Abstract
A recurrence of a juxtacortical chondroma of the finger after marginal excision prompted us to review the treatment of this condition. Although the recommended treatment is simple curettage or marginal excision, the reported recurrence rate is significantly higher for lesions in the hand than those in other locations and recurrences only occurred in patients who had local treatments which did not include excision of the adjacent bone cortex.
We report five patients with juxtacortical chondroma of the fingers. The first patient underwent marginal excision without resection of the underlying bone cortex. The other four patients underwent intralesional, marginal or wide excisions of tumour with resection of the bone cortex underlying the lesion. Recurrence was only seen in the patient who did not undergo resection of the bone cortex. Resection of the underlying bone cortex after excision of this tumour may be advisable for the treatment of this tumour in the hand to reduce the rate of recurrence.
INTRODUCTION
Juxtacortical chondroma of bone (Jaffe, 1956) was originally described as “periosteal chondroma” (Lichtenstein and Hall, 1952). It is a rare, slowly growing benign cartilaginous tumour that arises on the surface of bone. Two hundred and six cases have been reported (Bauer et al., 1982; Boriani et al., 1983; Cooke and Pearce, 1976; Dan et al., 1969; DeSantos and Spjut, 1981; Duran et al., 1975; Fornasier and McGonigal, 1977; Freiberg and Wilber, 1956; Gahhos and Cuono, 1984; Greenspan and Unni, 1993; Herrmann and Goth, 1981; Holder and Garna, 1987; Ishida et al., 1998; Jaffe, 1956; Kirchner et al., 1978; Le Saout et al., 1987; Lewis et al., 1990; Lichtenstein and Hall, 1952; Lorente et al., 2000; Marmor, 1964; Matsumori et al., 1966; Mora et al., 1988; Morisaki et al., 1996; Nojima et al., 1985; Nosanchuk and Kaufer, 1969; Pazzaglia and Ceciliani, 1985; Peidro et al., 1996; Rankin et al., 1986; Ricca et al., 2000; Rockwell et al., 1972; Savornin and Foult, 1989; Sinha et al., 1999; Spagnoli et al., 1995; Szendröi et al., 1990; Tillich et al., 1998; Varma et al., 1991; Wheelhouse and Griffin, 1982; Woertler et al., 2001; Wu, 1992). The metaphysis of a long tubular bone is usually involved, and the finger phalanx is one of the most commonly affected sites. The location of the tumours was reported in 183 of the 206 reported cases: it was located in the finger phalanx in 51, the femur in 49, the humerus in 39 and the tibia in 24 patients. The tumour may erode the underlying bone cortex without actually penetrating the medullary cavity. Intralesional curettage or marginal excision, viz. complete excision with a minimal margin of macroscopically normal tissue around the tumour, is generally recommended, and the recurrence rate has been described as very low (Bauer et al., 1982; Boriani et al., 1983). However, we have experienced a recurrent case of juxtacortical chondroma of the proximal phalanx of the thumb after simple marginal excision and literature review revealed a higher recurrence rate for lesions in the hand after intralesional or marginal excision.
This paper reports the authors’ surgical method of preventing recurrence of juxtacortical chondroma of the hand.
PATIENTS AND METHODS
Literature review
We reviewed cases reported in the literature, comparing the surgical outcome of lesions in the hand and other locations. Surgical procedures were described in 126 reported cases, of which 39 were in the hand (Boriani et al., 1983; Cooke and Pearce, 1976; Dan et al., 1969; DeSantos and Spjut, 1981; Fornasier and McGonigal, 1977; Freiberg and Wilber, 1956; Herrmann and Goth, 1981; Kirchner et al., 1978; Le Saout et al., 1987; Lichtenstein and Hall, 1952; Lorente et al., 2000; Marmor, 1964; Matsumori et al., 1966; Mora et al., 1988; Nojima et al., 1985; Nosanchuk and Kaufer, 1969; Pazzaglia and Ceciliani, 1985; Peidro et al., 1996; Ricca et al., 2000; Rockwell et al., 1972; Savornin and Foult, 1989; Sinha et al., 1999; Szendröi et al., 1990; Varma et al., 1991; Wheelhouse and Griffin, 1982; Lewis et al., 1990). We classified the surgical procedures into four categories: (1) intralesional excision without resection of the underlying bony cortex; (2) marginal excision without resection of the underlying bony cortex; (3) intralesional or marginal excision with resection of the underlying bony cortex; (4) wide excision. The lesions in the hand were treated by marginal excision without resection of the underlying bony cortex in 30 (77%) cases, intralesional or marginal excision with resection of the underlying cortex in eight (21%) cases and wide excision in 1 (3%) case.
Clinical series
The clinical records, radiographs, histological findings, surgical findings and hand function of our five patients were evaluated and are presented in Table 1. The diagnosis of juxtacortical chondroma was, ultimately, based on the clinical, X-ray and pathological findings.
There were three men and two women in our study, ranging in age from 7 to 70 (mean 26.5) years. All of the tumours occurred in the tubular bones of the hand, viz. the proximal phalanx of the thumb, the middle phalanx of the ring finger, the proximal phalanx of the ring finger, the middle phalanx of the little finger and the metacarpal of the little finger. Swelling without pain was the complaint in all patients. The duration of the symptoms before surgery ranged from 4 months to 13 years (mean 5 years 6 months). One patient had sustained a fracture at the lesion site 13 years before presentation but there was no evidence that this was a pathological fracture.
In all of our patients, radiographs revealed the typical scalloping of the adjacent bone cortex (Fig 1), the presence of calcification (Fig 2), and a soft tissue mass (Fig 3). MRI was available in two patients, and T1-weighted MR images revealed a homogeneously low signal intensity lesion (Fig 4), and T2-weighted MR images showed a homogeneously bright signal intensity lesion (Fig 5).
The first patient underwent marginal excision without excision of the underlying cortex. Three of the others were treated by marginal or intralesional excision with resection of the underlying bone cortex and one by wide excision including the adjacent bone cortex. Bone graft was added in one patient because of concern that a postoperative fracture might occur. The mean follow-up period was 7 (range 2–12) years. The physical findings, plain radiographs and functional results were compared to those of the contralateral upper limb according to Enneking’s scoring system at the final follow-up appointment (Enneking et al., 1993).
RESULTS
Literature review
Recurrence of juxtacortical chondroma after initial surgery was reported in seven patients (Mora et al., 1988; Nojima et al., 1985; Nosanchuk and Kaufer, 1969; Rockwell et al., 1972; Varma et al., 1991). Two cases had undergone intralesional excisions, and five had undergone marginal excisions as the initial procedure. In patients who had undergone intralesional or marginal excision with resection of the underlying cortex or wide excision, no recurrence was reported. The overall rate of recurrence was 6%. The reported rate of recurrence in the tubular bones of the hand was 15% (6 cases), compared with 1.2% (one case) for lesions elsewhere. This difference was statistically significant (χ2 for independence test: P<0.05).
Our series
Macroscopically, all of the tumours were blueish-white or yellowish-white cartilaginous lesions which were firm or hard, and, occasionally, had a definitive fibrous capsule. Their diameters ranged in size from 2.0 to 2.5 cm. Histologically, proliferation of lobulated hyaline cartilage within a fibrous capsule was seen. Focal calcification was observed in some areas. There was no cellular atypia or mitotic activity.
No recurrences have been observed during follow-up of four of the patients. The first patient, who underwent a marginal excision without resection of the adjacent cortex, had a recurrence. He declined further surgery.
The functional results, according to Enneking’s scoring system, were 100% in all of the patients, including the one with recurrence.
DISCUSSION
While assessment of recurrence rates from previous publications is not absolutely reliable, because of differences in length of follow-up and surgical techniques used, the reported recurrence rate would suggest a significantly higher rate for lesions in the hand than those in other locations. The recommended treatment for juxtacortical chondroma is simple curettage or marginal excision. Nosanchuk and Kaufer (1969) considered recurrence to be due to incomplete excision of the tumour or the formation of a new identical tumour. Jaffe has reported one tumour sticking to the adjacent bone cortex (Jaffe, 1956). We believe that incomplete excision of the adjacent bone cortex is probably the cause of local recurrence. It is also possible that, in some instances, the surgeon could not excise the lesion completely without restricting finger function. We have experienced no recurrence when the adjacent full thickness of the bone cortex underlying the lesion is excised. Although the number of the patients in this series was very small, we believe that this is a sensible surgical option.
The three major radiographic features of juxtacortical chondroma are: (1) scalloping or remodelling of the adjacent bony cortex, (2) the presence of a calcification and (3) the presence of a soft tissue mass (Cooke and Pearce, 1976; Jaffe, 1956; Kirchner et al., 1978). These features enable one to distinguish the lesion from the more common enchondroma and all were observed in our patients. Occasionally, some reactive periosteal new bone formation is seen and computed tomography can demonstrate indolent features of this benign lesion such as an unbroken cortex. MR imaging confirms that the marrow cavity is intact and can also assist the pre-operative diagnosis by demonstrating a subperiosteal mass with the typical features of a cartilaginous tumour (Varma et al., 1991).
Histologically, the lesion is a benign cartilaginous tumour. Atypical nuclei may be seen in some cases, including binucleated cells (Bauer et al., 1982; Boriani et al., 1983; Nojima et al., 1985; Nosanchuk and Kaufer, 1969). Care should be taken not to inadvertently diagnose a malignant tumour: this lesion can appear similar to a low-grade chondrosarcoma, but extension of the tumour into the marrow cavity is not present.