of interest None.

References

Figure 5. Photographs of finger ROM and the performance of activities of daily living 5 months after surgery. (a) Extension of fingers. (b) Flexion of fingers. (c) Using a knife.

did not have a work history that involved overuse of the hands or a history of trauma to the hand. High-dose, long-term, oral corticosteroid therapy could potentially be a cause of degeneration and increase the vulnerability of the flexor tendon. Jaccoud’s arthropathy in patients with SLE is characterized by degeneration of soft tissue mainly due to periarthritis [3]. Thus, the tendons of the patient in this report could have become increasingly vulnerable. In patients with rheumatoid arthritis (RA), spontaneous tendon rupture in the hand commonly occurs. The mechanism of tendon rupture involves tendon degeneration due to tenosynovitis or mechanical stress. Mechanical stress can be caused by instability of the carpal bones or the distal radioulnar joint, or by bony spurs with joint destruction [12]. Prominent synovitis is an infrequent complication of SLE. However, spontaneous tendon rupture, similar to what is known to occur in RA cases, can be anticipated due to increased vulnerability of the tendon caused by fibrinoid degeneration and mechanical stress [4]. There have not been any reports on flexor tendon rupture in patients with SLE.

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Spontaneous flexor tendon rupture in systemic lupus erythematosus: A case report.

Spontaneous flexor tendon rupture is an unusual complication of systemic lupus erythematosus (SLE) and has not previously been reported. While tendon ...
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