ReviewCurrent osteoporosis reports2017
Melorheostosis: a Rare Sclerosing Bone Dysplasia.
Review in Current osteoporosis reports, 2017. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 30 papers.
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Who cites it
30 citing papers in PubMed, 67 citations in OpenAlex.
- Melorheostosis: A Clinical, Pathologic, and Radiologic Case Series.The American journal of surgical pathology · 2019Trial
- Melorheostotic Bone Lesions Caused by Somatic Mutations in MAP2K1 Have Deteriorated Microarchitecture and Periosteal Reaction.Journal of bone and mineral research : the official journal of the American Society for Bone and Mineral Research · 2019Trial
- Melorheostosis: A Case Report on the Atypical Clinical Presentation and Management of a Rare Sclerosing Bone Disease.Cureus · 2026Article
- Zoledronic Acid in the Management of Melorheostosis of Radius and Ulna - A Rare Case Report with Literature Review.Journal of orthopaedic case reports · 2026Article
- Case Report: Combined posterolateral and direct anterior approaches for total hip arthroplasty in osteonecrosis of the femoral head with a rare massive osteophyte with literature review.Frontiers in surgery · 2026Article
- The melorheostosis with severe knee and ankle deformity treated by total knee arthroplasty with V-Y quadricepsplasty: a case report and literature review.BMC musculoskeletal disorders · 2025Review
- Melorheostosis: Clinical, radiological, and histopathological features with a literature review.Physiological reports · 2025Review
- Melorheostosis: A Rare Case Report with Pelvic and Lumbar Involvement.Journal of orthopaedic case reports · 2025Article
- Unraveling melorheostosis: insights into clinical features, diagnosis, and treatment.JBMR plus · 2025Review
- Clinical report of metacarpal melorheostosis: a rare disease with "the dripping candle wax" appearance on different imaging modalities.Oxford medical case reports · 2024Article
- Melorheostosis of the cervical and cervicothoracic spine: review of the literature and presentation of 3 new cases.Archives of orthopaedic and trauma surgery · 2024Review
- Bone Material Properties in Bone Diseases Affecting Children.Current osteoporosis reports · 2023Review
- Melorheostosis: A Review of the Literature and a Case Report.Medicina (Kaunas, Lithuania) · 2023Review
- Article
- Review
- Clinical characteristics of 10 Chinese patients with melorheostosis and identification of a somatic MAP2K1 variant in one case.Molecular genetics & genomic medicine · 2022Article
- Clinical and Histopathological Features of Scleroderma-like Disorders: An Update.Medicina (Kaunas, Lithuania) · 2021Review
- Bisphosphonate Therapy in the Management of Symptomatic Melorheostosis of Tibia.Journal of orthopaedic case reports · 2021Article
- Osteoblast Dysfunction in Non-Hereditary Sclerosing Bone Diseases.International journal of molecular sciences · 2021Review
- The combined prevalence of classified rare rheumatic diseases is almost double that of ankylosing spondylitis.Orphanet journal of rare diseases · 2021Article
Corrections and comments
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Authors and funding
2 authors at 2 institutions in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
purpose of reviewMelorheostosis is a rare sclerosing bone dysplasia that affects both cortical bone and adjacent soft tissue structures in a sclerotomal distribution. In this review, we describe the natural history, radiological features, proposed pathogenesis, and management options for this debilitating condition. RECENT
findingsSince its first description in 1922, about 400 cases of melorheostosis have been reported, either as single reports or in small case series. Melorheostosis affects the appendicular skeleton more commonly than the axial skeleton and usually presents with lower limb deformity. Diagnosis is based on a combination of clinical and radiological features that help differentiate this condition from other sclerosing bone dysplasias. LEM domain-containing protein 3 (LEMD3) gene mutations have been demonstrated in several familial cases, but these have been more strongly correlated with other hereditary dysplasias, such as osteopoikilosis, and are not thought to be the causative gene for melorheostosis. The exact etiology of classic sporadically occurring melorheostosis remains unknown, with possible causes being somatic LEMD3 mutations, somatic mutations in the bone morphogenetic protein/transforming growth factor-beta pathway, mutations in multiple genes, or other non-genetic causes. Management in recent years has involved nitrogen-containing bisphosphonates in addition to traditional orthopedic surgical approaches and physical therapy. Melorheostosis may present as mixed or atypical osseous involvement in addition to the classically described "dripping candle wax" appearance of hyperostosis. Some patients may have overlap with osteopoikilosis or Buschke-Ollendorff syndrome. In the future, better characterization of genetic and developmental factors predisposing to melorheostosis may lead to the development of targeted therapy for this condition, as well as for more commonly encountered skeletal abnormalities.
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