Parosteal aneurysmal bone cyst^☆

ABSTRACT

The incidence of aneurysmal bone cysts is 0.14 cases per 100,000 individuals. Parosteal aneurysmal bone cysts are the least prevalent subtype and represent 7% of all aneurysmal bone cysts. We present the case of a 38-year-old male patient with pain and bulging in his right arm for eight months. He had previously been diagnosed as presenting giant-cell tumor, but his slides were reviewed and his condition was then diagnosed as parosteal aneurysmal bone cyst. The patient was treated with corticosteroid and calcitonin infiltration into the lesion and evolved with clinical and radiological improvement within the first five weeks after the operation.

Keywords:
Aneurysmal bone cyst; Calcitonin; Corticosteroids; Infiltration

resumo

O cisto ósseo aneurismático tem uma incidência de 0,14 a cada 100 mil indivíduos. O subtipo parosteal é o menos prevalente, representa 7% de todos. Apresentamos um paciente mas culino, 38 anos, com dor e abaulamento em braço direito havia oito meses. Diagnosticado previamente como tumor de células gigantes, teve sua lâmina revisada e então foi feito o diagnóstico de cisto ósseo aneurismático parosteal. O paciente foi tratado com infiltração intralesional de corticosteroide e calcitonina e evoluiu com melhoria clínica e radiológica já nas primeiras cinco semanas pós-operatórias.

Palavras-chave:
Cisto ósseo aneurismático; Calcitonina; Corticosteroides; Infiltração.

Introduction

Aneurysmal bone cysts were first described byJaffe and Lichtenstein in 1942.1They account for 1-2% of all primary bone tumors and affect the metaphyseal region of long bones in children, adolescents and young adults.2^and3

This lesion typically develops inside bones.4Cysts located in the cortical bone are rare and account for 7-9.3% of all aneurysmal bone cysts.5^and6

Few cases have been reported in the literature. The approach used is individualized and varies according to the experience of each service. We present a case of parosteal aneurysmal bone cyst that was treated in accordance with our experience.

Case report

The patient was a 38-year-old man of mixed race with a complaint of pain and bulging in his right arm. It was of progressive nature and the patient had had the complaint for at least eight months. He said that he had not suffered any trauma or undergone previous surgery.

The patient, who had been attended previously at another service, underwent a biopsy from which the histopathological diagnosis was compatible with a giant-cell tumor. When he arrived at our service, because of the clinical and radiological characteristics of the slides (Fig. 1,Fig. 2,Fig. 3andFig. 4), a review of them was requested.

Fig. 1:
Radiographs of the right humerus in anteroposterior and lateral view.

Fig. 2:
Tomographic features.

Fig. 3:
Magnetic resonance.

Fig. 4:
Bone scintigraphy.

This review showed the presence of a lesion formed by cyst membranes that sometimes showed complete septation constituted by fusiform and multinucleated giant cells. Bone trabeculae dissociated by connective tissue were noted, along with neoformed bone trabeculae of reactive pattern, which led to the diagnosis of parosteal aneurysmal bone cyst.

Infiltration into the lesion using calcitonin and corticosteroid was indicated after reaching a group decision. In the fifth postoperative week, the lesion was already seen to be undergoing an ossification process (Fig. 5).

Fig. 5:
Postoperative radiographs.

Discussion

Aneurysmal bone cysts were first described byJaffe and Lichtenstein in 1942. According to the World Health Organization, they are characterized as benign cystic bone lesions composed of bone voids that are filled with blood and separated by septa of connective tissue containing fibroblasts, osteoclastic giant cells and reactive bone tissue. .1 2 3^amd7

These cysts account for 1-2% of all primary bone tumors and their incidence is 0.14 per 100,000 individuals.8The lesions affect the metaphyseal region of the long bones of children, adolescents and young adults.2^and3

The lesions generally develop inside bones and cause thinning of the cortex and possibly bone protrusion.4Localized cysts in the cortical bone are rare and were previously named subperiosteal giant cells or subperiosteal osteoclasis. .4In 1950, Lichtenstein9published an article that elucidated and differentiated parosteal aneurysmal bone cysts from subperiosteal giant cells, hemangiomas and osteogenic sarcomas.

In 1957, Sherman and Soong5classified aneurysmal bone cysts into three types: eccentric, parosteal and central. The parosteal subtype is the least frequent subtype, accounting for 7-9.3% of all aneurysmal bone cysts. .5^and6

Pain is the most prevalent symptom, and its duration may be weeks to months.2Radiographically, these cysts present as single eccentric and insufflative lesions that reach the periosteum and have well-defined margins.2^and3Their presence may be associated with onion-skin periosteal reactions and Codman's triangle.2^and6

Tomography helps in making the differential diagnosis of these lesions. They show liquid density and may clearly demonstrate the liquid levels.2^and6Scintigraphy shows that there is greater uptake at the periphery of the lesion.2In magnetic resonance imaging, the lesion is well defined, with lobulated outlines and liquid levels.²

The histology of aneurysmal bone cysts is characterized by voids filled with blood. These voids are covered by a single layer of undifferentiated cells. The solid tissue surrounding the lesion is composed of richly vascularized fibrosis.2Diagnostic differentiation between giant-cell tumors and osteosarcoma with telangiectasia is anatomopathologically complex.2

Because these are aggressive lesions, the treatment consists of curettage, with or without subsequent adjuvants such as bone grafts, bone marrow aspirate, cryotherapy, argon, phenol or calcitonin with corticosteroid injection into the lesion. .7^and10In our service, use of corticosteroids in association with calcitonin, injected into the lesion, is the preferred method for treating this type of lesion. Cases of resolution of lesions after an episode of fracturing or after a biopsy, or even spontaneously, have been described.7^and8Lesion recurrence is associated with young patients, previous aneurysmal bone cysts, location adjacent to a joint or growth plate, low mitotic count and presence of other open growth plates.8

Here, we presented a rare case of parosteal aneurysmal bone cyst in which the clinical, radiological and anatomopathological findings and presence of a multidisciplinary team were essential in order to completely elucidate the diagnosis.

Referências

• Jaffe HL, Lichtenstein L. Solitary unicameral bone cyst: with emphasis on the roentgen picture. The pathologic appearance and the pathogenesis. Archsurg. 1942;44(6):1004-25.
• Jesus-Garcia R. Diagnóstico e tratamento de tumores ósseos. 2 ed. Rio de Janeiro: Elsevier; 2013.
• Pietschmann MF, Oliveira AM, Chou MM, Ihrler S, Niederhagen M, Baur-Melnyk A, et al. Aneurysmal bone cysts of soft tissue represente true neoplasm. A report of two cases. J Bone Joint Surg Am. 2011;93(45):1-8.
• Kobayashi S, Hayakawa K, Takeno K, Baba H, Meir A. Parosteal aneurysmal bone cyst of the humerus with birdcage-like ossification on three-dimensional CT scanning: a case report. Joint Bone Spine. 2009;76(6):705-7.
• Sherman RS, Soong KY. Aneurysmal bone cyst: its roentgen diagnosis. Radiology. 1957;68(1):54-64.
• De Dios AMV, Bond JR, Shives TC, McLeod RA, Unni KK. Aneurysmal bone cyst. A clinicopathologic study of 238 cases. Cancer. 1992;69(12):2921-31.
• Reddy KIA, Sinnaeve F, Gaston CL, Grimer RJ, Carter SR. Aneurysmal bone cysts: do simple treatments work? Clin Orthop Relat Res. 2014;472(6):1901-10.
• Steffner RJ, Liao C, Stacy G, Atanda A, Attar S, Avedian R, et al. Factors associated with recurrence of primary aneurysmal bone cysts: is argon beam coagulation an effective adjuvant treatment?. J Bone Joint Surg Am 2011;93(21):e1221-9.
• Lichtenstein L. Aneurysmal bone cyst: a pathological entity commonly mistaken for giant cell tumor and occasionally for hemangioma and osteosarcoma. Cancer. 1950;3(2):279-89.
• Docquier PL, Dellove C. Treatment of aneurysmal bone cysts by introduction of demineralized bone and autogenous bone marrow.. J Bone Joint Surg Am 2005;87(10):2253-8.

• 2
  ☆Work performed at the Instituto Nacional de Traumatologia e Ortopedia (INTO), Rio de Janeiro, RJ, Brazil.

Publication Dates

• Publication in this collection
  Oct 2015

History

• Received
  04 June 2014
• Accepted
  23 Oct 2014