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DOI: 10.1016/j.jhsb.2003.11.001
Tumoral Calcinosis Infiltrating the Biceps Brachii Tendon – Excision and Reconstruction with Allograft: A Case ReportFrom the Section of Hand Surgery, Department of Plastic and Reconstructive Surgery, and the Department of Pathology, Istanbul Faculty of Medicine, Istanbul University, Istanbul, Turkey Correspondence: Atakan Aydin, MD, Orhantepe Mah., Kampyolu Sok, Palmiye Apt. No:17 D:5 Dragos Kartal, 81540 Istanbul, Turkey.; E-mail: atakanaydin{at}yahoo.com
Two patients with limited elbow motion due to tumoral calcinosis underwent resection of calcified masses around the elbow joints. This necessilated removal of the distal two thirds and three fourths of the biceps muscles. The resulting defects were reconstructed with homologous Achilles tendon grafts. Both patients regained M4 muscle power and no recurrences were observed.
Key Words: tumoral calcinosis • excision • allograft reconstruction
Tumoral calcinosis is a disorder with an obscure pathology characterized by deposition of calcium in periarticular areas, mainly around the hip, shoulder and elbow. Tumoral calcinosis presents with irregular "tumours" containing either a milky fluid, a white gritty, putty-like material or solid calcified masses. The condition has several alternative names, including lipocalcinogranulomatosis, lipocalcinogranulomatous bursitis, tumoral lipocalcinosis, calcified bursae, and calcifying collagenolysis (Hacihanefio  lu, 1978). There are usually no demonstrable abnormalities in calcium metabolism, though hyperphosphatemia (Abraham et al., 1996; Clarke et al., 1984; Demiryont et al., 1999) and elevated serum 1,25-dihydroxyvitamin D levels (Slavin et al., 1993) have been reported in some cases. The lesions of tumoral calcinosis are characterized clinically by the presence of irregular, painless, periarticular soft tissue calcifying masses (Harwood et al., 1996). Although the underlying joints are unaffected (Weiss and Goldblum, 2001), the lesions may extend into adjacent muscles and tendons (Smack et al., 1996; Savaci et al., 2000). Surgical excision is the preferred method of treatment, but recurrence is common, especially after incomplete removal. Radical excision may necessitate the excision of important periarticular structures.
Case 1 A 25-year-old lady had masses containing milky fluid around her ankles, elbows and buttocks, and in her thoracic and abdominal walls. They had been present since adolescence, and she had been diagnosed with and treated for tuberculosis, collagenosis, and other conditions. On physical examination, she had bilateral firm calcified masses predominantly around her knee and elbow joints (Fig 1). She had restricted elbow movements, especially extension. Blood levels for calcium, phosphate and vitamin D were normal. Biceps muscle invasion by the calcified masses was obvious on plain radiographs and magnetic resonance images. The patient underwent surgery and the subcutaneous lesions around the knees were excised. Exploration of both elbows and upper arms revealed involvement of the biceps muscles and tendons. The distal two-thirds of the left biceps muscle and tendon was affected by the calcinosis and had to be excised (Fig 1). The resulting defect was then reconstructed with homologous Achilles tendon graft (Biodynamics International, Germany). The graft was interwoven with the remaining biceps tendon distally, while proximally it was embedded in the biceps muscle (Fig 1). The anterior one-third of the right biceps muscle was also involved and resected, leaving the posterior parts of the muscle and tendon intact. Postoperatively, both arms were placed in a long arm cast for 4 weeks, after which physical therapy was commenced. Excellent elbow range of motion and M4 muscle power was achieved bilaterally (Fig 2). Histopathological examination revealed foci of calcification within fibrous tissue (Fig 3). No recurrences occurred during a 6-year follow-up period.
Case 2 A 20-year-old lady with lesions which discharged calcified material around the knee and elbow joints was referred to our clinic. Blood tests revealed normal calcium, phosphate and vitamin D levels. She underwent resection of the lesions which required excision of the anterior one-third of her right biceps muscle and the full thickness of distal three-quarters of her left biceps. The left biceps muscle was reconstructed with a homologous Achilles tendon graft. Postoperatively, both arms were placed in a long arm cast for 4 weeks, after which passive and active motion exercises commenced. At the end of physical rehabilitation, she had full elbow motion and M4 muscle power bilaterally. There was no recurrence during the 2-year follow-up.
Tumoral calcinosis is thought to be an exaggerated reparative reaction to bleeding after an injury which may be trivial. It results in the formation of synovial-like cavities and calcified masses. The disease occurs in about one-third of cases as a result of a genetic defect in the regulation of 25-hydroxy-1-alpha-hydroxylase in the kidney: this enzyme participates in vitamin D metabolism. It has also been reported in patients of varying ages who do not have hyperphosphatemia, but do have elevated 1,25-dihydroxyvitamin D levels. It also occurs in patients undergoing dialysis for renal failure (Slavin et al., 1993). Despite advances in the understanding of the etiology of the disease, prevention therapies generally fail and the treatment of tumoral calcinosis can be difficult. Treatment with various drug regimens and radiotherapy does not prevent recurrence, but diets which are low in phosphate and calcium and contain phosphate-binding antacids have been successful in some patients. Surgery is the preferred treatment, especially if the lesions are surrounded by a pseudocapsule. However, if the lesions extend as finger-like projections into adjacent tissues, complete surgical excision is extremely difficult. Tumoral calcinosis lesions demonstrate three stages. Stage 1 is the cellular phase and is characterized by foamy macrophage aggregates. In Stage 2 calcification occurs while Stage 3 is characterized by the presence of inactive calcified lesions. Surgical intervention in the second stage is difficult and incomplete resection commonly occurs as these lesions are poorly circumscribed and progressive. Stage 3 lesions are circumscribed and relatively quiescent, and these are easier to excise and less prone to recurrence. The underlying joints are normal in tumoral calcinosis, but the lesions tend to attach firmly to fascia, muscle and tendon (Weiss and Goldblum, 2001). Thus excision of advanced lesions may necessitate sacrifice of important periarticular structures, such as the biceps muscle and tendon as in our cases. Reconstruction of the biceps tendon with cadaveric Achilles tendon allografts has been described with satisfactory results (Sanchez-Sotelo et al., 2002), and we have achieved good outcomes and ranges of elbow motion despite using grafts that were 20 cm long.
Received for publication July 8, 2003. Accepted for publication November 3, 2003.
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Abstract
INTRODUCTION
lu U, Ertosun S (1999). Tumoral calcinosis (a clinopathologic study of 17 cases). The Turkish Journal of Pathology, 15: 32–34.