Zeynep Maraş ÖZDEMİR, Ayşegül SAĞIR KAHRAMAN, Cemile AYŞE GÖRMELİ, Reşit SEVİMLİ, Nusret AKPOLAT

Langerhans Cell Histiocytosis with Atypical Intervertebral Disc and Sacroiliac Joint Involvement Mimicking Osteoarticular Tuberculosis in an Adult

Background: Langerhans cell histiocytosis (LCH), typically found in children, is a rare single or mul-tisystem disorder with a wide range of clinical and radiological manifestations. Unusual presentations of LCH are occasionally encountered and it may be dif-ficult to distinguish LCH from an infection or a be-nign or malignant tumor.Results: A 35-year-old female presented with pain in her back and left buttock, malaise, and weight loss, with a duration of several months. Her laboratory test results were within the normal ranges except for the levels of acute phase reactants, which were elevated. Magnetic resonance imaging and computed tomogra-phy revealed a unilateral destructive sacroiliac lesion, and multiple vertebral lesions with adjacent discal in-volvement and extensive soft tissue extensions. She was initially misdiagnosed with multifocal osteoar-ticular tuberculosis. An open biopsy and joint curet-tage was performed. Histopathological examination showed that she had LCH.Conclusion: To the best of our knowledge, this is the first case of LCH associated with a destructive unilat-eral sacroiliac lesion, discal involvement, and involve-ment of the adjacent vertebrae, in an adult patient; the LCH mimicked osteoarticular tuberculosis. Disease onset in adulthood is rare, and this can potentially de-lay diagnosis. Familiarity with the imaging features of unusual LCH manifestations is necessary to ensure accurate diagnosis and appropriate treatment

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