Diagnosis and treatment of intramedullary osteosclerosis: a report of three cases and literature review

Abe, Kensaku; Yamamoto, Norio; Hayashi, Katsuhiro; Takeuchi, Akihiko; Miwa, Shinji; Igarashi, Kentaro; Higuchi, Takashi; Taniguchi, Yuta; Yonezawa, Hirotaka; Araki, Yoshihiro; Morinaga, Sei; Asano, Yohei; Tsuchiya, Hiroyuki

doi:10.1186/s12891-020-03758-5

Cited by 6 publications

(18 citation statements)

References 16 publications

(24 reference statements)

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“…None of these patients have a history of trauma or infection. No abnormal laboratory results including inflammatory reaction were observed [2][3][4], such in our case.…”

Section: Discussionmentioning

confidence: 57%

“…Computed Tomography (CT) scans showed, in these series intramedullary sclerosis and soft-tissue swelling adjacent to the cortex. The cortex was intact but thickened [2][3][4]8]. In the case of Balkissoon et al, CT scan proofs were not available such as in our case [5].…”

Section: Discussionmentioning

confidence: 80%

“…In the series of Kullanuch Chanchairujira et al, nine female patients with leg pain and imaging signs of IMOS were detected during a 25-years period [3]. Kensaku Abe et al reported three cases (one man, two women) of osteosclerotic lesions (femoral diaphyses, tibial diaphysis) examined between 2015 and 2019 [2]. Vasilios Skiadas et al presented the case of a 60-year-old woman with a bilateral midfemoral pain for 8 years [4].…”

Section: Discussionmentioning

confidence: 99%

“…The term intramedullary osteosclerosis was introduced by Abdul-Karim et al in 1988 to describe a rare pathology associated with new bone formation localized mainly in the diaphysis of the tibia in adult female patients [1]. It is a rare disease, without specific radiological signs except for the osteosclerotic lesion and it is not associated with a family history, infection, trauma or systemic disease [2]. Although the diagnosis of IMOS is confirmed after excluding other sclerotic lesions, it is not well recognized due to its rarity and absence of specific signs.…”

Section: Introductionmentioning

confidence: 99%

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A rare case of intramedullary osteosclerosis and literature review

mabrouk¹

2022

J Clin Images Med Case Rep

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Intramedullary Osteosclerosis (IMOS) is a rare idiopathic dysplasia of the long bones shafts. IMOS remain an un known diagnosis because of its rarity and the absence of specific features. Indeed, the diagnosis of IMOS can be confirmed only after excluding other differential diagnosis of osteosclerotic lesions. We report here a case of IMOS of the tibia shafts, diagnosed in a 53-year-old man. Medical history did not reveal a history of trauma or infection or familial bone disease and all other causes of osteosclerosis were excluded. The X-Rays showed extensive sclerotic intramedullary lesion of the tibia shafts, cortical bone thickening, and narrowing of the medullary cavity. No periosteal reaction or soft-tissue abnormality or fracture line were noted. The radiological signs were not pathognomonic, but the clinic, biology, histology, as well as the absence of medical or family history strongly suggested the diagnostic.

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“…None of these patients have a history of trauma or infection. No abnormal laboratory results including inflammatory reaction were observed [2][3][4], such in our case.…”

Section: Discussionmentioning

confidence: 57%

Section: Discussionmentioning

confidence: 80%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

See 2 more Smart Citations

A rare case of intramedullary osteosclerosis and literature review

mabrouk¹

2022

J Clin Images Med Case Rep

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“…The treatment strategy in IMOS has not yet been established. Conflicting data emerge from the use of non-steroidal anti-inflammatory drugs (NSAID) and greater relief is usually provided by surgery [ 35 ]. Biochemical analysis shows no alteration in bone metabolism.…”

Section: Intramedullary Osteosclerosismentioning

confidence: 99%

Osteoblast Dysfunction in Non-Hereditary Sclerosing Bone Diseases

Giardullo

Altomare

Rotondo

et al. 2021

IJMS

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A review of the available literature was performed in order to summarize the existing evidence between osteoblast dysfunction and clinical features in non-hereditary sclerosing bone diseases. It has been known that proliferation and migration of osteoblasts are concerted by soluble factors such as fibroblast growth factor (FGF), platelet-derived growth factor (PDGF), transforming growth factor (TGF), bone morphogenetic protein (BMP) but also by signal transduction cascades such as Wnt signaling pathway. Protein kinases play also a leading role in triggering the activation of osteoblasts in this group of diseases. Post-zygotic changes in mitogen-activated protein kinase (MAPK) have been shown to be associated with sporadic cases of Melorheostosis. Serum levels of FGF and PDGF have been shown to be increased in myelofibrosis, although studies focusing on Sphingosine-1-phosphate receptor was shown to be strongly expressed in Paget disease of the bone, which may partially explain the osteoblastic hyperactivity during this condition. Pathophysiological mechanisms of osteoblasts in osteoblastic metastases have been studied much more thoroughly than in rare sclerosing syndromes: striking cellular mechanisms such as osteomimicry or complex intercellular signaling alterations have been described. Further research is needed to describe pathological mechanisms by which rare sclerosing non hereditary diseases lead to osteoblast dysfunction.

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Evolutive radiological findings in an exceptional case of Cryptococcal and Coccidioidal co-infection in an immunocompetent male. A case report.

Montanara,

Albergo,

Castellana

et al. 2024

Preprint

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Background: Bone infections such as chronic fungal erosive osteomyelitis are rare. uncommon forms of bone infection. The widespread yeast of the Cryptococcus species, the endemic dimorphic fungus Coccidioides, and the simultaneous infection by multiple pathogen species are even less common, especially in immunocompetent hosts. These infections frequently have no symptoms and the clinical signs remain undetected, allowing the infection to worsen over weeks or months. Mycotic arthritis is one of the rarest clinical symptoms; it is hard to distinguish from other types of arthritis, which slows down the diagnosis procedure. Case presentation: In order to demonstrate the beginning and progression of radiological abnormalities in a case of aggressive fungal osteomyelitis, we provide the case of a 31-year-old male patient here. The man showed signs of extensive bone erosion and inflammatory involvement in his right knee and right hallux phalanx, although he had no prior history of immunodeficiency. The co-infection resulting from Cryptococcus Neoformans in his right knee and Coccidioides Immitis in his hallux were the reason for the injuries. Conclusions: While an acute, benign, and self eradicating lung infection is the predominant presentation for most cases of cryptococcosis and coccidioidomycosis, a small percentage of patients experience a devastating extrapulmonary condition, which can include arthritis. The pathogenic mechanism of bone involvement are unknown, and it often remains untreated. Here, we discuss radiographic evidence of particular bone inflammation during the early phase and later phases of the disease, since management of this chronic condition remains a challenge. We propose that imaging may mimic osseous neoplasia in persistent fungal diseases, such as Coccidioidomycosis and Cryptococcosis.

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Diagnosis and treatment of intramedullary osteosclerosis: a report of three cases and literature review

Cited by 6 publications

References 16 publications

A rare case of intramedullary osteosclerosis and literature review

A rare case of intramedullary osteosclerosis and literature review

Osteoblast Dysfunction in Non-Hereditary Sclerosing Bone Diseases

Evolutive radiological findings in an exceptional case of Cryptococcal and Coccidioidal co-infection in an immunocompetent male. A case report.

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