02 January 2025
: Case report
[In Press] A 20-Year-Old Woman with Metachronous Polyostotic Simple (Unicameral) Bone Cysts in 9 Sites: A Case Report
Challenging differential diagnosis, Unusual or unexpected effect of treatment, Diagnostic / therapeutic accidents, Rare disease
Nicole Liddy


DOI: 10.12659/AJCR.946165
Am J Case Rep In Press; DOI: 10.12659/AJCR.946165
Available online: 2025-01-02, In Press, Corrected Proof
Publication in the "In-Press" formula aims at speeding up the public availability of the pending manuscript while waiting for the final publication. The assigned DOI number is active and citable. The availability of the article in the Medline, PubMed and PMC databases as well as Web of Science will be obtained after the final publication according to the journal schedule
Abstract
BACKGROUND
Simple (unicameral) bone cysts (SBCs) are benign, fluid-filled bone lesions that are typically solitary and can be unicameral (single chamber) or septated. Most commonly affecting the long bones of children and adolescents, SBCs can be asymptomatic or associated with pain or fractures. Multifocal SBCs, featuring multiple cysts across different bones, are exceptionally rare and pose unique diagnostic and therapeutic challenges. Here, we report an exceptional case of a 20-year-old woman with multifocal SBCs affecting 9 known anatomical sites and discuss the unique diagnostic and therapeutic challenges.
CASE REPORT
A 20-year-old woman presented with right proximal tibia pain after a fall. Radiographs and MRI revealed radiolucent lesions in the proximal tibia, distal tibia, and fibula, with additional lesions identified on whole-body scintigraphy in the left proximal and distal tibia, left proximal humerus, and pelvis. Initial biopsy was suggestive of SBCs but inconclusive, and surgical biopsy was required for definitive diagnosis. Histologic evaluation confirmed SBCs, demonstrating cyst walls, cholesterol clefts, and fibrin-like deposits. Treatment included curettage, local adjuvant therapy with argon beam and doxycycline, bone grafting, and doxycycline sclerotherapy for pelvic and distal extremity lesions. At 17 months after treatment, the patient remained asymptomatic, with imaging showing sclerosis and near-complete resolution of the lesions.
CONCLUSIONS
This report presented a rare case of a young woman with multiple SBCs across 9 anatomical sites. Overall, the management of multifocal SBCs requires a multidisciplinary approach, involving orthopedic surgeons, radiologists, and pathologists for optimal outcomes.
Keywords: Bone Cysts; Case Reports; Doxycycline; Cysts
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