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J Surg Case Rep 2022 Jun 7;2022(6):rjac196. Epub 2022 Jun 7.

Cliniques Universitaires Saint-Luc, Service de Chirurgie Orthopédique et Traumatologique, Bruxelles, Belgium.

The occurrence of a pathological fracture in children requires a rigorous diagnostic approach in order to establish the etiology and to develop a precise therapeutic strategy. Several causes are associated with these fractures, the most frequent being benign tumors in children in developed countries and chronic osteomyelitis in developing countries. More rarely, malignant tumors must however always be considered. Read More

Clin Nucl Med 2022 Jul 5;47(7):635-637. Epub 2022 Apr 5.

From the Departments of Radiology.

Abstract: Ewing sarcoma is the second most common primary bone tumor in children. Typical Ewing sarcoma most frequently occurs in long bones and within the pelvis. ALES (adamantinoma-like Ewing sarcoma) is a rare subtype of Ewing sarcoma that is characterized by epithelial differentiation in addition to small round blue cells. Read More

Int J Surg Pathol 2022 Apr 25:10668969221081029. Epub 2022 Apr 25.

Department of Pathology, 378541City of Hope National Medical Center, Duarte, CA, USA.

. Adamantinoma-like Ewing sarcoma typically shows t(11;22) translocation and complex epithelial differentiation. It poses a diagnostic challenge, especially in the head and neck region, due to its under-recognition and significant histologic overlap with other malignancies. Read More

Zhonghua Bing Li Xue Za Zhi 2022 Mar;51(3):234-236

Department of Pathology, Beijing Jishuitan Hospital, Beijing 100035, China.

Bone Joint J 2022 Feb;104-B(2):302-308

Great Ormond Street Hospital for Children, London, UK.

Aims: Osteofibrous dysplasia (OFD) is a rare benign lesion predominantly affecting the tibia in children. Its potential link to adamantinoma has influenced management. This international case series reviews the presentation of OFD and management approaches to improve our understanding of OFD. Read More

J Ayub Med Coll Abbottabad 2021 Oct-Dec;33(Suppl 1)(4):S835-S840

Department Orthopaedic Surgery, Dow University of health sciences, Karachi, Pakistan.

Adamantinoma is a rare neoplastic bone tumor that has the potential to metastasize. The classic presentation is in the tibial region however; cases involving other bones of the body have been noticed. The tumour is very likely to be mistaken for other bone diseases and therefore it is important to investigate and study about its nature and thus differentiate it from other differentials. Read More

In Vivo 2021 Nov-Dec;35(6):3045-3052

Department of Anatomy - Histology-Embryology, University of Ioannina, Ioannina, Greece.

Adamantinoma is a biphasic tumor, with a low potential for malignancy, characterized by clusters of epithelial cells surrounded by a relatively bland spindle-cell osteofibrous component. The aim of the present study was to review the updated data regarding epidemiology; pathogenesis; clinical presentation; radiological, histopathological and ultrastructural findings; and treatment options of adamantinoma. In X-ray, it is usually seen as an eccentric and sometimes central, lobular, lytic lesion with sclerotic margins of overlapping radiolucency, and a characteristic 'soap-bubble' appearance. Read More

Zhonghua Bing Li Xue Za Zhi 2021 Aug;50(8):943-945

Department of Pathology, Xijing Hospital, the Fourth Military Medical University, Xi'an 710032, China.

Pathol Int 2021 Sep 30;71(9):614-620. Epub 2021 Jun 30.

Department of Diagnostic Pathology, National Hospital Organization Saitama Hospital, Saitama, Japan.

Ewing-like adamantinoma (EAD) is a rare bone tumor. It remains unclear whether EAD belongs to adamantinoma, Ewing sarcoma (ES), or an independent category. Herein, we present a case of femoral sarcoma previously diagnosed as EAD in a 26-year-old woman. Read More

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 2022 Jan 10;51(1):209-217. 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

Int J Surg Pathol 2021 Oct 11;29(7):798-803. Epub 2021 Mar 11.

National Health Laboratory Service, 121470Stellenbosch University, Cape Town, South Africa.

Adamantinoma-like Ewing sarcoma is a rare variant of Ewing sarcoma with histologic and immunohistochemical evidence of squamous differentiation. This variant most commonly occurs in the head and neck region with a few cases reported in the long bones of the limbs. It may be associated with poorer clinical outcome and could pose a diagnostic challenge, particularly if it occurs in older patients or as a metastatic lesion. 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

Int J Oral Maxillofac Surg 2021 Aug 6;50(8):1023-1026. Epub 2020 Dec 6.

Department of Oral and Maxillofacial Surgery, Peking University School and Hospital of Stomatology, Beijing, China. Electronic address:

Adamantinoma is an extremely rare tumour originating from bone that can be divided into classical and osteofibrous dysplasia (OFD)-like adamantinoma. This low-grade malignancy almost exclusively occurs in long bones, and only few cases of mandibular adamantinoma have been reported. Here, we report the case of a 30-year-old female with a 2-year history of right mandible tenderness. 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