RESUMO
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.
Assuntos
Melorreostose , Adolescente , Humanos , Melorreostose/complicações , Melorreostose/cirurgia , Melorreostose/diagnóstico , Dor , Manejo da Dor , Resultado do Tratamento , Doenças RarasRESUMO
We report a 34-year-old man who presented with hyperostosis of his right elbow associated with an inability to fully extend his elbow. The radiographic assessment revealed a classical dripping candle wax appearance of his proximal ulna suggestive of melorheostosis. Computed tomography was performed to identify the impingement point and aid in surgical planning. A targeted open excision biopsy via a Boyd incision was performed to excise the exophytic component that was causing the functional block. After surgery, he achieved full elbow extension and was able to return to his usual activity.
Assuntos
Articulação do Cotovelo , Melorreostose , Olécrano , Adulto , Cotovelo , Articulação do Cotovelo/diagnóstico por imagem , Articulação do Cotovelo/cirurgia , Humanos , Masculino , Melorreostose/diagnóstico por imagem , Melorreostose/cirurgia , Olécrano/diagnóstico por imagem , Olécrano/cirurgia , UlnaRESUMO
BACKGROUND: Melorheostosis is a bone hyperostosis disorder characterized by flowing bone thought to occur in a sclerodermal distribution. These bony lesions often arise in soft tissues adjacent to joints. Pain arises from joint limitations and impingements, but diffuse pain can also occur. CASE DESCRIPTION: We present a case of a patient with symptomatic compression of the common peroneal nerve by an extraosseous hyperostotic tissue island in a patient with melorheostosis. CONCLUSIONS: Melorheostosis is a rare bone overgrowth syndrome that can lead to joint mobility limitations and pain. In specific locations, it can also lead to tethering and symptomatic compression of the peripheral nerves.
Assuntos
Melorreostose/complicações , Síndromes de Compressão Nervosa/etiologia , Adulto , Diagnóstico Diferencial , Feminino , Fíbula , Humanos , Melorreostose/diagnóstico por imagem , Melorreostose/cirurgia , Síndromes de Compressão Nervosa/diagnóstico , Síndromes de Compressão Nervosa/cirurgia , Nervo FibularRESUMO
Melorheostosis is a very rare disorder characterized by irregular cortical thickening seen on radiographs. In this paper, we present a case of melorheostosis with microscopically confirmed soft-tissue components. The patient was a 51-year-old man who complained of severe pain in the lateral aspect of his right knee. The excision of an ossified soft-tissue lesion relieved intractable pain that had lasted 20 years. Microscopically, the cortex of the affected fibula was composed of thick compact bone and the soft-tissue component consisted of dense compact bone without endochondral ossification. The presence of soft-tissue osseous nodules around the joints is one of the specific conditions for melorheostosis and should be differentiated from synovial chondromatosis. The ossified soft-tissue lesion in our patient is to our knowledge the first reported case of the histologically confirmed soft-tissue component of melorheostosis, which differs from that of synovial chondromatosis.
Assuntos
Articulação do Joelho/diagnóstico por imagem , Melorreostose/diagnóstico por imagem , Meios de Contraste , Diagnóstico Diferencial , Humanos , Articulação do Joelho/patologia , Articulação do Joelho/cirurgia , Imageamento por Ressonância Magnética , Masculino , Melorreostose/patologia , Melorreostose/cirurgia , Pessoa de Meia-Idade , Tomografia Computadorizada por Raios XRESUMO
Melorheostosis is a rare dysplastic bone formation disease that can also affect the joints. We present a case of a patient with knee pain that was radiographically diagnosed as melorheostosis because of "dripping wax" image. An exploratory arthroscopy was made. In the joint, we found hyperplasic synovial tissue and an increased retropatellar Hoffa pad, which was surrounding an intra-articular ossification resulting from the disease. This was removed and led to a clinical and functional improvement.
Assuntos
Artroscopia , Melorreostose/patologia , Melorreostose/cirurgia , Feminino , Humanos , Hiperplasia , Articulação do Joelho/diagnóstico por imagem , Articulação do Joelho/patologia , Articulação do Joelho/cirurgia , Melorreostose/diagnóstico por imagem , Ossificação Heterotópica/diagnóstico por imagem , Ossificação Heterotópica/patologia , Ossificação Heterotópica/cirurgia , Radiografia , Sinovectomia , Membrana Sinovial/diagnóstico por imagem , Membrana Sinovial/patologia , Resultado do Tratamento , Adulto JovemRESUMO
Melorheostosis is a rare, progressive bone disease accompanied by hyperostosis and soft tissue fibrosis. While affected adults present with contracture and pain, children present with limb length discrepancy and deformity. We report the case of a 20-year-old woman with melorheostosis since childhood who presented with right hand deformity and numbness. Radiographs showed not only a combination of dense sclerosis and opacities, but also the classic 'flowing candle wax' appearance. Radiography can be used to identify melorheostosis, thus preventing unnecessary bone biopsies. Carpal tunnel release revealed the presence of a thickened flexor retinaculum and a degenerated median nerve distal to the retinaculum, but did not show hyperostosis. This case highlights the role of nerve decompression in melorheostosis and the importance of early identification of the disease to prevent unnecessary bone biopsies.
Assuntos
Síndrome do Túnel Carpal/diagnóstico por imagem , Melorreostose/diagnóstico por imagem , Síndrome do Túnel Carpal/complicações , Síndrome do Túnel Carpal/cirurgia , Diagnóstico Diferencial , Feminino , Fibrose/patologia , Mãos/patologia , Deformidades da Mão/diagnóstico por imagem , Deformidades da Mão/cirurgia , Humanos , Hipestesia , Nervo Mediano/cirurgia , Melorreostose/complicações , Melorreostose/cirurgia , Radiografia , Esclerose/diagnóstico por imagem , Adulto JovemRESUMO
BACKGROUND CONTEXT: Melorheostosis is a rare sclerosing bone disorder with a predilection for the appendicular skeleton. Involvement of the spine is infrequent and largely asymptomatic. Surgical treatment for spinal involvement is therefore uncommon with only one reported case of lumbar fusion for painful lumbosacral melorheostosis. PURPOSE: We report a case of lumbar melorheostosis causing disabling radiculopathy treated with nerve root decompression. CONCLUSIONS: Melorheostosis of the lumbar spine causing radicular symptoms has not been reported before. Our message from the management of this particular patient is to consider surgical option in symptomatic individuals.
Assuntos
Descompressão Cirúrgica , Vértebras Lombares/cirurgia , Melorreostose/complicações , Radiculopatia/etiologia , Adulto , Feminino , Humanos , Melorreostose/cirurgia , Radiculopatia/cirurgia , Raízes Nervosas Espinhais/cirurgia , Resultado do TratamentoAssuntos
Descompressão Cirúrgica , Articulação do Joelho/cirurgia , Melorreostose/cirurgia , Procedimentos Ortopédicos/métodos , Patela/cirurgia , Feminino , Humanos , Articulação do Joelho/diagnóstico por imagem , Perna (Membro)/diagnóstico por imagem , Melorreostose/diagnóstico por imagem , Radiografia , Adulto JovemRESUMO
Melorheostosis is a rare disease that usually burdens the patient with painful disability or soft tissue compromise. The treatment is usually symptomatic and conservative. Patients with severe and complicated forms of the disease may require surgery. Involvement of the distal part of a limb usually carries more morbidity, such as tumefaction pain, cosmetic and psychosocial or functional problems that render conservative treatment unsatisfactory to patients. In our series, surgical debulking or decompression of the mass effect provided prompt symptom relief.
Assuntos
Melorreostose/cirurgia , Dor/cirurgia , Doenças Raras/cirurgia , Adulto , Descompressão Cirúrgica , Feminino , Humanos , Melorreostose/complicações , Melorreostose/diagnóstico por imagem , Dor/complicações , Dor/diagnóstico por imagem , Radiografia , Doenças Raras/complicações , Doenças Raras/diagnóstico por imagem , Resultado do TratamentoRESUMO
Melorrheostosis is a rare osteosclerotic bone dysplasia of obscure etiology. The typical radiographic features are flowing candle wax, sub-periosteal bone and streaky endosteal bone formation in diaphyseal and epiphyseal area with sclerotomal pattern mainly involving appendicular skeleton. It is rarely associated with nerve palsies. The authors report a case of melorrheostotic mass causing sciatic neuropraxia and to the best of their knowledge it is the first case reported in the English language literature.
Assuntos
Melorreostose/complicações , Síndromes de Compressão Nervosa/etiologia , Nervo Isquiático/patologia , Neuropatia Ciática/etiologia , Quadril/diagnóstico por imagem , Quadril/patologia , Quadril/fisiopatologia , Humanos , Masculino , Melorreostose/cirurgia , Síndromes de Compressão Nervosa/cirurgia , Radiografia , Amplitude de Movimento Articular , Neuropatia Ciática/cirurgia , Resultado do Tratamento , Adulto JovemRESUMO
Melorheostosis is an uncommon and rare linear hyperostosis, which can be complicated by soft tissue changes. We present a case of this disorder in the humerus, clinically referred because of carpal tunnel syndrome. Although treatment is usually conservative, in this case, a neurolysis and resection of the sclerotic bone were done with good clinical result.
Assuntos
Síndrome do Túnel Carpal/etiologia , Úmero , Melorreostose , Adulto , Descompressão Cirúrgica/métodos , Denervação/métodos , Diagnóstico Diferencial , Eletrodiagnóstico , Feminino , Humanos , Imageamento por Ressonância Magnética , Melorreostose/complicações , Melorreostose/diagnóstico , Melorreostose/cirurgia , Osteotomia/métodos , Dor/etiologia , Compostos Radiofarmacêuticos , Amplitude de Movimento Articular , Doenças Raras , Medronato de Tecnécio Tc 99m , Resultado do TratamentoRESUMO
Melorheostosis is a rare disorder marked by increased bony sclerosis on radiographs. In addition to bone changes, the skin and soft tissues overlying affected bone often demonstrate increased fibrosis, which can create joint contracture. These can all affect surgical planning for a patient with melorheostosis. This article reviews the literature and describes the surgical intervention and 4-year follow-up of a 10-year-old boy with melorheostosis.
Assuntos
Deformidades Congênitas do Pé/cirurgia , Melorreostose/cirurgia , Atividades Cotidianas , Criança , Contratura/etiologia , Contratura/cirurgia , Deformidades Congênitas do Pé/diagnóstico por imagem , Deformidades Congênitas do Pé/etiologia , Hallux/cirurgia , Humanos , Ílio/transplante , Masculino , Melorreostose/complicações , Melorreostose/diagnóstico por imagem , Ossos do Metatarso/cirurgia , Osteotomia , Complicações Pós-Operatórias , Radiografia , Tendões/cirurgiaAssuntos
Pé Equino/diagnóstico , Melorreostose/diagnóstico , Neoplasias Ósseas/diagnóstico , Criança , Doença Crônica , Diagnóstico Diferencial , Pé Equino/etiologia , Pé Equino/cirurgia , Feminino , Pé/diagnóstico por imagem , Pé/patologia , Hemangioma/diagnóstico , Humanos , Imageamento por Ressonância Magnética , Melorreostose/complicações , Melorreostose/cirurgia , Osteoma/diagnóstico , Osteomielite/diagnóstico , Osteopecilose/diagnóstico , Radiografia , Resultado do TratamentoRESUMO
Melorheostosis is a rare, benign, and sporadically occurring osteosclerosis of unknown cause. The onset of the disease is usually in early adulthood. Melorheostosis affects both genders, develops progressively, and is usually limited to one side of the human body. The sclerosis originates predominantly from the cortices of the long bones of the lower limbs and rarely the upper limbs. Frequently, the sclerosis involves the soft tissue surrounding the affected bones which may cause limitations in the range of motion, contractures, deformities, and pain. Melorheostosis is usually diagnosed by radiograms. Pain relief and restoration of the full range of motion are the primary goals of the therapeutic approach. A good outcome cannot always be achieved and a recurrence of the disease happens very often.
Assuntos
Artralgia/etiologia , Artralgia/prevenção & controle , Instabilidade Articular/etiologia , Instabilidade Articular/cirurgia , Melorreostose/diagnóstico por imagem , Melorreostose/cirurgia , Adulto , Feminino , Humanos , Melorreostose/complicações , Pessoa de Meia-Idade , Radiografia , Resultado do TratamentoRESUMO
We report a case of a 47-year-old man who presented with progressive loss of motion and pain in the right shoulder. Radiographs of the shoulder demonstrated dense ossification in the glenoid and humeral head with extension into the periarticular soft tissues. CT and MRI scans confirmed the radiographic findings and also revealed ossification of the glenoid labrum. A radiographic diagnosis of melorheostosis, an uncommon benign sclerosing bone dysplasia, was made. Because of the patient's severe symptomatology, he underwent total shoulder arthroplasty. Histological analysis of the resected masses was consistent with melorheostosis with a few areas covered by a cartilage cap. This case illustrates several uncommon but important features of melorheostosis, including mechanical obstruction of joint motion requiring joint replacement, ossification of the glenoid labrum, and cartilage-covering portions of the intra-articular masses, not to be confused with cartilage-producing tumors.
Assuntos
Artropatias/diagnóstico , Melorreostose/diagnóstico , Ossificação Heterotópica/diagnóstico , Amplitude de Movimento Articular , Artroplastia/métodos , Diagnóstico Diferencial , Humanos , Artropatias/etiologia , Artropatias/cirurgia , Imageamento por Ressonância Magnética , Masculino , Melorreostose/complicações , Melorreostose/cirurgia , Pessoa de Meia-Idade , Ossificação Heterotópica/etiologia , Ossificação Heterotópica/cirurgia , Doenças Raras , Índice de Gravidade de Doença , Articulação do Ombro/diagnóstico por imagem , Articulação do Ombro/patologia , Articulação do Ombro/cirurgia , Dor de Ombro/etiologia , Dor de Ombro/cirurgia , Tomografia Computadorizada por Raios XRESUMO
Melorheostosis is a rare nongenetic developmental anomaly of the cortical bone. We present a 40-year-old woman who was diagnosed with melorheostosis affecting the ribs only. Chronic pain and cosmetic deformity were her presenting symptoms. She underwent posterolateral thoracotomy and excision of the affected ribs, with a satisfactory outcome of this uncommon presentation.
Assuntos
Melorreostose/diagnóstico por imagem , Melorreostose/cirurgia , Costelas , Adulto , Biópsia por Agulha , Doença Crônica , Feminino , Seguimentos , Humanos , Procedimentos Ortopédicos/métodos , Dor/diagnóstico , Dor/etiologia , Medição da Dor , Radiografia Torácica , Tomografia Computadorizada por Raios X , Resultado do TratamentoRESUMO
Melorheostosis is a rare condition which can cause soft tissue joint contractures. We present a case of melorheostosis causing disabling knee joint contracture, treated successfully by total knee arthroplasty.
Assuntos
Artroplastia do Joelho , Contratura/cirurgia , Articulação do Joelho/cirurgia , Melorreostose/cirurgia , Adulto , Contratura/fisiopatologia , Feminino , Humanos , Articulação do Joelho/fisiopatologia , Melorreostose/fisiopatologiaRESUMO
INTRODUCTION: Melorheostosis is a rare bony dysplasia and often recognised just sporadically by chance. CASE REPORT: We present a case of a 15 year old girl who presented a melorheostosis of the left foot. After birth there was recognized a shortening and deformity of the 2nd toe on the left foot. Furthermore she had an interphalangeal hallux valgus that displaced the 2nd toe increasingly. Thus in the last years there were more and more difficulties to wear normal shoes. Conservative therapy was not successful. We performed a lengthening extending osteotomy of the 2nd toe (a modified Weil osteotomy) and an Akin osteotomy of the interphalangeal hallux valgus. Since the surgical procedure the patient is out of any complaints. DISCUSSION: We demonstrate the radiologic and histologic findings and discuss the relevant literature and possible etiology.
Assuntos
Fios Ortopédicos , Deformidades Congênitas do Pé/cirurgia , Melorreostose/congênito , Osteotomia , Adolescente , Osso e Ossos/patologia , Feminino , Deformidades Congênitas do Pé/patologia , Humanos , Melorreostose/patologia , Melorreostose/cirurgia , Periósteo/patologiaRESUMO
A 24-year-old female developed, in infancy, progressive right upper and lower limb muscle and soft tissue contractures and had a diagnosis of melorheostosis made on X-ray and pathological specimens. At the age of 11 years she began to have pain in the right hip and lower limb and this later became the dominant feature. She ultimately required amputation through the right hip joint and prosthetic fitting. She now has independent mobility with her prosthesis and has had no recurrence of pain. Her right arm remains flexed, shortened and contracted, but some hand function is retained. A review of the medical literature is discussed.