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Recurrent Focal Myositis Presenting as Flexion Contractures of the FingersWestmead Hospital, Sydney, New South Wales, Australia E-mail: bsnell{at}gmp.usyd.edu.au Dear Sir, A 19 year-old male with no significant previous medical history presented with progressive contractures of the left middle and ring fingers, which had developed over a 3-year period. There was no history of trauma and he did not recall a family history of neurological, musculoskeletal or connective tissue diseases. On examination, the range of motion of the proximal interphalangeal (PIP) joints of the middle and ring fingers was +60°/+120° with the wrist in the neutral position. PIP extension improved with wrist flexion and worsened with wrist extension. MRI of the forearm showed a rim of hyperintense signal on the superficial margin of the flexor compartment, predominantly in the belly of the flexor digitorum superficialis muscle. Surgical exploration of the left forearm revealed patchy fibrosis of the radial side of the muscle belly of flexor digitorum superficialis and an anomalous belly of this muscle extending from the radial side of the normal muscle belly to the mid radius. No other muscles of the flexor compartment were affected. Excision of the affected tissue of flexor digitorum superficialis was undertaken. The histology of the excised muscle showed scattered inflammatory infiltrates with both hypertrophic and atrophic fibres. There were large areas of interstitial fibrosis with scattered areas of basophilic regenerating fibres. This histology was consistent with a diagnosis of focal myositis. Initially, he had a satisfactory clinical outcome, but re-presented 6 months later with recurrence of disease, manifesting in the same manner but now affecting all of the flexor digitorum superficialis tendons. The recurrent disease began to manifest itself around 14 weeks after the initial operation, initially in the middle and ring fingers, followed shortly after by flexion deformities of the index and little fingers. The recurrent disease was severe, with flexion deformities of 90° of all four PIP joints when the wrist was in neutral position. On this occasion, excision of the entire flexor digitorum superficialis muscle belly was performed. On follow-up at 3 months, other than a +10° extension deficit of the PIP joint of the middle finger, all of the fingers were straight when the wrist was in the neutral position. In full finger flexion, the fingertip to distal palmar crease distance was 2 cm for all four fingers. Due to social factors, the patient was subsequently lost to further follow-up. Focal myositis was first described by Heffner et al. (1977) as a non-malignant, pseudotumour of skeletal muscle which often presents as a painful soft tissue mass in one limb, most commonly the lower limb, with no associated systemic pathology (Finger and Dennis, 1995; Kisielinski et al., 2002; Manganelli et al., 2002). A case in the hand has previously been described, presenting as a painful mass (Maguire et al., 1988). In the current case, however, the patient presented with a functional deficit of his hand and without the typical, painful soft tissue mass. To our knowledge, there have been no previous cases, which have presented in this manner. Whilst spontaneous regression have been reported (Manganelli et al., 2002), surgical resection of the affected muscle tissue has usually been necessary and has mostly resulted in a favorable clinical outcome (Gordon and Madhok, 1999).
Finger D, Dennis G (1995). Focal myositis. Journal of Rheumatology, 22: 188–189.[Web of Science][Medline] [Order article via Infotrieve] Gordon M, Madhok R (1999). Recurrent focal myositis. Rheumatology, 38: 1295–1296. Heffner RV, Armbrustmacher VW, Earle KM (1977). Focal myositis. Cancer, 40: 301–306.[CrossRef][Web of Science][Medline] [Order article via Infotrieve] Kisielinski K, Miltner O, Sellhaus B, Kruger S, Goost H, Siebert CH (2002). Recurrent focal myositis of the peroneal muscles. Rheumatology, 41: 1318–1322. Maguire JK, Milford LW, Pitcock JA (1988). Focal myositis of the hand. Journal of Hand Surgery, 13A: 140–142.[CrossRef][Medline] [Order article via Infotrieve] Manganelli S, De Stefano R, Malandrini A, Selvi E, Frati E, Gambelli S, Marcolongo R (2002). Bilateral recurrent focal myositis of gastrocnemius muscles after BCG vaccination. Rheumatology, 41: 1074–10765.
Journal of Hand Surgery (European Volume), Vol. 32, No. 6,
714 (2007)
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