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

Abstract Background Intramedullary osteosclerosis (IMOS) is a rare condition without specific radiological findings except for the osteosclerotic lesion and is not associated with family history and infection, trauma, or systemic illness. Although the diagnosis of IMOS is confirmed after excluding o...

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Main Authors: Kensaku Abe, Norio Yamamoto, Katsuhiro Hayashi, Akihiko Takeuchi, Shinji Miwa, Kentaro Igarashi, Takashi Higuchi, Yuta Taniguchi, Hirotaka Yonezawa, Yoshihiro Araki, Sei Morinaga, Yohei Asano, Hiroyuki Tsuchiya
Format: Article
Language:English
Published: BMC 2020-11-01
Series:BMC Musculoskeletal Disorders
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Online Access:http://link.springer.com/article/10.1186/s12891-020-03758-5
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author Kensaku Abe
Norio Yamamoto
Katsuhiro Hayashi
Akihiko Takeuchi
Shinji Miwa
Kentaro Igarashi
Takashi Higuchi
Yuta Taniguchi
Hirotaka Yonezawa
Yoshihiro Araki
Sei Morinaga
Yohei Asano
Hiroyuki Tsuchiya
author_facet Kensaku Abe
Norio Yamamoto
Katsuhiro Hayashi
Akihiko Takeuchi
Shinji Miwa
Kentaro Igarashi
Takashi Higuchi
Yuta Taniguchi
Hirotaka Yonezawa
Yoshihiro Araki
Sei Morinaga
Yohei Asano
Hiroyuki Tsuchiya
author_sort Kensaku Abe
collection DOAJ
description Abstract Background Intramedullary osteosclerosis (IMOS) is a rare condition without specific radiological findings except for the osteosclerotic lesion and is not associated with family history and infection, trauma, or systemic illness. Although the diagnosis of IMOS is confirmed after excluding other osteosclerotic lesions, IMOS is not well known because of its rarity and no specific feature. Therefore, these situations might result in delayed diagnosis. Hence, this case report aimed to investigate three cases of IMOS and discuss imaging findings and clinical outcomes. Case presentation All three cases were examined between 2015 and 2019. The location of osteosclerotic lesions were femoral diaphyses in the 60-year-old man (Case 1) and 41-year-old woman (Case 2) and tibial diaphysis in the 44-year-old woman (Case 3). All cases complained of severe pain and showed massive diaphyseal osteosclerotic lesions in plain radiograms and computed tomography (CT) scans. Cases 2 and 3 were examined using the triphasic bone scan, and a fusiform-shaped intense area of the tracer uptake on delayed bone image was detected in both cases without (Case 2) or slightly increased vascularity (Case 3) on the blood pool image, which was reported as a specific finding of IMOS. Open biopsy was performed in all cases, and histologic section showed trabecular bone sclerosis with hypocellular fibrous tissues, finally diagnosed as IMOS. The pain was sharply improved after biopsy and kept at the latest follow-up periods (34, 33, and 6 months in Cases 1, 2, and 3, respectively). Conclusions Massive sclerotic lesions with severe pain in the diaphyseal region of long bones should be considered as IMOS to avoid the delayed diagnosis, although other sclerotic bony lesions should be carefully excluded. Triphasic bone scan with a fusiform-shaped intense area of tracer uptake on delayed bone image and without or slightly increased vascularity on the blood pool image will help confirm IMOS. The role of open biopsy was to confirm the diagnosis of IMOS and to give the severe pain relief immediately in the three cases, although more cases and long-term follow-up are necessary.
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spelling doaj.art-d360b30d774a409da4d58dbf1b2405f72022-12-21T18:18:30ZengBMCBMC Musculoskeletal Disorders1471-24742020-11-012111810.1186/s12891-020-03758-5Diagnosis and treatment of intramedullary osteosclerosis: a report of three cases and literature reviewKensaku Abe0Norio Yamamoto1Katsuhiro Hayashi2Akihiko Takeuchi3Shinji Miwa4Kentaro Igarashi5Takashi Higuchi6Yuta Taniguchi7Hirotaka Yonezawa8Yoshihiro Araki9Sei Morinaga10Yohei Asano11Hiroyuki Tsuchiya12Department of Orthopaedic Surgery, Graduate School of Medical Sciences, Kanazawa UniversityDepartment of Orthopaedic Surgery, Graduate School of Medical Sciences, Kanazawa UniversityDepartment of Orthopaedic Surgery, Graduate School of Medical Sciences, Kanazawa UniversityDepartment of Orthopaedic Surgery, Graduate School of Medical Sciences, Kanazawa UniversityDepartment of Orthopaedic Surgery, Graduate School of Medical Sciences, Kanazawa UniversityDepartment of Orthopaedic Surgery, Graduate School of Medical Sciences, Kanazawa UniversityDepartment of Orthopaedic Surgery, Graduate School of Medical Sciences, Kanazawa UniversityDepartment of Orthopaedic Surgery, Graduate School of Medical Sciences, Kanazawa UniversityDepartment of Orthopaedic Surgery, Graduate School of Medical Sciences, Kanazawa UniversityDepartment of Orthopaedic Surgery, Graduate School of Medical Sciences, Kanazawa UniversityDepartment of Orthopaedic Surgery, Graduate School of Medical Sciences, Kanazawa UniversityDepartment of Orthopaedic Surgery, Graduate School of Medical Sciences, Kanazawa UniversityDepartment of Orthopaedic Surgery, Graduate School of Medical Sciences, Kanazawa UniversityAbstract Background Intramedullary osteosclerosis (IMOS) is a rare condition without specific radiological findings except for the osteosclerotic lesion and is not associated with family history and infection, trauma, or systemic illness. Although the diagnosis of IMOS is confirmed after excluding other osteosclerotic lesions, IMOS is not well known because of its rarity and no specific feature. Therefore, these situations might result in delayed diagnosis. Hence, this case report aimed to investigate three cases of IMOS and discuss imaging findings and clinical outcomes. Case presentation All three cases were examined between 2015 and 2019. The location of osteosclerotic lesions were femoral diaphyses in the 60-year-old man (Case 1) and 41-year-old woman (Case 2) and tibial diaphysis in the 44-year-old woman (Case 3). All cases complained of severe pain and showed massive diaphyseal osteosclerotic lesions in plain radiograms and computed tomography (CT) scans. Cases 2 and 3 were examined using the triphasic bone scan, and a fusiform-shaped intense area of the tracer uptake on delayed bone image was detected in both cases without (Case 2) or slightly increased vascularity (Case 3) on the blood pool image, which was reported as a specific finding of IMOS. Open biopsy was performed in all cases, and histologic section showed trabecular bone sclerosis with hypocellular fibrous tissues, finally diagnosed as IMOS. The pain was sharply improved after biopsy and kept at the latest follow-up periods (34, 33, and 6 months in Cases 1, 2, and 3, respectively). Conclusions Massive sclerotic lesions with severe pain in the diaphyseal region of long bones should be considered as IMOS to avoid the delayed diagnosis, although other sclerotic bony lesions should be carefully excluded. Triphasic bone scan with a fusiform-shaped intense area of tracer uptake on delayed bone image and without or slightly increased vascularity on the blood pool image will help confirm IMOS. The role of open biopsy was to confirm the diagnosis of IMOS and to give the severe pain relief immediately in the three cases, although more cases and long-term follow-up are necessary.http://link.springer.com/article/10.1186/s12891-020-03758-5Intramedullary osteosclerosis99mTc- methylene diphosphonate (MDP) triphasic bone scanOpen biopsy
spellingShingle Kensaku Abe
Norio Yamamoto
Katsuhiro Hayashi
Akihiko Takeuchi
Shinji Miwa
Kentaro Igarashi
Takashi Higuchi
Yuta Taniguchi
Hirotaka Yonezawa
Yoshihiro Araki
Sei Morinaga
Yohei Asano
Hiroyuki Tsuchiya
Diagnosis and treatment of intramedullary osteosclerosis: a report of three cases and literature review
BMC Musculoskeletal Disorders
Intramedullary osteosclerosis
99mTc- methylene diphosphonate (MDP) triphasic bone scan
Open biopsy
title Diagnosis and treatment of intramedullary osteosclerosis: a report of three cases and literature review
title_full Diagnosis and treatment of intramedullary osteosclerosis: a report of three cases and literature review
title_fullStr Diagnosis and treatment of intramedullary osteosclerosis: a report of three cases and literature review
title_full_unstemmed Diagnosis and treatment of intramedullary osteosclerosis: a report of three cases and literature review
title_short Diagnosis and treatment of intramedullary osteosclerosis: a report of three cases and literature review
title_sort diagnosis and treatment of intramedullary osteosclerosis a report of three cases and literature review
topic Intramedullary osteosclerosis
99mTc- methylene diphosphonate (MDP) triphasic bone scan
Open biopsy
url http://link.springer.com/article/10.1186/s12891-020-03758-5
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