<span name="style_bold">Osteopoikilosis</span>: <span name="style_bold">Report of a case and review of the current literature</span>

None; None; None

Abstracts

A 19-year-old-male from Guápiles with history of congenital torticollis and obstructive urolithiasis was seen in the orthopedic clinic because of symetrical hyperostic spots in both hips and sacroilliac joints detected incidentally when an intravenous pyelogram was performed on the patient. The patient manifested pain in both lower limbs of several months’ length. Hip and knee radiographs showed symmetrical sclerotic bone lesions suggestive of osteopoikilosis. A bone gammagraphy study showed no pathological findings. The possibility of any other associated medical condition was ruled out. Osteopoikilosis requires no medical treatment.

osteopoikilosis; displasia; osteosclerosis

Paciente masculino de diecinueve años de edad, proveniente de Guápiles, con antecedente de tortículis congénita y urolitiasis obstructiva a repetición, con un cuadro clínico de varios meses de evolución de dolor en miembros inferiores, fue referido por lesiones enostóticas simétricas en ambas caderas y articulaciones sacroiliacas, como hallazgos incidentales de un estudio de pielograma intravenoso. Radiografías de cadera y rodilla mostraron lesiones escleróticas simétricas, características de osteopoiquilosis. El estudio de gamagrafía óseo descarta hallazgos óseos patológicos. Se descarta también cualquier otra condición asociada que pueda requerir tratamiento médico. Además, su condición ósea no requiere ningún tipo de intervención.

osteopoiquilosis; displasia; osteoesclerosis

A 19-year-old-male from Guápiles with history of congenital torticollis and obstructive urolithiasis was seen in the orthopedic clinic because of symetrical hyperostic spots in both hips and sacroilliac joints detected incidentally when an intravenous pyelogram was performed on the patient. The patient manifested pain in both lower limbs of several months’ length. Hip and knee radiographs showed symmetrical sclerotic bone lesions suggestive of osteopoikilosis. A bone gammagraphy study showed no pathological findings. The possibility of any other associated medical condition was ruled out. Osteopoikilosis requires no medical treatment.

osteopoikilosis; displasia; osteosclerosis

It is important to emphasize that in this case, the patient denies family history of osteopoikilosis. A history of hereditary bone disease in his family is not known either. This is expected; since it is mostly a subclinical disorder, if a radiological study in family members does not exist, its identification in other subjects is difficult.

Osteopoikilosis

José Carlos Alonso-Escalante¹, Esteban Zamora-Estrada², Faylan Esquivel-Solís³.

Orthopedics and Radiology Department, “Rafael Angel Calderón Guardia” Hospital. Medicine School, University of Costa Rica.
Contact information: jcalonso@racsa.co.cr

Abstract

Keywords: osteopoikilosis, displasia, osteosclerosis

¹This entity is acortical bone ectopiainspongy bone. It is characterized by the presence of hyperostosis foci at any point in the skeleton, predominantly in the metaphysis and epiphysis of long bones, as well as in the carpal and tarsal bones.²

³

^2,4According to the literature reviewed, this is the first osteopoikilosis case described in Costa Rica, thus, it is a significant contribution to the study of this disorder.

5 mm and juxta-articularin knees, hips and sacroiliac joints (Figures 1 and 2). Furthermore, by means of a neck radiograph, the possible presence of a soft tissue mass is documented. Afterwards, a neck ultrasound is performed, which rules out pathologies. A whole body bone scan shows normal skeletal findings.

⁵

^1-3 As in this case, the detection of osteopoikilosis tends to be accidental, when a radiologic study is requested for some other reason and it evidences the characteristic bone lesions.⁵

⁶

⁷

^2,3

Conflicts of interest: Authors report no conflict of interest.

Figure 2

Figure 2

Figure 1

References

1. Greenspam A. Sclerosing bone displasias a target-site approach. Skeletal Radiol 1991; 20: 561-583.
2. Baasanjav S, Jamsheer A, Kolanczyk M, Horn D, Latos T, Hoffman K et al. Osteopoikilosis and multiple exostoses caused by novel mutations in LEMD3 and EXT1 genes respectively - coincidence within one family BMC BMC Med Genet 2010; 11:110-117.
3. Ruiz-Fernández M, León-García F, Ruiz-González A, Rodríguez- Palmero M. Osteopatía condensante diseminada: definición y actitud. BSCP Can Ped 2003; 27: 401-405.
4. Sutherland C. Osteopoikilosis. Radiology 1935; 25: 470-479.
5. Sclerotic Lesions of Bone. Department of Radiology University of Washington, 2008. En http://www.rad.washington.edu/academics/academic-sections/msk/teaching-materials/online-musculoskeletal-radiology-book/sclerotic-lesions-of-bone.

» http://www.rad.washington.edu/academics/academic-sections/msk/teaching -materials/online- musculoskeletal-radiology-book/sclerotic-lesions-of-bone
6. Khot R, Sikarwar J, Gupta R, Sharma G. Osteopoikilosis: A case report. Ind J Radiol Imag 2005; 15: 453-454.
7. Serdaroglu M, Capkin E, Uçuencue F, Tosun M. Case report of a patient with osteopoikilosis. Rheumatol Int 2007; 27: 683-686.

en_bart10

Received: September 2, 2011 Accepted: February 9, 2012

References

1. Greenspam A. Sclerosing bone displasias a target-site approach. Skeletal Radiol 1991; 20: 561-583.
2. Baasanjav S, Jamsheer A, Kolanczyk M, Horn D, Latos T, Hoffman K et al. Osteopoikilosis and multiple exostoses caused by novel mutations in LEMD3 and EXT1 genes respectively - coincidence within one family BMC BMC Med Genet 2010; 11:110-117.
3. Ruiz-Fernández M, León-García F, Ruiz-González A, Rodríguez- Palmero M. Osteopatía condensante diseminada: definición y actitud. BSCP Can Ped 2003; 27: 401-405.
4. Sutherland C. Osteopoikilosis. Radiology 1935; 25: 470-479.
5. Sclerotic Lesions of Bone. Department of Radiology University of Washington, 2008. En http://www.rad.washington.edu/academics/academic-sections/msk/teaching-materials/online-musculoskeletal-radiology-book/sclerotic-lesions-of-bone.

» http://www.rad.washington.edu/academics/academic-sections/msk/teaching -materials/online- musculoskeletal-radiology-book/sclerotic-lesions-of-bone
6. Khot R, Sikarwar J, Gupta R, Sharma G. Osteopoikilosis: A case report. Ind J Radiol Imag 2005; 15: 453-454.
7. Serdaroglu M, Capkin E, Uçuencue F, Tosun M. Case report of a patient with osteopoikilosis. Rheumatol Int 2007; 27: 683-686.

en_bart10

Received: September 2, 2011 Accepted: February 9, 2012

Publication Dates

Publication in this collection
18 Sept 2012
Date of issue
June 2012

History

Received
02 Sept 2011
Accepted
09 Feb 2012

[1] 1. Greenspam A. Sclerosing bone displasias a target-site approach. Skeletal Radiol 1991; 20: 561-583.

[2] 2. Baasanjav S, Jamsheer A, Kolanczyk M, Horn D, Latos T, Hoffman K et al. Osteopoikilosis and multiple exostoses caused by novel mutations in LEMD3 and EXT1 genes respectively - coincidence within one family BMC BMC Med Genet 2010; 11:110-117.

[3] 3. Ruiz-Fernández M, León-García F, Ruiz-González A, Rodríguez- Palmero M. Osteopatía condensante diseminada: definición y actitud. BSCP Can Ped 2003; 27: 401-405.

[4] 4. Sutherland C. Osteopoikilosis. Radiology 1935; 25: 470-479.

[5] 5. Sclerotic Lesions of Bone. Department of Radiology University of Washington, 2008. En http://www.rad.washington.edu/academics/academic-sections/msk/teaching-materials/online-musculoskeletal-radiology-book/sclerotic-lesions-of-bone.

» http://www.rad.washington.edu/academics/academic-sections/msk/teaching -materials/online- musculoskeletal-radiology-book/sclerotic-lesions-of-bone

[6] 6. Khot R, Sikarwar J, Gupta R, Sharma G. Osteopoikilosis: A case report. Ind J Radiol Imag 2005; 15: 453-454.

[7] 7. Serdaroglu M, Capkin E, Uçuencue F, Tosun M. Case report of a patient with osteopoikilosis. Rheumatol Int 2007; 27: 683-686.

<b>Osteopoikilosis</b>: <b>Report of a case and review of the current literature</b>

Abstracts

References

References

Publication Dates

History