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Erschienen in: Pediatric Radiology 2/2019

15.11.2018 | Original Article

Ulnar focal cortical indentation: a progressive, deforming variant of focal fibrocartilaginous dysplasia

verfasst von: Alyssa R. Goldbach, Dan A. Zlotolow, Sarah D. Fenerty, Jordan Rapp, Grigory Gershkovich, Sayed Ali

Erschienen in: Pediatric Radiology | Ausgabe 2/2019

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Abstract

Background

Focal fibrocartilaginous dysplasia is a rare growth disturbance of bone resulting in deformity. In the ulna, focal fibrocartilaginous dysplasia is particularly rare, and the characteristic fibroligamentous tether can result in progressive deformity with progressive dislocation of the radial head. The fibroligamentous tether is similar in appearance and function to the Vickers ligament seen in Madelung deformity. The imaging features of ulnar focal fibrocartilaginous dysplasia include a unilateral angular deformity in the ulna with a radiolucent cortical defect and surrounding sclerosis, with secondary radial bowing or radial head dislocation. Focal fibrocartilaginous dysplasia of the ulna has been described using the term ulnar focal cortical indentation.

Objective

To review the clinical and radiologic features of four cases of ulnar focal cortical indentation occurring in pediatric patients.

Materials and methods

We retrospectively reviewed picture archiving and communication system and electronic medical record imaging findings with surgical correlation of ulnar focal cortical indentation at two large specialty pediatric hospitals.

Results

Ulnar focal cortical indentation lesions typically arise in the ulnar metaphysis and result in angular forearm deformities with progressive radial deformity including radial head dislocation. Early surgical intervention prevents progression of the deformity and retains range of motion.

Conclusion

Ulnar focal cortical indentation, although rare, is likely a progressive form of focal fibrocartilaginous dysplasia resulting in significant deformity and disability. Early recognition of the characteristic imaging features is important for early surgical intervention to preserve range of motion and prevent radial head dislocation.
Literatur
1.
Zurück zum Zitat Bell SN, Campbell PE, Cole WG, Menelaus MB (1985) Tibia vara caused by focal fibrocartilaginous dysplasia. Three case reports. J Bone Joint Surg 67:780–784CrossRef Bell SN, Campbell PE, Cole WG, Menelaus MB (1985) Tibia vara caused by focal fibrocartilaginous dysplasia. Three case reports. J Bone Joint Surg 67:780–784CrossRef
2.
Zurück zum Zitat Gottschalk HP, Light TR, Smith P (2012) Focal fibrocartilaginous dysplasia in the ulna: report on 3 cases. J Hand Surg 37:2300–2303CrossRef Gottschalk HP, Light TR, Smith P (2012) Focal fibrocartilaginous dysplasia in the ulna: report on 3 cases. J Hand Surg 37:2300–2303CrossRef
3.
Zurück zum Zitat Kazuki K, Hiroshima K, Kawahara K (2005) Ulnar focal cortical indentation: a previously unrecognized form of ulnar dysplasia. J Bone Joint Surg 87:540–543CrossRef Kazuki K, Hiroshima K, Kawahara K (2005) Ulnar focal cortical indentation: a previously unrecognized form of ulnar dysplasia. J Bone Joint Surg 87:540–543CrossRef
4.
Zurück zum Zitat Nakura A, Kawabata H, Tamura D, Sugita A (2017) Focal fibrocartilaginous dysplasia in the ulna with the radial head dislocation: a case report and literature review. J Pediatr Orthop B 26:41–47CrossRefPubMed Nakura A, Kawabata H, Tamura D, Sugita A (2017) Focal fibrocartilaginous dysplasia in the ulna with the radial head dislocation: a case report and literature review. J Pediatr Orthop B 26:41–47CrossRefPubMed
5.
6.
Zurück zum Zitat Lincoln TL, Birch JG (1997) Focal fibrocartilaginous dysplasia in the upper extremity. J Pediatr Orthop 17:528–532PubMed Lincoln TL, Birch JG (1997) Focal fibrocartilaginous dysplasia in the upper extremity. J Pediatr Orthop 17:528–532PubMed
7.
Zurück zum Zitat Choi IH, Kim CJ, Cho TJ et al (2000) Focal fibrocartilaginous dysplasia of long bones: report of eight additional cases and literature review. J Pediatr Orthop 20:421–427PubMed Choi IH, Kim CJ, Cho TJ et al (2000) Focal fibrocartilaginous dysplasia of long bones: report of eight additional cases and literature review. J Pediatr Orthop 20:421–427PubMed
8.
Zurück zum Zitat Eren A, Cakar M, Erol B et al (2008) Focal fibrocartilaginous dysplasia in the humerus. J Pediatr Orthop 17:148–151CrossRef Eren A, Cakar M, Erol B et al (2008) Focal fibrocartilaginous dysplasia in the humerus. J Pediatr Orthop 17:148–151CrossRef
9.
Zurück zum Zitat Ringe KI, Schirg E, Rosenthal H et al (2009) Unilateral tibia vara in a toddler caused by focal fibrocartilaginous dysplasia. J Radiol Case Rep 3:14–17PubMedPubMedCentral Ringe KI, Schirg E, Rosenthal H et al (2009) Unilateral tibia vara in a toddler caused by focal fibrocartilaginous dysplasia. J Radiol Case Rep 3:14–17PubMedPubMedCentral
10.
Zurück zum Zitat Pavone V, Testa G, Riccioli M et al (2014) The natural history of focal fibrocartilaginous dysplasia in the young child with tibia vara. Eur J Orthop Surg Traumatol 24:579–586CrossRefPubMed Pavone V, Testa G, Riccioli M et al (2014) The natural history of focal fibrocartilaginous dysplasia in the young child with tibia vara. Eur J Orthop Surg Traumatol 24:579–586CrossRefPubMed
11.
Zurück zum Zitat Smith NC, Carter PR, Ezaki M (2004) Focal fibrocartilaginous dysplasia in the upper limb: seven additional cases. J Pediatr Orthop 24:700–705CrossRefPubMed Smith NC, Carter PR, Ezaki M (2004) Focal fibrocartilaginous dysplasia in the upper limb: seven additional cases. J Pediatr Orthop 24:700–705CrossRefPubMed
12.
Zurück zum Zitat Nakase T, Yasui N, Araki N et al (1998) Florid periosteal reaction and focal fibrocartilaginous dysplasia. Skelet Radiol 27:646–649CrossRef Nakase T, Yasui N, Araki N et al (1998) Florid periosteal reaction and focal fibrocartilaginous dysplasia. Skelet Radiol 27:646–649CrossRef
13.
Zurück zum Zitat Ando A, Hatori M, Hosaka M et al (2008) A patient with focal fibrocartilaginous dysplasia in the distal femur and review of the literature. Tohoku J Exp Med 215:307–312CrossRefPubMed Ando A, Hatori M, Hosaka M et al (2008) A patient with focal fibrocartilaginous dysplasia in the distal femur and review of the literature. Tohoku J Exp Med 215:307–312CrossRefPubMed
14.
Zurück zum Zitat Jouve JL, Kohler R, Mubarak SJ et al (2007) Focal fibrocartilaginous dysplasia (‘fibrous periosteal inclusion’): an additional series of eleven cases and literature review. J Pediatr Orthop 27:75–84CrossRefPubMed Jouve JL, Kohler R, Mubarak SJ et al (2007) Focal fibrocartilaginous dysplasia (‘fibrous periosteal inclusion’): an additional series of eleven cases and literature review. J Pediatr Orthop 27:75–84CrossRefPubMed
15.
Zurück zum Zitat Ali S, Kaplan S, Kaufman T et al (2015) Madelung deformity and Madelung-type deformities: a review of the clinical and radiological characteristics. Pediatr Radiol 45:1856–1863CrossRefPubMed Ali S, Kaplan S, Kaufman T et al (2015) Madelung deformity and Madelung-type deformities: a review of the clinical and radiological characteristics. Pediatr Radiol 45:1856–1863CrossRefPubMed
16.
Zurück zum Zitat Welborn MC, Stevens P (2017) Correction of angular deformities due to focal fibrocartilaginous dysplasia using guided growth: a preliminary report. J Pediatr Orthop 37:e183–e187CrossRefPubMed Welborn MC, Stevens P (2017) Correction of angular deformities due to focal fibrocartilaginous dysplasia using guided growth: a preliminary report. J Pediatr Orthop 37:e183–e187CrossRefPubMed
17.
Zurück zum Zitat Navarro OM, Caro-Domínguez P (2017) Bone tumors of the pediatric foot: imaging appearances. Pediatr Radiol 47:739–749CrossRefPubMed Navarro OM, Caro-Domínguez P (2017) Bone tumors of the pediatric foot: imaging appearances. Pediatr Radiol 47:739–749CrossRefPubMed
Metadaten
Titel
Ulnar focal cortical indentation: a progressive, deforming variant of focal fibrocartilaginous dysplasia
verfasst von
Alyssa R. Goldbach
Dan A. Zlotolow
Sarah D. Fenerty
Jordan Rapp
Grigory Gershkovich
Sayed Ali
Publikationsdatum
15.11.2018
Verlag
Springer Berlin Heidelberg
Erschienen in
Pediatric Radiology / Ausgabe 2/2019
Print ISSN: 0301-0449
Elektronische ISSN: 1432-1998
DOI
https://doi.org/10.1007/s00247-018-4294-6

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