Melorheostosis

01/01/2010 - "Melorheostosis and its treatment with intravenous zoledronic acid."
06/21/2013 - "We report an adult patient with a very painful melorheostosis, who  improved after treatment with zoledronate, either on symptoms or on bone scans. "
06/21/2013 - "Successful treatment of pain in melorheostosis with zoledronate, with improvement on bone scintigraphy."
01/01/2010 - "We suggest that melorheostosis can be treated with intravenous zoledronic acid and that treatment can be monitored by the use of a specific bone resorption marker."
01/01/2010 - "We report a case of melorheostosis, a rare bone disorder characterised by mesodermal dysplasia, and its successful and prolonged treatment with the intravenous bisphosphonate zoledronic acid. "

07/01/1986 - "Successful symptomatic treatment of melorheostosis with nifedipine."
07/01/1986 - "We describe a patient with melorheostosis who showed improvement in pain and vasomotor function after treatment with nifedipine. "

03/01/2009 - "Report a new case of melorheostosis of the ribs improved by pamidronate infusions A 36-year-old man without any medical history was admitted for a history of one month painful tumefaction on the 7th left rib. "
03/01/2009 - "Atypical form of melorheostosis improved by pamidronate."

09/27/2018 - "To the best of our knowledge, this is the first case report on denosumab treatment for melorheostosis. "
09/27/2018 - "Clinical improvement in a patient with monostotic melorheostosis after treatment with denosumab: a case report."
09/27/2018 - "This is the first report indicating that denosumab has a place in the treatment of melorheostosis when the effect of bisphosphonate treatment is insufficient."
09/27/2018 - "A 20-year-old Danish woman with melorheostosis in her right femoral shaft and disabling pain in the affected area, whose symptoms did not in the long term respond to zoledronic acid, experienced continuous remission of pain after treatment with denosumab. "

05/01/1978 - "Technetium-99m pyrophosphate bone images should prove helpful when superimposed osseous disease must be differentiated from osteopoikilosis, osteopathia striata, or melorheostosis."
05/01/1978 - "Technetium-99m pyrophosphate bone images were contrasted with radiographic findings in two patients with osteopoikilosis, one with osteopathia striata, and one with melorheostosis. "

01/01/2017 - "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. "
03/01/2000 - "Taken together, our results suggest that altered expression of several adhesion proteins may contribute to the development of hyperostosis and concomitant soft tissue abnormalities of melorheostosis, with betaig-h3 in particular playing an important role in osteogenesis."
08/01/2007 - "Recent studies have reported loss of function mutations in the LEMD3 gene, encoding an inner nuclear membrane protein that influences Smad signaling, as a cause of osteopoikilosis, Buschke-Ollendorff syndrome, and melorheostosis. "

12/01/2020 - "We herein report a case of melorheostosis affecting left femur and tibia which was diagnosed based on clinical and radiological features and managed with bisphosphonates. "
12/01/2002 - "Atypical form of active melorheostosis and its treatment with bisphosphonate."
09/01/2021 - "Bisphosphonate Therapy in the Management of Symptomatic Melorheostosis of Tibia."
09/27/2018 - "This is the first report indicating that denosumab has a place in the treatment of melorheostosis when the effect of bisphosphonate treatment is insufficient."
01/01/2010 - "We report a case of melorheostosis, a rare bone disorder characterised by mesodermal dysplasia, and its successful and prolonged treatment with the intravenous bisphosphonate zoledronic acid. "

04/01/2009 - "The diagnosis of melorheostosis was made on the basis of the characteristic distribution, location, and combined radiographic, CT, and radionuclide imaging features of the abnormalities. "
06/01/1994 - "Radionuclide imaging in forme fruste of melorheostosis."
08/01/2001 - "We retrospectively reviewed the CT (n=7) and/or MR imaging findings (n=12) of 17 patients with characteristic radiographic findings of melorheostosis (undulating cortical hyperostosis with marked uptake on radionuclide bone scintigraphy). "
01/01/2000 - "It must be distinguished from melorheostosis, osteopathia striata and fundamentally from osteoblastic bone metastases, on the basis of the clinical, radiological (roentgenographs, computed tomography and magnetic resonance) and radionuclide scanning characteristics. "
06/01/1994 - "Radionuclide bone scans demonstrated a moderately increased uptake of radiopharmaceutical localized to the "flowing" cortical hyperostosis of melorheostosis observed radiographically. "

09/22/2023 - "VEGF Secretion Drives Bone Formation in Classical MAP2K1+ Melorheostosis."
01/01/2022 - "In conclusion, this work identifies the role of several important molecular mediators in the pathogenesis of melorheostosis, including MAPK/ERK kinase 1, phosphorylated ERK1/2, and VEGF, all of which have clinically available pharmacologic inhibitors, which could be further explored as therapeutic targets."
01/01/2022 - "Blocking VEGF with bevacizumab reduces the proangiogenic effect of affected fibroblasts in both the matrigel and direct coculture angiogenesis models, indicating that elevated VEGF secretion is a key mediator of increased angiogenesis in melorheostosis tissue. "

05/01/1978 - "In melorheostosis, focal increased radiopharmaceutical accumulation appeared in each radiographically abnormal area. "
06/01/1994 - "Radionuclide bone scans demonstrated a moderately increased uptake of radiopharmaceutical localized to the "flowing" cortical hyperostosis of melorheostosis observed radiographically. "

09/27/2018 - "Clinical improvement in a patient with monostotic melorheostosis after treatment with denosumab: a case report."
06/21/2013 - "We report an adult patient with a very painful melorheostosis, who  improved after treatment with zoledronate, either on symptoms or on bone scans. "
07/01/1986 - "We describe a patient with melorheostosis who showed improvement in pain and vasomotor function after treatment with nifedipine. "
09/27/2018 - "A 20-year-old Danish woman with melorheostosis in her right femoral shaft and disabling pain in the affected area, whose symptoms did not in the long term respond to zoledronic acid, experienced continuous remission of pain after treatment with denosumab. "

04/01/2014 - "This case highlights the role of nerve decompression in melorheostosis and the importance of early identification of the disease to prevent unnecessary bone biopsies. "
09/01/2022 - "Detection of the fibromatous component in multilevel extensive intradural lipomatosis associated with melorheostosis is helpful in planning selection of the level of surgical excision and decompression."
04/01/2020 - "We report a case of melorheostosis involving the T9 vertebra presenting with unilateral radicular pain managed surgically by decompression and posterior stabilization. "
08/01/2013 - "We report a case of lumbar melorheostosis causing disabling radiculopathy treated with nerve root decompression. "

01/01/2018 - "Debridement arthroplasty is safe and effective in treating elbow stiffness associated with Melorheostosis."
05/13/2015 - "Vascular Malformations Corresponding to Sclerotomes in Multifocal Melorheostosis: Painful Hip and Knee Contractures Treated with Total Joint Arthroplasty."
04/01/2015 - "Vascular Malformations Corresponding to Sclerotomes in Multifocal Melorheostosis: Painful Hip and Knee Contractures Treated with Total Joint Arthroplasty: A Case Report."

01/01/2018 - "Debridement arthroplasty is safe and effective in treating elbow stiffness associated with Melorheostosis."

01/01/2020 - "These data reveal antagonism of TGF-β/SMAD3 activation by BMP signaling in SMAD3 mutation-positive endosteal melorheostosis, which may guide future therapies."
01/01/2017 - "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."
01/31/2015 - "Melorheostosis is incurable, the therapy is mostly focused on maintaining patient quality of life. "
09/01/2021 - "Bisphosphonate Therapy in the Management of Symptomatic Melorheostosis of Tibia."