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Linear morphoea with spontaneous fractures

SIR, A 4-year-old girl was sent to our hospital in March 2006 because of skin changes on her right leg which had been noticed by her parents in January 2004. Approximately 1 year after the appearance of these changes, in December 2004, she had sustained a fracture of the distal right tibia. In August 2005 she developed a second fracture of the distal right tibia, very close to the former. Neither of these fractures was preceded by trauma. The girl had never been treated with oral or topical steroids for her skin lesion. On physical examination there were linear, atrophic, hyperpigmented and indurated band-like skin lesions from the internal side of her right thigh to the shin of her right leg (Fig. 1a). Joint mobility of the ankle was unaffected. Laboratory investigations, including full blood cell count, biochemistry, antinuclear antibody test, antisingle-stranded DNA and anticentromere antibody were negative or within the normal range. X-ray of the leg revealed a fracture callus near a spiroideal fracture line without displacement of bone fragments in the distal portion of the right tibia. The bone also was slightly demineralized (Fig. 1b), probably due to immobilization for the treatment of the latter fracture. Lumbar spine densitometry (L1–L4) yielded a Z score of )2Æ0, which was considered to be at the lower limit of normality for her age. Serum calcium, phosphorus and alkaline phosphatase levels were within normal limits. The girl was diagnosed as having linear morphoea in whom two lytic lesions developed in the tibia. Treatment with oral calcium carbonate 625 mg daily and vitamin D 200 IU daily was then begun. Even though linear morphoea is a disorder of connective tissues with primary changes usually confined to skin and subcutaneous tissues, it may also involve the underlying muscle, periosteum and bone, resulting in functional impairment and cosmetic disfigurement. Many children develop severe atrophy of the extremities, deformities, contractures and limb length discrepancies. Structural changes may also occur in the osseous tissue. Thus, bone changes in the form of thinning, hypoplasia and shortening may ensue. The association of linear morphoea with melorheostosis and osteopoikilosis has also been recognized. Likewise, bone remodelling deformity and erosion of the periosteum have previously been described in association with localized or linear morphoea. However, osteolytic bone lesions have been described only once, in the calcaneus bone of a 3-year-old girl with localized morphoea. Most previously reported cases of bone resorption associated with scleroderma have involved the systemic form of scleroderma. Erosive changes in the distal interphalangeal joints and mandibular resorption are some radiographic findings described in patients with systemic sclerosis. Osteolysis of the distal end of the radius, ulna, acromioclavicular joints, ribs and cervical spine has also been described. Weber et al. reported bone fractures in a 28-year-old woman with progressive systemic sclerosis. The fractures occurred in bilateral mandibular angles, and were caused by a fall. A case of progressive systemic sclerosis with spontaneous fracture of the mandible has been reported. Vasculitis of the small muscular arteries is thought to be the origin of the ischaemic osteolytic lesions appearing in systemic scleroderma, and progressive bone resorption might predispose to fractures. The pathogenesis of osteolysis in patients with linear morphoea is not known. Greenberg et al. found vascular changes characteristic of scleroderma associated with bone lysis and proposed a common vascular aetiology for bone lysis in both systemic scleroderma and morphoea. Alternatively, bone