A 20-Year-Old Woman with Metachronous Polyostotic Simple (Unicameral) Bone Cysts in 9 Sites: A Case Report.

IF 1 Q3 MEDICINE, GENERAL & INTERNAL American Journal of Case Reports Pub Date : 2025-02-17 DOI:10.12659/AJCR.946165
Nicole Liddy, Sten H Kajitani, John M Gross, Ying Zou, Brock Lindsey, Albert J Aboulafia
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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.

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背景 单纯性(单腔)骨囊肿(SBC)是一种良性、充满液体的骨病变,通常为单发,可以是单腔(单腔)或隔腔。骨囊肿最常见于儿童和青少年的长骨,可无症状,也可伴有疼痛或骨折。多灶性 SBC,即在不同的骨骼上长出多个囊肿,非常罕见,给诊断和治疗带来了独特的挑战。在此,我们报告了一例 20 岁女性的特殊病例,她患有影响 9 个已知解剖部位的多灶性 SBC,并讨论了独特的诊断和治疗难题。病例报告 一名 20 岁女性在摔倒后出现右胫骨近端疼痛。X光片和核磁共振成像显示胫骨近端、胫骨远端和腓骨有放射性病变,全身闪烁扫描发现左侧胫骨近端和远端、左侧肱骨近端和骨盆有其他病变。最初的活组织检查提示为 SBC,但尚无定论,需要进行手术活组织检查才能明确诊断。组织学评估证实了 SBC,显示出囊壁、胆固醇裂隙和纤维蛋白样沉积物。治疗方法包括刮宫术、氩气束和强力霉素局部辅助治疗、植骨术以及针对骨盆和四肢远端病变的强力霉素硬化疗法。治疗 17 个月后,患者仍无症状,影像学显示病变硬化并接近完全消退。结论 本报告介绍了一例罕见的年轻女性多发性 SBC 病例,该病例涉及 9 个解剖部位。总之,多灶性 SBC 的治疗需要多学科的参与,包括骨科医生、放射科医生和病理学家,才能取得最佳疗效。
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来源期刊
American Journal of Case Reports
American Journal of Case Reports Medicine-Medicine (all)
CiteScore
1.80
自引率
0.00%
发文量
599
期刊介绍: American Journal of Case Reports is an international, peer-reviewed scientific journal that publishes single and series case reports in all medical fields. American Journal of Case Reports is issued on a continuous basis as a primary electronic journal. Print copies of a single article or a set of articles can be ordered on demand.
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