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Diagn Cytopathol 2020 Jun 19. 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 Apr 25. 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

Diagn Interv Imaging 2014 Jun 11;95(6):621-3. Epub 2014 Mar 11.

Pediatric Radiology Department, hôpital Jeanne-de-Flandre, université de Lille 2, CHRU de Lille, avenue Eugène-Avinée, 59037 Lille cedex, France. Electronic address:

Am J Surg Pathol 2014 Mar;38(3):402-9

Departments of *Laboratory Medicine and Pathology †Radiology, Mayo Clinic, Rochester, MN.

Parosteal osteosarcoma is a surface-based osteosarcoma that often exhibits deceptively bland cytologic features, hindering diagnosis in small biopsies or when correlative radiologic imaging is not readily available. A number of benign and malignant fibro-osseous lesions, including fibrous dysplasia (FD) and low-grade central osteosarcoma, fall within the morphologic differential diagnosis of parosteal osteosarcoma. Somatic mutations in GNAS, encoding the α-subunit of the heterotrimeric G protein complex (Gsα), occur in FD and McCune-Albright syndrome but have not been reported in parosteal osteosarcoma. Read More

J Clin Oncol 2014 May 27;32(15):e57-60. Epub 2014 Jan 27.

Children's Hospital Los Angeles; and Keck School of Medicine, University of Southern California, Los Angeles, CA

Zhonghua Bing Li Xue Za Zhi 2013 Oct;42(10):702-3

Email:

Clin Radiol 2014 Feb 5;69(2):200-8. Epub 2013 Nov 5.

Department of Radiology, Aberdeen Royal Infirmary, Foresterhill, Aberdeen, UK.

Fibro-osseous lesions of the bone are well-recognized primary bone tumours. However, given the degree of overlap of imaging findings and variation in management of various sub-types, it is a widely accepted practice to perform a biopsy to obtain histopathological confirmation of the diagnosis. The following is a summary of the epidemiology, clinicopathological features, and review of the imaging features of fibro-osseous lesions, including osteofibrous dysplasia, osteofibrous dysplasia-like adamantinoma, adamantinoma, and lesions that closely mimic them. Read More

Am J Orthop (Belle Mead NJ) 2013 Sep;42(9):420-2

Resident Physician, Department of Orthopaedic Surgery, Boston University Medical Center, Massachusetts.

We report the case of a 56-year-old woman who had severe leg pain and whose radiographs initially suggested metastatic carcinoma, lymphoma, osteogenic sarcoma, or adamantinoma. Results of multiple biopsies confirmed a diagnosis of Rosai-Dorfman disease, which typically presents in children and young adults (mean age at onset, 20 years). Read More

Zhonghua Bing Li Xue Za Zhi 2013 Jun;42(6):398-9

Acta Orthop 2013 Apr 15;84(2):224-6. Epub 2013 Feb 15.

Department of Orthopaedic Surgery, Center for Medical Imaging North East Netherlands, University Medical Center Groningen, University of Groningen, the Netherlands.

Ethiop Med J 2012 Apr;50(2):185-92

Black Lion University Hospital, Addis Ababa University, Addis Ababa, Ethiopia.

Unlabelled: Adamantinoma of long bones is an extremely rare tumor with no report, to authors' knowledge from neither Ethiopia nor Africa. We are reporting a 25 year old female college student with one and half years history of right mid leg pain and swelling with radiologically and histologically proven adamantinoma of right tibia. At CURE Hospital, she had undergone complete excision of the tumor with histologically documented clean margins. Read More

Orthopade 2011 Dec;40(12):1121-42

Klinik für Orthopädie und Sportorthopädie, Klinikum rechts der Isar, Technische Universität München, Ismaninger Straße 22, 81675, München, Deutschland.

Among human neoplasms, primary malignant bone tumors are fairly rare. They present an incidence rate of roughly 10 cases per 1 million inhabitants per year. During childhood (<15 years), the percentage of malignant bone tumors amounts to 6% of all infantile malignancies. Read More

Orthop Traumatol Surg Res 2011 Dec 11;97(8):877-80. Epub 2011 Nov 11.

Pediatric orthopedic surgery department, Saint-Vincent de Paul hospital, 74, avenue Denfert-Rochereau, 75014 Paris, France.

Because of the relative frequency of osteofibrous dysplasia (OFD) and the gravity of adamantinoma, it is important to know whether there is a link between these two entities. A young boy had been followed from the age of 5 years for OFD of the right tibia. At the age of 10, biopsy performed because of pain, revealed OFD-like adamantinoma. Read More

Clin Imaging 2011 Nov-Dec;35(6):483-7

Department of Radiology, Winthrop-University Hospital, Mineola, NY 11501, USA.

Adamantinoma is a rare primary bone malignancy with a predilection for the tibial cortex. Metastases have been reported to the long bones, lung, pleura and spine. We present a 26-year-old pregnant woman with metastatic disease to the posterior cul-de-sac, lungs, liver and retroperitoneum, which collectively have not been previously reported in a single patient to our knowledge. Read More

Clin Nucl Med 2011 Sep;36(9):788-90

Division of Nuclear Medicine, Department of Radiology and Medical Imaging, King Khalid University Hospital, King Saud University, Riyadh, Kingdom of Saudi Arabia.

J Craniofac Surg 2011 May;22(3):1163-5

Maxillo-facial Surgery Department of La Sapienza University, II Faculty of Medicine and Surgery at Sant' Andrea Hospital, Rome, Italy.

The adamantinoma is characterized by a high rate of local recurrence. Because of this peculiarity, radical treatment is generally preferred than conservative surgery. Resection with free margins is associated with lower risk of recurrence but is not recurrence free; thus, the importance of long and scrupulous follow-up is evident. Read More

J Orthop Sci 2011 Mar 16;16(2):190-5. Epub 2011 Feb 16.

Department of Orthopedic Surgery, Kyungpook National University College of Medicine, Daegu, Korea.

Background: We describe a method for the direct application of MR images to navigation-assisted bone tumor surgery as an alternative to CT-MRI fusion.

Method: Six patients with an orthopedic malignancy were employed for this method during navigation-assisted tumor resection. Tumor types included osteosarcoma (4), high-grade chondrosarcoma (1), and adamantinoma (1). Read More

Pathology 2011 Feb;43(2):174-6

Radiol Case Rep 2011 6;6(4):546. Epub 2015 Nov 6.

Osteofibrous dysplasia (OFD) is a rare bone tumor affecting young individuals. The differential diagnosis between OFD and adamantinoma may be challenging in some cases on imaging. We present a case of OFD and discuss the key imaging and histological findings. Read More

Orthopedics 2010 Nov 2;33(11):845. Epub 2010 Nov 2.

Division of Orthopedic Surgery, Kingsbrook Jewish Medical Center, Brooklyn, New York, USA.

The differential diagnosis of a tibial intracortical diaphyseal lesion includes osteoid osteoma, periosteal chondroma, nonossifying fibroma, osteofibrous dysplasia, and adamantinoma. While osteoid osteomas represent 5% of all primary bone tumors, little is understood about intracortical chondromas. Intracortical chondroma was first described in 1990 and 7 reported cases have since been published. Read More

Ann Diagn Pathol 2011 Oct 1;15(5):347-54. Epub 2010 Jun 1.

Department of Pathology, Medical College of Wisconsin, Milwaukee, 53226, United States.

Adamantinoma is a rare neoplasm that characteristically involves the tibia. In many instances, typical location within the tibia, very slow course, and a typical radiographic appearance can strongly suggest the correct diagnosis. We present a case that has both unusual radiographic findings and uncharacteristic histology. Read More

Rheumatol Int 2013 Feb 28;33(2):485-7. Epub 2010 Jul 28.

Punjab Institute of Nuclear Medicine, Jail Road, Adjacent PMC/CPSP Regional Office, Faisalabad, Pakistan.

This is a case report of a young physically active patient who presented with pain in the right leg. Plain radiograph and bone scan showed lesions mimicking stress fracture. Follow-up investigations (repeated plain radiography and three phase bone scan) and histopathological examination proved it as a case of multifocal adamantinoma. Read More

Foot (Edinb) 2009 Mar 30;19(1):58-61. Epub 2009 Jan 30.

Musculoskeletal Oncology Unit, Royal Orthopaedic Hospital, Birmingham B31 2AP, UK.

Adamantinoma is rare, representing less than 1% of all primary malignant bone tumours. It is a slow growing low grade malignant tumour which is often clinically, radiologically and histologically mistaken for many other tumours like Ewing's sarcoma, synovial sarcoma, fibrosarcoma, metastatic carcinoma, fibrous cortical defect and osteofibrous dysplasia. Adamantinoma commonly occurs in the tibia. Read More

Pathology 2010 Feb;42(2):190-3

Orthopedics 2009 Dec;32(12):926

Department of Orthopedics and Traumatology-Spine Surgery, Ospedale Maggiore C A Pizzardi, Bologna, Italy.

Adamantinoma is a rare tumor with an indolent course that occurs most commonly in the tibia. It is locally aggressive, and local recurrences are described after resection. Pain is the most common symptom. Read More

Clin Podiatr Med Surg 2010 Jan;27(1):157-65

First Department of Orthopaedics, Attikon University General Hospital, Athens University Medical School, 15123 Amarousio, Athens, Greece.

Adamantinoma of the long bones is a rare primary malignant bone tumor accounting for less than 1% of all primary malignant bone tumors. Most typical imaging findings include heterogeneous osteolytic cortical lesions showing multiple sharply circumscribed lucent zones of various sizes with sclerotic bone surroundings, interspersing between, and extending above and below the lucent zones by the thinning and bulging cortex. Advanced or recurrent lesions may be associated with destruction of the cortex and soft tissue extension. Read More

J Int Med Res 2009 Jul-Aug;37(4):1238-45

First Department of Orthopaedics, Attikon General University Hospital, Athens University Medical School, Athens, Greece.

In this case report, a 75-year old male presented with a mass on the anterior surface of the mid-shaft of the right tibia. Imaging studies showed a well-circumscribed radiolucent lesion in the anterior tibial cortex, without soft tissue extension. Plain radiographs and computed tomography scan of the chest were negative. Read More

Acta Chir Belg 2009 Jan-Feb;109(1):126-9

Orthopaedic Department Medical School, University of Ioannina, Ioannina, Greece.

We describe a case of adamantinoma of the distal tibia in a 31-year-old woman. Segmental resection of the part of the tibia containing the tumour in normal bone was done and the bone defect was bridged with bone transport and external fixation. The tibia healed uneventfully and one year after the operation the patient resumed full activity. Read More