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J Craniofac Surg 2022 May 12;33(3):e342-e345. Epub 2021 Oct 12.

Department of Plastic and Reconstructive Surgery, Seoul National University Bundang Hospital, Seongnam.

Background: Fibrous dysplasia (FD) is a rare sporadic benign disease, which involves from single to several bones with unilateral distribution. Recently, image-based surgical navigation systems have played a significant role in surgical process on neurological and orthopedic operations. However, because an intraoral approach can visualize the field for maxillary surgery, there are few cases using endoscopes for excision of FD. Read More

Indian J Radiol Imaging 2022 Mar 1;32(1):145-148. Epub 2022 May 1.

Department of Radiodiagnosis and Imaging, All India Institute of Medical Sciences, Rishikesh, Uttarakhand, India.

Fibrous dysplasia has various ways of presentation including syndromic associations and secondary changes within the lesion. We present a case of a 21-year-old female with craniofacial fibrous dysplasia, presenting with proptosis and intermittent blurring of vision due to focal fibrous dysplasia involving the frontal bone with secondary aneurysmal bone formation that was provisionally diagnosed on imaging and confirmed on histopathology. This case demonstrates the typical imaging findings of fibrous dysplasia with seldom encountered secondary aneurysmal bone cyst formation and also discusses about the pathology and management of the craniofacial fibrous dysplasia. Read More

J Equine Vet Sci 2022 Jun 15:104048. Epub 2022 Jun 15.

D.V.M., M.Sc., University of Teramo, Faculty of Veterinary Medicine, Loc. Piano d'Accio, 64100, Teramo, Italy.

A 11-year-old, Haflinger breed mare developed a slowly growing mass, protruding on the ventral aspect of the mandible. On palpation, the lesion was pedunculated, very firm, cold and not painful. Radiographic examinations showed a radiopaque, multi-lobular, "mushroom-like" lesion, which arose from the mandibular cortex without involving the bone marrow cavity. Read More

J Bone Miner Res 2022 Jun 13. Epub 2022 Jun 13.

Metabolic Bone Disorders Unit, National Institute of Dental and Craniofacial Research, National Institutes of Health, Bethesda, MD, USA.

Fibrous dysplasia (FD) is a rare mosaic disorder resulting in fractures, pain, and disability. Bone lesions appear during childhood and expand during skeletal growth. The rate at which FD lesions progress and the biochemical determinants of FD lesion formation have not been established, making it difficult to investigate and implement preventative therapies. Read More

Cureus 2022 Mar 23;14(3):e23428. Epub 2022 Mar 23.

Radiology, Buraidah Central Hospital., Buraidah, SAU.

Uremic leontiasis ossea (ULO) is a rare disease characterized by extensive thickening of the cranium, resulting in a characteristic, lion-like facial appearance. It is considered the most severe osseous complication of renal dystrophy. Although rare, ULO can occur even in young patients, which can be catastrophic, as it can not only lead to life-threatening conditions but also multiple complications that cause severe determent to the quality of life. Read More

Front Med (Lausanne) 2022 15;9:857079. Epub 2022 Mar 15.

Department of Anesthesiology, Critical Care and Pain Medicine, Boston Children's Hospital and Harvard Medical School, Boston, MA, United States.

Patients diagnosed with McCune-Albright Syndrome (MAS) frequently manifest craniofacial fibrous dysplasia (FD). Craniofacial FD can impinge nerve fibers causing visual loss as well as craniofacial pain. Surgical decompression of affected nerves is performed, with variable efficacy, in an attempt to restore function or alleviate symptoms. Read More

Front Neurol 2022 16;13:855157. Epub 2022 Mar 16.

Department of Anesthesiology, Critical Care and Pain Medicine, Boston Children's Hospital and Harvard Medical School, Boston, MA, United States.

Patients with fibrous dysplasia (FD) often present with craniofacial lesions that affect the trigeminal nerve system. Debilitating pain, headache, and migraine are frequently experienced by FD patients with poor prognosis, while some individuals with similar bone lesions are asymptomatic. The clinical and biological factors that contribute to the etiopathogenesis of pain in craniofacial FD are largely unknown. Read More

Korean J Intern Med 2022 05 29;37(3):695-696. Epub 2022 Mar 29.

Faculty of Medicine, School of Medicine, National Yang Ming Chiao Tung University, Taipei, Taiwan.

Arch Oral Biol 2022 May 4;137:105389. Epub 2022 Mar 4.

Adelaide Dental School, The University of Adelaide, Adelaide, SA, Australia; South Australian Health and Medical Research Institute, Adelaide, SA, Australia; Mesenchymal Stem Cell Laboratory, School of Biomedicine, The University of Adelaide, Adelaide, SA, Australia; Cleft and Craniofacial SA, Women's and Children's Hospital, Adelaide, SA, Australia.

Objective: EFNB1 mutation causes craniofrontonasal dysplasia (CFND), a congenital syndrome associated with craniomaxillofacial anomalies characterised by coronal craniosynostosis, orbital hypertelorism, and midface dysplasia. The aim of this murine study was to investigate the effect of the EfnB1 conditional gene deletion in osteoprogenitor cells on the craniomaxillofacial skeletal morphology.

Design: The skulls of male and female mice, in which EfnB1 was deleted by Cre (a site-specific DNA recombinase) under the control of the Osterix (Osx) promoter (EfnB1), were compared to those without EfnB1 deletion (Osx:Cre control) at two ages (4 and 8 weeks; n = 6 per group). Read More

Quant Imaging Med Surg 2022 Mar;12(3):1988-2001

Department of Radiology, The University of Hong Kong-Shenzhen Hospital, Shenzhen, China.

Background: This study evaluated the clinical characteristics and imaging findings of 112 patients with irregular and flat bone osteosarcoma (IFBO).

Methods: The age, gender, location, tumor size, density and signal intensity, osteoid matrix, periosteal reaction, and histological subtypes were analyzed for 112 patients with IFBO.

Results: A total of 112 patients with IFBO, including 64 males and 48 females, with a mean age of 34. Read More

Neurol India 2022 Jan-Feb;70(1):174-175

Department of Anaesthesia and Critical Care, PGIMER, Chandigarh, India.

Am J Otolaryngol 2022 May-Jun;43(3):103404. Epub 2022 Feb 26.

Weill Cornell Medical College, Department of Otolaryngology - Head and Neck Surgery, 2315 Broadway, 3rd Floor, New York, NY 10024, United States of America. Electronic address:

Benign bony tumors of the skull base and paranasal sinuses are uncommon entities, with an overall higher incidence in males. Benign bony tumors may lead to local expansion with resultant mass effect of potentially critical structures. Some benign bony tumors may undergo malignant transformation. Read More

Head Neck Pathol 2022 Feb 26. Epub 2022 Feb 26.

Department of Oral Pathology and Microbiology, Centre for Dental Education and Research, All India Institute of Medical Sciences, Delhi, Pin 110029, India.

Benign fibro-osseous lesions (BFOLs) are a diverse group of lesions showing considerable degree of overlap with low grade osteosarcoma (LGOS). Further, de-differentiated osteosarcoma (DOS) is usually indistinguishable from conventional high-grade OS (COS) if LGOS foci are not identified. Thus, there is a need for adjunctive immunohistochemical markers to differentiate OS from benign FOLs as well as DOS from COS. Read More

J Pers Med 2022 Jan 31;12(2). Epub 2022 Jan 31.

Department of Cardiovascular and Thoracic Surgery, "Iuliu Hatieganu" University of Medicine and Pharmacy Cluj Napoca, 8, Victor Babes, St., 400012 Cluj-Napoca, Romania.

Arrhythmogenic right ventricular dysplasia (ARVD) is a rare genetic condition of the myocardium, with a significantly high risk of sudden death. Recent genetic research and improved understanding of the pathophysiology tend to change the ARVD definition towards a larger spectrum of myocardial involvement, which includes, in various proportions, both the right (RV) and left ventricle (LV), currently referred to as ACM (arrhythmogenic cardiomyopathy). Its pathological substrate is defined by the replacement of the ventricular myocardium with fibrous adipose tissue that further leads to inadequate electrical impulses and translates into varies degrees of malignant ventricular arrythmias and dyskinetic myocardium movements. Read More

Int J Environ Res Public Health 2022 02 9;19(4). Epub 2022 Feb 9.

Oral and Maxillofacial Radiology, Applied Oral Sciences and Community Dental Care, Faculty of Dentistry, University of Hong Kong, Hong Kong, China.

(1) Background: Many radiolucent jaw lesions exist, and they often show a radiographic resemblance, rendering diagnosis a challenging act. Closely related lesions should be frequently mentioned together in the academic literature, which might be helpful for junior practitioners in determining their differential diagnosis. The usefulness of bibliometric analysis in this respect has yet to be demonstrated. Read More

Diagnostics (Basel) 2022 Jan 19;12(2). Epub 2022 Jan 19.

Clinic of Cranio-Maxillofacial and Oral Surgery, Center of Dental Medicine, University of Zurich, 8032 Zurich, Switzerland.

Background: Fibro-osseous lesions (FOL) of the jaw represent a rare, benign group of lesions that share similar clinical, radiological, and histopathological features and are characterized by progressive, variable replacement of healthy bone tissue by fibrous connective tissue.

Methods: This retrospective study aimed to evaluate the incidence of fibro-osseous lesions and to reassess the efficacy of case-specific treatment management from a clinical, radiological, and histopathological perspective based on 14 years of data.

Results: Forty-four patients with a radiological and/or histopathological diagnosis of benign FOLs were identified and re-evaluated. Read More

J Pediatr Orthop 2022 May-Jun 01;42(5):e492-e500

Department of Orthopaedic Surgery, Pediatric Hospital "Bambino Gesù", Palidoro, Rome, Italy.

Background: In children, intramedullary nailing (IN) has been proposed as the best treatment when the femur and tibia are totally affected by fibrous dysplasia (FD). However, in younger children IN must be repeated to maintain stabilization of the affected skeletal segment during growth. We report the long-term results in a cohort of patients in whom more than two-thirds of cases had IN repeated during growth. Read More

J Craniofac Surg 2022 Feb 24. Epub 2022 Feb 24.

Department of Otolaryngology-Head and Neck Surgery, Chonnam National University Medical School & Hwasun Hospital, Hwasun, South Korea.

Introduction: Fibrous dysplasia (FD) is a benign, idiopathic skeletal disorder, in which the normal bone and marrow are replaced by fibrous tissue and immature woven bone. The authors aim to analyze the long-term results of patients diagnosed with FD of the paranasal sinuses via imaging or surgical biopsy.

Methods: The authors retrospectively enrolled 21 patients with FD of the paranasal sinuses from 2006 to 2021. Read More

Beijing Da Xue Xue Bao Yi Xue Ban 2022 Feb;54(1):54-61

Department of Oral Pathology, Peking University School and Hospital of Stomatology & National Center of Stomatology & National Clinical Research Center for Oral Diseases & National Engineering Laboratory for Digital and Material Technology of Stomatology & Beijing Key Laboratory of Digital Stomatology, Beijing 100081, China.

Objective: To compare the clinicopathologic features and prognosis of the different types of fibrous dysplasia (FD) of cranio-maxillofacial region, so as to provide a new reference for clinicians to treat these patients and make prognostic judgement.

Methods: Clinical records, radiographic data and pathological information of 105 patients diagnosed with FD or McCune-Albright syndrome (MAS) at the Department of Oral Pathology, Peking University Hospital of Stomatology from January 2013 to December 2020 were collected. The patients were divided into 4 groups: monostotic FDs, polyostotic FDs, MAS and a specific type called craniofacial fibrous dysplasia (CFD) limited in the craniofacial region. Read More

Medicine (Baltimore) 2022 Feb;101(6):e28815

Department of Pediatrics, Graduate School of Medicine, Gifu University, Yanagido, Gifu, Gifu, Japan.

Rationale: McCune-Albright syndrome (MAS) is a rare disorder characterized by clinical findings, which includes fibrous dysplasia (FD). FD is a benign tumor that leads to increased rates of bone fracture. In some MAS cases with FD, facial deformities, severe pain, and orbital neuropathies are complicated. Read More

Radiol Case Rep 2022 Mar 31;17(3):825-831. Epub 2021 Dec 31.

Department of Radiology, AORN Santobono Pausilipon, Pediatric Hospital, Naples, Italy.

Osteofibrous dysplasia (OFD) is a nonneoplastic tumor-like lesion, made up of fibrous matrix with immature bone tissue surrounded by osteoblasts, occurring usually in the cortex of tibial diaphysis. OFD is usually seen in the first decade of life and, according to literature, it is rarely seen in the newborn period. Diagnosis of congenital OFD in the newborn is challenging because it is uncommon in this age group and can be confused with other bone benign or malignant lesions. Read More

Imaging Sci Dent 2021 Dec 11;51(4):461-466. Epub 2021 Aug 11.

Department of Oral Medicine and Radiology, New Horizon Dental College and Research Institute, Bilaspur, Chhattisgarh, India.

Fibro-osseous lesions are a poorly defined category of conditions affecting the jaws and craniofacial bones, and include developmental lesions, reactive or dysplastic lesions, and neoplasms. Fibrous dysplasia and ossifying fibroma are the 2 main types of fibro-osseous lesions affecting the jaw, and ossifying fibroma is a true benign neoplasm of the bone-forming tissues with several well-recognized variants ranging from innocuous to extensively aggressive lesions. However, multiple simultaneous fibro-osseous lesions of the jaw bones involving all quadrants are exceedingly rare. Read More

Br J Oral Maxillofac Surg 2022 02 31;60(2):162-167. Epub 2021 Mar 31.

Institute of Forming Technology & Equipment, Shanghai Jiao Tong University, Shanghai, China; Institute of Medical Robotics, Shanghai Jiao Tong University, Shanghai, China; Quanzhou Normal University, Fujian, China. Electronic address:

The aim of this study was to apply an augmented reality (AR) navigation technique based on a head- mounted display in the treatment of craniofacial fibrous dysplasia and to explore the feasibility and the value of AR in craniofacial surgery. With preoperative planning and three-dimensional simulation, the normal anatomical contours of the deformed area were recreated by superimposing the unaffected side on to the affected side. We completed the recontouring procedures in real time with the aid of an AR navigation system. Read More

Zhongguo Xiu Fu Chong Jian Wai Ke Za Zhi 2021 Dec;35(12):1525-1530

Department of Joint Surgery, the First People's Hospital of Wenling, Wenling Zhejiang, 317500, P.R.China.

Objective: To investigate the effectiveness of sliding trochanteric osteotomy (STO) in total hip arthroplasty (THA) for Crowe type Ⅳ developmental dysplasia of the hip (DDH).

Methods: Between July 2016 and April 2021, 52 patients (57 hips) with Crowe type Ⅳ DDH who underwent primary THA were enrolled according to inclusion criteria. There were 5 males and 47 females with a median age of 51 years (range, 18-76 years). Read More

Medicine (Baltimore) 2021 Dec;100(49):e28138

Department of Orthopedic Surgery, Nagoya University Graduate School and School of Medicine, 65 Tsurumai, Showa, Nagoya, Japan.

Rationale: Fibrous dysplasia is a rare disorder that results in fractures, pain, and disability and can affect any bone in the body. The treatment of symptomatic fibrous dysplasia is determined based on the affected bones. Although some lesions are often too extensive for surgical procedures, there are currently no effective or recommended medical treatments available for them. Read More

Bone 2022 02 4;155:116270. Epub 2021 Dec 4.

Department of Drug Design and Pharmacology, University of Copenhagen, Copenhagen, Denmark. Electronic address:

Background: Fibrous dysplasia (FD) is a rare genetic bone disorder resulting in an overproduction of cAMP leading to a structurally unsound tissue, caused by a genetic mutation in the guanine nucleotide-binding protein gene (GNAS). In order to better understand this disease, several animal models have been developed with different strategies and features.

Objective: Conduct a systematic review to analyze and compare animal models with the causative mutation and features of FD. Read More

JBJS Case Connect 2021 11 22;11(4). Epub 2021 Nov 22.

Shriners Hospitals for Children-Portland, Portland, Oregon.

Case: We report an 18-year-old patient with a clinical phenotype consistent with severe osteogenesis imperfecta (OI) with frequent fractures, short stature, shortening and bowing of extremities, and unusual radiographic features of severe fibrous dysplasia, including lytic lesions and a "ground-glass" appearance. Genetic testing for the patient was notable for a c.119C>T (p. Read More

Surg Pathol Clin 2021 Dec 7;14(4):707-721. Epub 2021 Oct 7.

Department of Orthopaedic Oncology, Harvard Medical School, Massachusetts General Hospital, 55 Fruit Street, Boston, MA 02114, USA.

Fibrous and fibro-osseous tumors are some of the most common benign lesions involving bones. Although many of the histomorphologic features of these tumors overlap significantly, an interdisciplinary approach helps to consolidate the classification of these tumors. Herein, the clinical, radiologic, and pathologic features of lesions within these categories are described. Read More

Acta Ortop Mex 2021 Mar-Apr;35(2):221-225

Anatomía Patológica. Centro Médico ABC. México.

Mazabraud syndrome was first described in 1926 by Henschen, consisting of the association between bone fibrodysplasia and one or more intramuscular myxomas. The study paper conducted by Mazabraud et al. in 1967, described an association between the two pathologies. Read More