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

Year 2016, Volume: 33 Issue: 5, 573 - 577, 01.09.2016

Zeynep Maraş Özdemir Ayşegül Sağır Kahraman Cemile Ayşe Görmeli Reşit Sevimli Nusret Akpolat

Abstract

Background: Langerhans cell histiocytosis (LCH),
typically found in children, is a rare single or multisystem
disorder with a wide range of clinical and
radiological manifestations. Unusual presentations of
LCH are occasionally encountered and it may be difficult
to distinguish LCH from an infection or a benign
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 tomography
revealed a unilateral destructive sacroiliac lesion,
and multiple vertebral lesions with adjacent discal involvement
and extensive soft tissue extensions. She
was initially misdiagnosed with multifocal osteoarticular
tuberculosis. An open biopsy and joint curettage
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 unilateral
sacroiliac lesion, discal involvement, and involvement
of the adjacent vertebrae, in an adult patient; the
LCH mimicked osteoarticular tuberculosis. Disease
onset in adulthood is rare, and this can potentially delay
diagnosis. Familiarity with the imaging features
of unusual LCH manifestations is necessary to ensure
accurate diagnosis and appropriate treatment.

Keywords

Langerhans cell histiocytosis, sacroiliac joint, intervertebral disc, tuberculosis, osteoarticular

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