Fibrous dysplasia with secondary aneurysmal bone cyst—a rare case report and literature review

Rau, Levy Hermes; Reinheimer, Angélica; Meurer, Maria Inês; Marodin, Aline; Espezim, Catherine Schmitz; Klüppel, Leandro Eduardo; FERNANDES, Paula Cristina dos Santos Vaz; Rivero, Elena Riet Correa

doi:10.1007/s10006-019-00741-w

Cited by 13 publications

(6 citation statements)

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“…These findings were compatible with concomitant fibroosseous injury and ABC. The patient was disease-free at the time of his 10-month follow-up [20].…”

Section: Resultsmentioning

confidence: 96%

Incidence and major clinical approaches of aneurismatic bone cysts: a concise systematic review

Vasconcelos¹,

Bannak²,

Naves³

et al. 2022

MedNEXT

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Introduction: Aneurysmal bone cysts (ABC) are pseudocysts expansive lesions. The World Health Organization (WHO) categorizes primary ABCs as cystic neoplasms that contain fibrous septa separating numerous blood-filled spaces. Approximately 70% of ABC cases are primary, while 30% are secondary. Objective: The present study aimed to carry out a concise systematic review of the main clinical findings of aneurysmal bone cysts, highlighting the incidence and prevalence, as well as the main forms of treatment. Methods: The rules of the Systematic Review-PRISMA Platform were followed. The research was carried out from May 2022 to July 2022 and developed based on Scopus, PubMed, Science Direct, Scielo, and Google Scholar. The quality of the studies was based on the GRADE instrument and the risk of bias was analyzed according to the Cochrane instrument. Results: A total of 143 articles were found. In total, 73 articles were evaluated and 10 were included and evaluated in the systematic review. The symmetric funnel plot does not suggest a risk of bias. After prospecting the main literary findings, it was generally evidenced that the aneurysmal bone cyst (ABC) is a rare lesion that is more frequently found in young adults and children. It may have unpredictable behavior, with a high recurrence rate after treatment. Standard treatment consists of curettage plus local adjuvants and bone grafting to fill the void. In anatomic sites with difficult surgical access, percutaneous procedures are used (injection of sclerosing agents, radiofrequency thermal ablation, or selective arterial embolization. Medical management with bisphosphonates or denosumab has also been advocated. Conclusion: Primary ABCs involving the maxillofacial region are extremely rare and even more in patients with advanced age. Next-generation sequencing confirmed the presence of a USP6-CDH11 fusion gene, consistent with classification as a primary aneurysmal bone cyst. Sclerotherapy provided an effective and minimally invasive treatment for aneurysmal bone cysts and is particularly useful for deep injuries, challenging surgical access, and potentially injurious vital structures.

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“…These findings were compatible with concomitant fibroosseous injury and ABC. The patient was disease-free at the time of his 10-month follow-up [20].…”

Section: Resultsmentioning

confidence: 96%

Incidence and major clinical approaches of aneurismatic bone cysts: a concise systematic review

Vasconcelos¹,

Bannak²,

Naves³

et al. 2022

MedNEXT

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“…ABC formation could be attributed to a local circulatory disturbance causing a markedly elevated venous pressure and the development of enlarged vascular spaces within the affected bone [7,25]. This hemodynamic disturbance could be secondary to local trauma or the presence of a pre-existing lesion in the affected bone [7,25]. Clinically, COD usually presents slow-growing, painless lesions, whereas the ABC presents a rapidly growing, painful lesion.…”

Section: Discussionmentioning

confidence: 99%

“…Free hemorrhage was frequently interspersed in the artifactual spaces throughout COD [ 6 ]. ABC formation could be attributed to a local circulatory disturbance causing a markedly elevated venous pressure and the development of enlarged vascular spaces within the affected bone [ 7 , 25 ]. This hemodynamic disturbance could be secondary to local trauma or the presence of a pre-existing lesion in the affected bone [ 7 , 25 ].…”

Section: Discussionmentioning

confidence: 99%

“…The aneurysmal bone cyst (ABC) is a benign, osteolytic, rapidly growing, expansile lesion of the bone, which predominantly occurs in the metaphysis of long bones, such as the femur, tibia, and spine [7,8]. Its occurrence in the jaws is uncommon [7]. Radiographically, ABCs are characterized by ballooned cortical expansion from unilocular to multilocular radiolucent lesions [8].…”

Section: Introductionmentioning

confidence: 99%

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Concomitant cemento-osseous dysplasia and aneurysmal bone cyst of the mandible: a rare case report with literature review

Yeom

Yoon

2020

BMC Oral Health

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Background Concomitant cemento-osseous dysplasia (COD) and aneurysmal bone cyst (ABC) are rare in the head and neck region. In our search of the English language literature, we found only one case report describing the simultaneous occurrence of COD and ABC in the head and neck region. Here, we report a case of COD associated with ABC. Further, we performed a systematic search of the literature to identify studies on patients with COD associated with nonepithelial lined cysts of the jaws. Case presentation The patient was a 32-year-old woman who was referred from a private dental clinic because of a cystic lesion below the mandibular right first molar. She had no pain or significant systemic disease. After performing panoramic radiography and cone-beam computed tomography, the imaging diagnosis was COD with a cystic lesion, such as ABC or solitary bone cyst. Excisional biopsy was performed, which revealed concomitant COD and ABC. Conclusion This case of ABC associated with COD provides insight for the diagnostic process of radiographically mixed lesions with cystic changes.

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“…По литературным данным, заболевание наиболее часто встречается в детском возрасте -от 6 до 15 лет (без привязки к полу) и составляет от 2% до 18% из всех опухолевых образований костей скелета [5]. Поражение костей свода черепа среди данной патологии, по сведениям некоторых авторов, составляет 20%, из которых 20-40% приходится на челюстно-лицевую область [6,7]. 30% дисплазий имеют моноосальный вариант и нередко сочетаются с другими доброкачественными новообразованиями костной ткани, например с остеомой.…”

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A Clinical Case of Multi-Stage Surgical Treatment of a Patient With Extensive Fibrous Dysplasia of the Skull and Subsequent Reconstruction With Individual Titanium Implants

Mishinov¹,

Kopylov²,

Pendyurin³

et al. 2022

СПНО (MPSE)

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Фиброзная дисплазия относится к группе доброкачественных (опухолеподобных) заболеваний. В основе патогенеза лежит нарушение остеобластического процесса в костной ткани, вследствие чего происходит ее замещение на волокнисто-соединительную (фиброзную) ткань. Наиболее часто заболевание встречается в детском возрасте (от 6 до 15 лет) и составляет от 2% до 18% от всех опухолевых образований костей скелета. Поражение костей свода черепа среди данной патологии составляет не менее 20%. В ряде случаев данное заболевание медленно прогрессирует в течение всей жизни, что приводит к тяжелым косметическим и неврологическим осложнениям. Малигнизация при фиброзной остеодисплазии встречается редко, в 0,4-1% случаев. Цель исследования: демонстрация клинического наблюдения пациента с выполненным многоэтапным хирургическим лечением по поводу обширного поражения костей свода и основания черепа фиброзной дисплазией с последующим закрытием дефекта костей черепа двумя индивидуальными титановыми имплантатами, изготовленными путем трехмерной печати. Преимущество данного метода и эффективность использования индивидуальных титановых имплантатов в этапном лечении фиброзной дисплазии костей свода и основания черепа позволяют достигнуть высокой степени радикальности при резекции патологически измененной кости с сохранением желаемых косметических результатов, удовлетворяющих больных. Ключевые слова: нейрохирургия, фиброзная дисплазия, доброкачественные опухоли костей черепа, дефект черепа, краниопластика, 3D-печать.

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Fibrous dysplasia with secondary aneurysmal bone cyst—a rare case report and literature review

Cited by 13 publications

References 50 publications

Incidence and major clinical approaches of aneurismatic bone cysts: a concise systematic review

Incidence and major clinical approaches of aneurismatic bone cysts: a concise systematic review

Concomitant cemento-osseous dysplasia and aneurysmal bone cyst of the mandible: a rare case report with literature review

A Clinical Case of Multi-Stage Surgical Treatment of a Patient With Extensive Fibrous Dysplasia of the Skull and Subsequent Reconstruction With Individual Titanium Implants

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