American Journal of Physical Medicine & Rehabilitation:
Kürklü, Mustafa MD; Özkan, Hüseyin MD; Kömürcü, Mahmut MD; Tunay, Servet MD; Başbozkurt, Mustafa MD
From Gülhane Military Medical Academy, Department of Orthopedics and Traumatology, Ankara, Turkey.
All correspondence and requests for reprints should be addressed to Mustafa Kürklü, GATA Ortopedi ve Travmatoloji A.D., 06018 Etlik, Ankara, Türkiye.
A 26-yr-old man (military staff) was seen for his complaint of left-foot pain for the last 17 mos. On detailed questioning, he described the pain as ensuing after military exercise (3–5 km running, pentathlon, decathlone) but not during routine daily activities or during rest. The physical examination (including the skin) was normal except for pain on the dorsal side of the second metatarsal bone during resistive plantar flexion of the second toe. Radiologic evaluation of the feet showed massive sclerotic changes in the second metatarsal and phalangeal bones on the left side (Fig. 1). Laboratory tests, including complete blood count, erythrocyte sedimentation rate, C-reactive protein, and tumor markers, were completely normal. Computed tomography of the feet and triphasic bone scintigraphy were both suggestive of melorheostosis (Figs. 2–3). Eventually, an open biopsy was carried out, and the pathologic diagnosis was consistent with nonspecific, reactive, regenerative, small, bony tissue, without any malignant changes. The patient was followed conservatively with a plantar orthosis (medial longitudinal arc and first metatarsal support), and he was instructed to avoid the aforementioned exercises. After 26 mos of follow-up, despite the persistence of the radiologic findings, the patient is currently quite well, with no painful symptoms unless he performs military exercise.
Melorheostosis is a rare disorder characterized by dense sclerotic bone. A single limb (lower limbs more than the upper ones) is most often affected with dense, eccentric involvement of one or more bones. The x-ray appearance is described to be somewhat similar to wax flowing along the side of a candle.1 Soft-tissue abnormalities are common, and skin lesions resemble scleroderma. Joint swelling, muscle contractures, and tendon and ligament shortening, causing pain and limited range of motion, can be encountered.1 The diagnosis is usually established before the age of 20.2
We have reported this case for two reasons. First, we would like to draw the attention of physicians to this rare disorder, which could be diagnosed somewhat late and without other relevant clinical findings except for bone lesions. Secondly, we also imply that biopsy results may sometimes be noncontributory, and thus radiologic diagnosis—with computed tomography and/or scintigraphy—should become the mainstay. Last, but not least, conservative treatment with pain management and daily life modifications seem to be all that is necessary.
1. Waddell C, Demos TC, Lomasney L, McCarthy R: The case: your diagnosis? Orthopedics 2005;28:720–5
2. Morris JM, Samilson RL, Corely CR: Melorheostosis: review of the literature and report of an interesting case with a 19-year follow-up. J Bone Joint Surg Am 1963;45:1191–206
© 2007 Lippincott Williams & Wilkins, Inc.