sternoclavicular hyperostosis with pathological fracture of the clavicle—a case report

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Injury, Int. J. Care Injured 34 (2003) 464–466 Case report Sternoclavicular hyperostosis with pathological fracture of the clavicle—a case report S. Raja, A. Goel, A. Paul University Department of Orthopaedic Surgery, Manchester Royal Infirmary, Oxford Road, Manchester M13 9WL, UK Accepted 1 August 2002 1. Introduction Sternoclavicular hyperostosis is a well recognised but un- common condition. It is a disease of unknown etiology, which is characterised by periosteal reaction and endosteal hyperossification of the sternum, clavicles, and upper ribs as well as ossification of the surrounding soft tissues [4]. The presentation is either isolated or part of sternoclavicu- lar hyperostosis or synovitis, acne, pustulosis, hyperostosis, osteitis syndrome (SAPHO). Pathological fracture of clavicle in sternoclavicular hyper- ostosis has to our knowledge been described in post-mortem specimen [2] and not as a case report. We are reporting this rare case of sternoclavicular hyperostosis complicated by pathological fracture of clavicle. 2. Case report A 21 years old lady presented with acute pain in her left clavicle after she had lifted a heavy object. She gave a his- tory of intermittent discomfort in the region of left clavicle present for 12 months prior to this event. She was well with Fig. 1. Plain radiograph taken at time of presentation showing sclerosis with periosteal reaction in medial half of left clavicle. Corresponding author. Tel.: +44-161-276-1234; fax: +44-161-276-8006. no constitutional symptoms. She had no features of SAPHO syndrome. Clinical examination confirmed a tender swelling along the medial half of the left clavicle with loss of terminal 15 of abduction and forward flexion of the left shoulder. No prominent veins or signs of local inflammation were noted. There was no lymphadenopathy. Blood tests showed a normal full blood count, differential, erythrocyte sedimentation rate, C reactive protein and was negative for rheumatoid factor. Plain radiographs showed sclerosis of the medial half of the clavicle with periosteal reaction (Fig. 1). The contralat- eral clavicle appeared normal. A radioisotope scan showed increased uptake at the medial end of the left clavicle (Fig. 2). A computed tomography (CT scan) of the clavicle showed a hyperostotic medial half with a transverse fracture in the mid clavicle (Fig. 3). An open biopsy of the clavicle was performed and bone tissue obtained from two sites, the medial end and shaft showed hyperostosis with no evidence of infection or neo- plasia. Aerobic, anaerobic and mycobacterial cultures were neg- ative. She was treated with a broad arm sling and nons- teroidal anti-inflammatory drugs. After 4 weeks the acute 0020-1383/02/$ – see front matter © 2002 Elsevier Science Ltd. All rights reserved. PII:S0020-1383(02)00259-0

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Page 1: Sternoclavicular hyperostosis with pathological fracture of the clavicle—a case report

Injury, Int. J. Care Injured 34 (2003) 464–466

Case report

Sternoclavicular hyperostosis with pathological fractureof the clavicle—a case report

S. Raja, A. Goel, A. Paul∗University Department of Orthopaedic Surgery, Manchester Royal Infirmary, Oxford Road, Manchester M13 9WL, UK

Accepted 1 August 2002

1. Introduction

Sternoclavicular hyperostosis is a well recognised but un-common condition. It is a disease of unknown etiology,which is characterised by periosteal reaction and endostealhyperossification of the sternum, clavicles, and upper ribsas well as ossification of the surrounding soft tissues[4].The presentation is either isolated or part of sternoclavicu-lar hyperostosis or synovitis, acne, pustulosis, hyperostosis,osteitis syndrome (SAPHO).

Pathological fracture of clavicle in sternoclavicular hyper-ostosis has to our knowledge been described in post-mortemspecimen[2] and not as a case report. We are reporting thisrare case of sternoclavicular hyperostosis complicated bypathological fracture of clavicle.

2. Case report

A 21 years old lady presented with acute pain in her leftclavicle after she had lifted a heavy object. She gave a his-tory of intermittent discomfort in the region of left claviclepresent for 12 months prior to this event. She was well with

Fig. 1. Plain radiograph taken at time of presentation showing sclerosis with periosteal reaction in medial half of left clavicle.

∗ Corresponding author. Tel.:+44-161-276-1234;fax: +44-161-276-8006.

no constitutional symptoms. She had no features of SAPHOsyndrome. Clinical examination confirmed a tender swellingalong the medial half of the left clavicle with loss of terminal15◦ of abduction and forward flexion of the left shoulder. Noprominent veins or signs of local inflammation were noted.There was no lymphadenopathy.

Blood tests showed a normal full blood count, differential,erythrocyte sedimentation rate, C reactive protein and wasnegative for rheumatoid factor.

Plain radiographs showed sclerosis of the medial half ofthe clavicle with periosteal reaction (Fig. 1). The contralat-eral clavicle appeared normal. A radioisotope scan showedincreased uptake at the medial end of the left clavicle(Fig. 2). A computed tomography (CT scan) of the clavicleshowed a hyperostotic medial half with a transverse fracturein the mid clavicle (Fig. 3).

An open biopsy of the clavicle was performed and bonetissue obtained from two sites, the medial end and shaftshowed hyperostosis with no evidence of infection or neo-plasia.

Aerobic, anaerobic and mycobacterial cultures were neg-ative. She was treated with a broad arm sling and nons-teroidal anti-inflammatory drugs. After 4 weeks the acute

0020-1383/02/$ – see front matter © 2002 Elsevier Science Ltd. All rights reserved.PII: S0020-1383(02)00259-0

Page 2: Sternoclavicular hyperostosis with pathological fracture of the clavicle—a case report

S. Raja et al. / Injury, Int. J. Care Injured 34 (2003) 464–466 465

Fig. 2. Isotope bone scan taken at time of presentation showing increased uptake in medial half of left clavicle.

Fig. 3. CT scan taken at time of presentation showing hyperostotic medial half of left clavicle with a transverse fracture.

pain settled. A magnetic resonance imaging (MRI) obtainedat 1 year follow up showed a healed fracture (Fig. 4).

3. Discussion

Sternoclavicular hyperostosis was first described in theJapanese literature[4]. The main presenting symptoms arepain and swelling. The usual age of presentation is between30 and 50 years. Sonozaki et al.[5] reported a male pre-ponderance while Sartoris et al.[3] and Chigira et al.[1]reported a slight female preponderance. The differentialdiagnosis includes infection and tumour. The pathological

lesions are characterised by periosteal and endosteal hyper-ossification of the sternum, clavicles and upper ribs as wellas ossification of the surrounding soft tissues[4].

Plasmacytic infiltration with liberation of an agent forosteoblastic induction or osteoclast inhibition has beenpostulated as an explanation for bone production in a va-riety of conditions including sternoclavicular hyperostosis[3].

Fragility of the bone due to remodelling has been sug-gested in sternoclavicular hyperostosis[3]. However, biome-chanical testing in dominant endosteal hyperostosis revealedthat both cortical and cancellous bones were stiffer than nor-mal [6]. Histological evaluation revealed that metaphyseal

Page 3: Sternoclavicular hyperostosis with pathological fracture of the clavicle—a case report

466 S. Raja et al. / Injury, Int. J. Care Injured 34 (2003) 464–466

Fig. 4. MRI scan at 1 year follow up showing healed fracture.

trabeculae and cortical thickness were greater in dominantendosteal hyperostosis than in normal bone[6].

The diagnostic yield of radiography is poor and mislead-ing, particularly in the early stages, even on specific viewsof the sternoclavicular joint.

A CT scan demonstrates clearly the extent of the hyperos-tosis, the presence of a soft tissue mass or a direct mechanicaleffect of the hyperostosis on the vascular structures. Labo-ratory investigations are non-specific. Moderately increasedESR, CRP and leucocytes have been described[1,5].

Treatment has been ineffective. Clinical symptoms (chestpain and pustulosis) have occasionally improved follow-ing antibiotic treatment[1]. Nonsteroidal anti-inflammatorydrugs control symptoms and may prevent bone formation.

Surgical resection of the medial end of the clavicle andnew bone has been reported[5]. This condition is probablyself-limiting and tends to become quiescent after a decadeor longer of exacerbation and remissions[1].

References

[1] Chigira M, Maehara S, Nagase M, Ogimi T, Udagawa E. Sterno-costoclavicular hyperostosis: a report of 19 cases with specialreference to etiology and treatment. J Bone Joint Surg Am 1986;68A:103–11.

[2] Lagier R, Arroyo J, Fallet GH. Sternocostoclavicular hyperostosisradiological and pathological study of a specimen with ununitedclavicular fracture. Pathol Res Practice 1986;181(5):596–603.

[3] Sartoris DJ, Schreiman JS, Kerr R, Resnik CS, Resnick D.Sternocostoclavicular hyperostosis: a review and report of 11 cases.Radiology 1986;158(1):125–8.

[4] Sonozaki H, Furusawa S, Seki H, Kurokawa T, Tateishi A, Kabata K.Four cases with symmetrical ossification between the clavicles andthe first ribs of both sides. Kanto J Orthop Trauma 1974;5:244–7.

[5] Sonozaki H, Azuma A, Okai K, Nakamura K, Fukuoka S, Tateishi A,et al. Clinical features of 22 cases with inter-sterno-costo-clavicularossification: a new rheumatic syndrome. Arch Orthop Traumat Surg1979;95:13–22.

[6] Beals RK, Mcloughlin SW, Teed RL, Mcdonald C. Dominant endostealhyperostosis. J Bone Joint Surg Am 2001;83:1643–9.