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

Abstract

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