A focus on melorheostosis disease: a literature review and case report of femoral-acetabular impingement due to melorheostosis treated with surgical hip osteoplasty

Submitted: 22 July 2023
Accepted: 25 October 2023
Published: 22 March 2024
Abstract Views: 259
PDF: 12
Publisher's note
All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article or claim that may be made by its manufacturer is not guaranteed or endorsed by the publisher.


Objective. Melorheostosis is a rare, non-hereditary, benign bone disease characterized by abnormal bone growth. Generally, melorheostosis develops during childhood or adolescence and progresses gradually over time. This disease represents a true challenge to the physician because of its variability due to location, extension of the affected bone, and involvement of associated soft tissue. Pain management, physical therapy, and surgery may be recommended, depending on the individual case. This review aims to get an overview of the latest evidence relating to epidemiology, clinical and radiographic characteristics, diagnosis, and possible therapeutic strategies for melorheostosis and describe our experience through a clinical case.

Methods. We designed a comprehensive literature search on melorheostosis in MEDLINE (via Pubmed) up to April 2023 and reviewed reports published in international journals.

Results. The purpose is to highlight the importance of a multidisciplinary approach in the management of a rare disease such as melorheostosis. We discuss the role of different physicians, including genetists, rheumatologists, physiatrists, physical therapists, and orthopedic surgeons, in providing accurate diagnoses and effective treatments. We conducted a comprehensive review of the literature on the treatment of melorheostosis to support these findings. In addition, the article presents a case study of a patient suffering from melorheostosis, focusing on difficulties in reaching a correct diagnosis and attempts towards conservative and surgical interventions. The patient underwent hip arthroplasty, and the final result was an improvement in function and a reduction in pain.

Conclusions. Managing melorheostosis can be challenging, and there is no standardized treatment for this condition at the moment.



PlumX Metrics


Download data is not yet available.


Vaishya R, Vaish A. Melorheostosis: dripping candle wax disease. Indian J Med Res 2020; 152: S45-6. DOI: https://doi.org/10.4103/ijmr.IJMR_1745_19
Kotwal A, Clarke BL. Melorheostosis: a rare sclerosing bone dysplasia. Curr Osteoporos Rep 2017; 4: 335-42. DOI: https://doi.org/10.1007/s11914-017-0375-y
Léri A, Joanny M. Une affection non décrite des os: Hyperostose “en ember” sur toute la longueur d’un ember ou “mélorhéostose”. Bull Mem Soc Med Hop (Paris) 1922; 46: 1141-5.
Suresh S, Muthukumar T, Saifuddin A. Classical and unusual imaging appearances of melorheostosis. Clin Radiol 2010; 65: 593-600. DOI: https://doi.org/10.1016/j.crad.2010.02.004
Iordache S, Cursaru A, Serban B, Costache M, Spiridonica R, Cretu B, et al. Melorheostosis: a review of the literature and a case report. Medicina (Kaunas) 2023; 59: 869. DOI: https://doi.org/10.3390/medicina59050869
Gasparyan AY, Ayvazyan L, Blackmore H, Kitas GD. Writing a narrative biomedical review: considerations for authors, peer reviewers, and editors. Rheumatol Int 2011; 31: 1409-17. DOI: https://doi.org/10.1007/s00296-011-1999-3
Zhang C, Dai W, Yang Y, Tang Q, Yao Z. Melorheostosis and a review of the literature in China. Intractable Rare Dis Res 2013; 2: 51-4. DOI: https://doi.org/10.5582/irdr.2013.v2.2.51
Wordsworth P, Chan M. Melorheostosis and osteopoikilosis: a review of clinical features and pathogenesis. Calcif Tissue Int 2019; 104: 530-43. DOI: https://doi.org/10.1007/s00223-019-00543-y
Happle R. Melorheostosis may originate as a type 2 segmental manifestation of osteopoikilosis. Am J Med Genet A 2004; 125A: 221-3. DOI: https://doi.org/10.1002/ajmg.a.20454
Murray RO, McCredie J. Melorheostosis and the sclerotomes: a radiological correlation. Skeletal Radiol 1979; 4: 57-71. DOI: https://doi.org/10.1007/BF00349329
Fryns JP. Melorheostosis and somatic mosaicism. Am J Med Genet 1995; 58: 199. DOI: https://doi.org/10.1002/ajmg.1320580221
Jha S, Fratzl-Zelman N, Roschger P, Papadakis GZ, Cowen EW, Kang H, et al. Distinct clinical and pathological features of melorheostosis associated with somatic MAP2K1 mutations. J Bone Miner Res 2019; 34: 145-56. DOI: https://doi.org/10.1002/jbmr.3577
Sinigaglia L. Metabolic bone diseases: an overview. Reumatismo 2014; 66: 109-11. DOI: https://doi.org/10.4081/reumatismo.2014.783
Chimenti MS, Di Stefani A, Conigliaro P, Saggini A, Urbani S, Giunta A, et al. Histopathology of the skin in rheumatic diseases. Reumatismo 2018; 70: 187-98. DOI: https://doi.org/10.4081/reumatismo.2018.1049
Ihde LL, Forrester DM, Gottsegen CJ, Masih S, Patel DB, Vachon LA, et al. Sclerosing bone dysplasias: review and differentiation from other causes of osteosclerosis. Radiographics 2011; 31: 1865-82. DOI: https://doi.org/10.1148/rg.317115093
Freyschmidt J. Melorheostosis: a review of 23 cases. Eur Radiol 2001; 11: 474-9. DOI: https://doi.org/10.1007/s003300000562
Smith GC, Pingree MJ, Freeman LA, Matsumoto JM, Howe BM, Kannas SN, et al. Melorheostosis: a retrospective clinical analysis of 24 patients at the Mayo Clinic. PM R 2017; 9: 283-8. DOI: https://doi.org/10.1016/j.pmrj.2016.07.530
Davis DC, Syklawer R, Cole RL. Melorheostosis on three-phase bone scintigraphy. Case report. Clin Nucl Med 1992; 17: 561-4. DOI: https://doi.org/10.1097/00003072-199207000-00006
Fornasari D, Coaccioli S. Pharmacology of pain. Reumatismo 2014; 66: 14-7. DOI: https://doi.org/10.4081/reumatismo.2014.759
Sarzi-Puttini P, Atzeni F, Lanata L, Bagnasco M, Colombo M, Fischer F, et al. Pain and ketoprofen: what is its role in clinical practice? Reumatismo 2010; 62: 172-88. DOI: https://doi.org/10.4081/reumatismo.2010.172
Shin SJ, Nam U, Kim SR, Kim HJ, Dimitriou D, Li G, et al. Vascular malformations corresponding to sclerotomes in multifocal melorheostosis: painful hip and knee contractures treated with total joint arthroplasty: a case report. JBJS Case Connect 2015; 5: e40. DOI: https://doi.org/10.2106/JBJS.CC.N.00159
Gagliardi GG, Mahan KT. Melorheostosis: a literature review and case report with surgical considerations. J Foot Ankle Surg 2010; 49: 80-5. DOI: https://doi.org/10.1053/j.jfas.2009.08.004
Donáth J, Poór G, Kiss C, Fornet B, Genant H. Atypical form of active melorheostosis and its treatment with bisphosphonate. Skeletal Radiol 2002; 31: 709-13. DOI: https://doi.org/10.1007/s00256-002-0576-y
Slimani S, Nezzar A, Makhloufi H. Successful treatment of pain in melorheostosis with zoledronate, with improvement on bone scintigraphy. BMJ Case Rep 2013; 2013: bcr2013009820. DOI: https://doi.org/10.1136/bcr-2013-009820
Gnoli M, Staals EL, Campanacci L, Bedeschi F, Faletra F, Gallone S, et al. Melorheostosis and osteopoikilosis clinical and molecular description of an Italian case series. Calcif Tissue Int 2019; 105: 215-21. DOI: https://doi.org/10.1007/s00223-019-00565-6
Lurati A, Laria A, Faggioli P, Tamburello A, Castelnovo L, Mazzone A. Neridronate in bone marrow edema syndrome. Efficacy and safety of two therapeutic regimens. Beyond Rheumatology 2020; 2: e273. DOI: https://doi.org/10.4081/br.2020.34
Molfetta L, Florian A, Saviola G, Frediani B. Bone marrow edema: pathogenetic features. Clin Ter 2022; 173: 434-9.

How to Cite

Ruggiero, M., Conforti, A., Culcasi, A., Mazzanti, C., Sibahi, G., Rani, N., & Sartini, S. (2024). A focus on melorheostosis disease: a literature review and case report of femoral-acetabular impingement due to melorheostosis treated with surgical hip osteoplasty. Reumatismo, 76(1). https://doi.org/10.4081/reumatismo.2024.1621