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Radiol Case Rep 2021 Aug 7;16(8):1934-1940. Epub 2021 Jun 7.

Department of Radiology, The Ohio State University Wexner Medical Center, Columbus 43210, Ohio, USA.

Metastatic adamantinoma involving the spine is an extremely rare occurrence. In this case report, we present a patient with recurrent adamantinoma of the tibia which was found to have metastasized to the spine. The metastatic involvement was diagnosed pathologically, status post CT guided percutaneous core needle biopsy, performed after the patient returned with concerning symptoms and imaging findings suggestive of metastasis. Read More

Skeletal Radiol 2021 Jun 10. Epub 2021 Jun 10.

Sparsh Orthopedic Oncology, Ahmedabad, Gujrat, India.

Adamantinoma-like Ewing sarcoma is uncommonly reported in the skeletal sites, including small bones of the feet.A 15-year-old girl presented with pain and swelling in her left foot, leading to difficulty in walking for 8 months. Plain radiograph revealed an ill-defined, lytic-sclerotic lesion without significant periosteal reaction in her left calcaneus. Read More

BMJ Case Rep 2021 Jun 3;14(6). Epub 2021 Jun 3.

Department of Gynaecological Oncology, Oxford University Hospitals NHS Foundation Trust, Oxford, Oxfordshire, UK

Adamantinoma is a rare low-grade malignancy of the appendicular skeleton with unclear histogenesis. We present the case of a 65-year-old woman with known recurrent and metastatic right tibial disease despite clear resection margins. On further investigation, a positron emission tomography-CT scan identified a primary breast lesion and an 11 cm mass in the right iliac fossa of suspected ovarian malignancy amenable to surgical resection. Read More

Orthop Traumatol Surg Res 2021 Mar 10:102888. Epub 2021 Mar 10.

Department of Pediatric Orthopedics, Fuzhou Second Hospital Affiliated to Xiamen University, 47th Shangteng Road of Cangshan District, Fuzhou 350007, Fujian Province, China. Electronic address:

Background: Osteofibrous dysplasia (OFD) is a rare non neoplastic, self-limited intracortical fibro-osseous lesion that most commonly affects the diaphysis of the tibia and fibula of children, the best treatment is still debated. Therefore we performed a retrospective study in children mostly under 10 years old with OFD aiming to determine whether early surgery is necessary and which is the best treatment.

Hypothesis: Symptomatic OFD lesions should be treated proactively, and appropriate treatment can achieve favorable outcome. Read More

Clin Nucl Med 2021 Jul;46(7):e365-e367

Department of Radiology, University of British Columbia.

Abstract: Adamantinomas are rare malignant primary bone tumors, which typically arise in young patients, are generally low-grade tumors, and classically arise from the anterior tibial diaphysis. We present the case of a 70-year-old woman who underwent repeat imaging with FDG PET for a pathologically proven case of femoral adamantinoma who experienced a relatively rapid development of metastatic disease with an atypical distribution of lesions. Read More

J Orthop Surg Res 2020 Jul 16;15(1):268. Epub 2020 Jul 16.

Department of Orthopaedic Oncology Surgery, Beijing Jishuitan Hospital, Peking University, Number 31, Xinjiekoudongjie Street, Xicheng District, Beijing, 100035, China.

Background: The clinical and molecular characteristics of osteofibrous dysplasia (OFD)-like adamantinoma (AD) differ from those of classic AD. Most reports about OFD-like AD are case reports or small case series. More cases from different centers are still warranted. Read More

Diagn Cytopathol 2020 Dec 19;48(12):E4-E6. Epub 2020 Jun 19.

Department of Pathology, Johns Hopkins University School of Medicine, Baltimore, Maryland, USA.

Adamantinoma-like Ewing sarcoma (ALES) is a rare variant of Ewing sarcoma family of tumors (ESFTs). ALES is characterized by translocations of the EWSR1 (Ewing sarcoma breakpoint region 1) gene on chromosome 22, morphologic features of small round blue cell tumors with focal squamous differentiation, and a unique immunoprofile. Herein, we report a 21-year-old patient who presented with severe, acute onset back pain. Read More

J Surg Oncol 2020 Aug 25;122(2):273-282. Epub 2020 Apr 25.

Department of Surgery, Orthopaedic Service, Memorial Sloan-Kettering Cancer Center, New York, New York.

Background And Objectives: Adamantinomas are primary, low-grade malignant tumors of the bone that have metastatic potential to the lungs, lymph nodes, and other regions. The rarity of this disease and its nonspecific symptoms complicate diagnosis.

Materials And Methods: Records for 20 patients who underwent treatment for adamantinoma from 1975 to 2018 were reviewed for demographic, clinical, and pathological data, treatment details, postoperative complications, and outcomes. Read More

Radiol Case Rep 2019 Nov 5;14(11):1330-1333. Epub 2019 Sep 5.

Department of Medical imaging, Hefei Second People's Hospital of Wannan Medical College, Guangde Road, Hefei, 230011, China.

Adamantinoma of the bone is a rare low-grade bony tumor that accounts for less than 1% of all primary bone tumors. On imaging, adamantinoma may be similar to other tumors such as osteofibrous dysplasia, for which the treatment protocol is completely different. Therefore, correct diagnosis and staging of adamantinoma ensures that the patient will undergo appropriate surgery. Read More

Hum Pathol 2019 09 26;91:123-128. Epub 2018 Nov 26.

Department of Laboratory Medicine and Pathology, Mayo Clinic, Rochester, MN 55905. Electronic address:

Adamantinoma of the long bones is a rare, typically low-grade malignant tumor that frequently involves the tibia. Radiographically, adamantinoma is characteristically a lytic, intracortical, and expansile lesion with variable margins. Histologically, adamantinoma is a bimorphic neoplasm, composed of epithelial and osteofibrous elements. Read More

Int J Surg Pathol 2019 Apr 25;27(2):193-202. Epub 2018 Jul 25.

1 Tata Memorial Centre, Mumbai, India.

A dedifferentiated adamantinoma is a rare subtype of an adamantinoma, associated with a relatively aggressive clinical course, with less than 10 such cases reported so far. A 25-year-old-male presented with pain in his right leg of 1-year duration. Imaging disclosed a well-defined lytic, destructive lesion in his proximal tibia with a cortical break and a soft tissue component. Read More

JBJS Case Connect 2018 Jul-Sep;8(3):e51

Department of Radiology, Universitaetsklinik Balgrist, Zurich, Switzerland.

Case: A 38-year-old man presented with multifocal, partially confluent osteolytic lesions in the proximal dia-meta-epiphyseal region of the proximal aspect of the left tibia, which had been found incidentally when a radiograph was made after a rotational knee injury. When the results of a percutaneous core needle biopsy proved inconclusive, an open biopsy was performed. Osteofibrous dysplasia (OFD) with scattered groups of plump cells with a rhabdoid phenotype, shown to express both vimentin and pan-cytokeratin, was found. Read More

J Pediatr Orthop 2018 Aug;38(7):e404-e410

Department of Orthopaedic Surgery, Seoul National University Hospital.

Background: Our study of a large patient group reports on the behavior and postoperative recurrence of osteofibrous dysplasia (OFD).

Methods: We reviewed the medical records of 55 patients who were diagnosed with OFD of the tibia and showed typical features of this tumor. The patients' presentation, disease course, history of pathologic fracture, typical radiographic features, surgical treatment history, and surgical results were investigated. Read More

BMJ Case Rep 2018 May 15;2018. Epub 2018 May 15.

Department of Radiology, Alder Hey NHS Foundation Trust, Liverpool, UK.

J Orthop Surg (Hong Kong) 2017 May-Aug;25(2):2309499017713940

4 Orthopaedic and Traumatology Department, Sanglah General Hospital, Faculty of Medicine, Udayana University, Denpasar, Bali, Indonesia.

Amputation still considered as primary choice of malignancy treatment in distal tibia. Bone recycling with liquid nitrogen for reconstruction following resection of malignant bone tumours offers many advantages. We presented four patients with osteosarcoma, Ewing sarcoma, adamantinoma and recurrent giant cell tumour over distal tibia. Read More

Clin Nucl Med 2017 Aug;42(8):e375-e376

From the Departments of *Radiology, and †Pathology, Kagoshima University Graduate School of Medical and Dental Sciences, Kagoshima, Japan.

A 17-year-old girl had pain in the right front lower leg while road racing 2 months earlier. Radiography and CT showed osteolytic lesions in the medial part of the bilateral tibia. Bone scintigraphy showed focally increased radiotracer uptake in the bilateral tibia. Read More

Head Neck Pathol 2017 Dec 7;11(4):541-545. Epub 2017 Feb 7.

Institute of Pathology, Technical University of Munich (TUM), Munich, Germany.

The term "sinonasal undifferentiated carcinoma (SNUC)" has been coined in 1986 for a highly aggressive sinonasal tract epithelial neoplasm showing distinctive morphology, but lacking any specific line of differentiation. Recent developments resulted in a dynamic splitting of new entities traditionally included in the spectrum of SNUC. Sinonasal NUT-midline carcinoma, adamantinoma-like Ewing family tumors and most recently, SMARCB1(INI1)-deficient sinonasal carcinoma are the main entities defined by specific genetic aberrations. Read More

World Neurosurg 2016 Nov 21;95:618.e7-618.e12. Epub 2016 Aug 21.

Department of Neurosurgery, Johns Hopkins Hospital, Baltimore, Maryland, USA; Department of Oncology, Johns Hopkins Hospital, Baltimore, Maryland, USA; Department of Otolaryngology-Head and Neck Surgery, Johns Hopkins University School of Medicine, Baltimore, Maryland, USA. Electronic address:

Background: Craniopharyngiomas are uncommon benign locally aggressive epithelial tumors mostly located in the sellar and suprasellar regions. An infrasellar origin of these tumors is rare.

Case Description: The authors report a 22-year-old male patient with a purely infrasellar adamantinomatous craniopharyngioma centered in the nasopharynx with extension into the posterior nasal septum, sphenoid sinus, and clivus. Read More

J Med Case Rep 2016 Jun 23;10(1):185. Epub 2016 Jun 23.

Department of Orthopedic Surgery, Keio University School of Medicine, 35 Shinanomachi, Shinjuku-ku, Tokyo, 160-8582, Japan.

Background: Adamantinoma arising in the femur is extremely rare. We report a case of an adamantinoma occurring in the right medial femoral condyle that was diagnosed 5 years after the primary surgery.

Case Presentation: A 74-year-old Asian woman first complained of right knee pain without any cause. Read More

Rev Esp Med Nucl Imagen Mol 2016 Sep-Oct;35(5):341-3. Epub 2016 Mar 14.

Nuclear Medicine, Spedali Civili Brescia, Brescia, Italy.

World J Nucl Med 2016 Jan-Apr;15(1):50-2

Department of Medical Imaging and Clinical Oncology, Stellenbosch University, and Tygerberg Hospital, South Africa.

Adamantinoma is a rare locally aggressive osteolytic tumor that is found 90% of the time in the diaphysis of the tibia with the remaining lesions found in the fibula and long tubular bones. A case of adamantinoma of the tibia is presented. The added value of nuclear medicine investigations in the workup of this patient is described. Read More

APMIS 2016 Apr 14;124(4):278-90. Epub 2016 Jan 14.

Department of Surgical Oncology (Bone and Soft Tissues), Tata Memorial Centre, Mumbai, India.

Primary intraosseous myoepithelial tumours, including carcinomas are rare tumours. The concept of histopathological spectrum of these tumours is evolving. We describe clinicopathological and immunohistochemical features of five myoepithelial carcinomas, including molecular cytogenetic results in one case. Read More

Global Spine J 2015 Aug 25;5(4):e7-e12. Epub 2014 Oct 25.

Department of Pathology, Anatomia e Istologia Patologica, Istituto Ortopedico Rizzoli, Bologna, Italy.

Study Design Case report and literature review. Objective To present the first case of metastatic adamantinoma of the spine with immunohistochemical confirmation and an updated literature review. Summary of Background Data Spinal metastatic disease could be difficult to diagnose because of the multiple differential diagnoses involved. Read More

Acta Orthop Traumatol Turc 2015 ;49(2):210-2

Memorial Hospital, Department of Orthopedics and Traumatology, İstanbul, Turkey.

We report a case of a very rarely seen osteofibrous dysplasia-like adamantinoma (OFDLA) of the lower leg in a 3-month-old male infant, making it the youngest case in the literature. OFDLA is typically regarded as a benign lesion; however, due to its convertibility into classical adamantinoma, it is recommended to evaluate it as a pre-malignant lesion. After OFDLA diagnosis with biopsy, our case underwent surgical resection and reconstruction with a large allograft. Read More

Arch Argent Pediatr 2015 Jan;113(1):e26-30

Servicio de Ortopedia y Traumatología, Hospital Nacional de Pediatría Prof. Dr. Juan P. Garrahan.

Adamantinoma is a primary tumor of long bones, which affects mainly the shaft of the tibia, and is extremely rare in pediatrics. It frequently presents during the second decade of life, with a slight predominance in males. It is a low grade tumor, with local aggressiveness and low rate of metastasis and recurrence once it is completely removed. Read More

J Neuropathol Exp Neurol 2015 Jan;74(1):95-7

University of Colorado Denver School of Medicine Anschutz Medical Campus Aurora, Colorado

BMC Res Notes 2014 Oct 18;7:736. Epub 2014 Oct 18.

Department of Orthopaedics, Graduate School of Medical Science, Kyoto Prefectural University of Medicine, Kawaramachi-Hirokoji, Kamigyo-ku, Kyoto 602-8566, Japan.

Background: Primary pulmonary synovial sarcoma (PPSS) is rare. We describe a case of PPSS complicated by tibial adamantinoma that required differentiation from lung metastasis.

Case Presentation: A 39-year-old Japanese woman presented with hemoptysis, dyspnea, and a well-defined tumor measuring 3. Read More

Acta Orthop 2014 Dec 20;85(6):663-9. Epub 2014 Aug 20.

Department of Orthopedic Surgery , University of Groningen, University Medical Center Groningen, Groningen , the Netherlands.

Background And Purpose: In orthopedic oncology, computer-assisted surgery (CAS) can be considered an alternative to fluoroscopy and direct measurement for orientation, planning, and margin control. However, only small case series reporting specific applications have been published. We therefore describe possible applications of CAS and report preliminary results in 130 procedures. Read More

Orthopedics 2014 Jun;37(6):362, 420-2

A 73-year-old woman with no significant past medical history presents with a palpable lump in the midshaft of the left tibia and intermittent mild discomfort for the past 8 months. Read More