Abstract
Osteopoikilosis, a rare benign bone disease, is usually an incidental radiographic finding without therapeutic considerations. It is characterized by multiple sclerotic bone lesions, which are asymptomatic in most of cases. The challenge is to distinguish osteopoikilosis from other, especially malignant, bone diseases. In this case report, we present a 65-year-old male patient, who was incidentally diagnosed with osteopoikilosis in our department. The diagnosis was based on the exclusion of differential diagnosis, typical imaging findings in X-rays, CT scan and PET/CT scan. In this work, we discuss recent literature on the disease, including prevalence, pathogenesis, clinical manifestation, as well as differential diagnosis.
This is a preview of subscription content, log in via an institution to check access.
Similar content being viewed by others
References
Fisk GR. Osteopoikilosis (familial disseminated osteosclerosis). Proc R Soc Med. 1953;46(2):103–5.
Benli IT, Akalin S, Boysan E, Mumcu EF, Kiş M, Türkoğlu D. Epidemiological, clinical and radiological aspects of osteopoikilosis. J Bone Joint Surg Br. 1992;74(4):504–6.
Nielsen GP, Rosenberg AE. Diagnostic pathology: bone: Elsevier; 2017.
Al-Bourini O, Lotz J, Wienbeck S. Radiologische Bildgebung bei Osteopoikilose. RöFo - Fortschritte auf dem Gebiet der Röntgenstrahlen und der bildgebenden Verfahren. 2018;190(6):499–502.
Boulet C, et al. Sclerosing bone dysplasias: genetic, clinical and radiology update of hereditary and non-hereditary disorders. Br J Radiol. 2016;89(1062):20150349.
Serdaroğlu M, Capkin E, Uçüncü F, Tosun M. Case report of a patient with osteopoikilosis. Rheumatol Int. 2007;27(7):683–6.
Hellemans J, Preobrazhenska O, Willaert A, Debeer P, Verdonk PCM, Costa T, et al. Loss-of-function mutations in LEMD3 result in osteopoikilosis, Buschke-Ollendorff syndrome and melorheostosis. Nat Genet. 2004;36(11):1213–8.
Woyciechowsky TG, Monticielo MR, Keiserman B, Monticielo OA. Osteopoikilosis: what does the rheumatologist must know about it? Clin Rheumatol. 2012;31(4):745–8.
Korkmaz MF, Elli M, Özkan MB, Bilgici MC, Dağdemir A, Korkmaz M, et al. Osteopoikilosis: report of a familial case and review of the literature. Rheumatol Int. 2015;35(5):921–4.
Rusu A, Cardoneanu A, Burlui A, Rezus E. Osteopoikilosis - a case report. Arch Clin Cases. 2015;2(2):81–5.
Joseph RO, Thomas J, Shenoy P. Osteopoikilosis and ankylosing spondylitis: strange bedfellows: a case report. Int J Rheum Dis. 2019;22(6):1756–185X.13594.
Lüke U, von Ditfurth B, Dumont C. Osteopoikilosis. Dtsch Aerzteblatt. 2019.
Liu S, Zhou X, Song A, Huo Z, Liu Y, Wang Y. Osteopoikilosis. Postgrad Med J. 2019. postgradmedj-2019-136705.
Pal R, Dutta P, Bhadada SK, Bolla T, Bhansali A. Islands of bone within bone. Postgrad Med J. 2019;95(1123):289.
Ureten K. Osteopoikilosis in a patient with rheumatoid arthritis complicated with dry eyes. Rheumatol Int. 2007;27(11):1079–82.
Crepaldi G, Saracco M, Rovera G, Scarati M, Lomater C. Osteopoikilosis hiding active sacroiliitis in psoriatic arthritis. Rheumatology. 2019.
Correa Llano MG, Mora J, Torner F, Inarejos E, Cruz O. Osteopoikilosis with germline LEMD3 mutation mimicking bone metastases in a girl with a concurrent secreting mixed germ cell tumor. J Pediatr Hematol Oncol. 2019:1.
Madeira N, Ganhão S, Ferreira RM, Duarte AC, Freitas R, Miranda LC. Osteopoikilosis: case series from Portuguese Rheumatology Centers. Acta Reumatol Port. 44(1):78–83.
Gnoli M, Staals EL, Campanacci L, Bedeschi MF, Faletra F, Gallone S, et al. Melorheostosis and osteopoikilosis clinical and molecular description of an Italian case series. Calcif Tissue Int. 2019;105(2):215–21.
Author information
Authors and Affiliations
Corresponding author
Ethics declarations
Conflict of Interest
The authors declare that they have no conflict of interest.
Ethics Declarations
All procedures performed in studies involving human participants were in accordance with 1964 Helsinki declaration and its later amendments or comparable ethical standards. Informed consent was obtained from all individual participants included in the study.
Additional information
Publisher’s Note
Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.
This article is part of the Topical Collection on Medicine
Rights and permissions
About this article
Cite this article
Voulgari, M.L., Kellner, H. A 65-year-old Patient with Osteopoikilosis and Bilateral Pleural Effusion. Case Report. SN Compr. Clin. Med. 2, 809–813 (2020). https://doi.org/10.1007/s42399-020-00297-x
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s42399-020-00297-x