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in 2005 , an otherwise healthy 55 year - old female patient , with a primary diagnosis of hip osteoarthritis and otherwise unremarkable history , underwent bilateral staged tha at another institution .
a cementless system with a ceramic - on - ceramic articulation was used on both occasions ( r3 acetabulum , synergy femoral stem ; smith & nephew , memphis , tn , usa ) .
commencing early postoperatively , the patient had recurrent dislocation of her right arthroplasty ( fig .
1a ) as often as 3 - 4 times a year , which she managed to reduce on her own .
six years postoperatively , the patient sustained a fracture of the right ceramic head ( fig .
the acetabular prosthesis was exchanged with a tantalum shell and an ultra - high molecular weight polyethylene liner .
the fractured ceramic head was exchanged with one made of cobalt - chromium alloy ( continuum acetabulum , versys head ; zimmer , warsaw , in , usa ) ( fig .
six months post - revision , the patient developed erythema nodosum of the left leg and bilateral chorioretinitis and papilledema .
a chest computed tomography ( ct ) revealed diffuse radiopaque nodules in both lungs and enlargement of the right hilum ( fig .
biopsy of the right lung showed fibrosis and extensive inflammation with multiple non - necrotizing granulomata , consisting of histiocytes , multinucleated giant cells , and lymphocytes ( fig .
the patient received corticosteroids and azathioprine for six months , which led to clinical and radiological improvement . between 2013 and 2015 , echocardiograms and chest magnetic resonance imaging showed severe cardiomyopathy with the ejection fraction decreasing from 70% to 35% and evidence of non - ischemic type fibrosis of the lower and posterior ventricular wall , as well as moderate dilation of the left ventricle .
physical examination revealed a 76 kg , 159 cm tall woman , with a severely antalgic gait and painful , decreased range of motion of the right hip .
the presence of a pseudotumor , extensive osteolysis , and the history of a fractured ceramic implant exchanged with a metal alloy head and a polyethylene liner , aroused suspicion for metallosis .
a hip aspiration produced black fluid with high concentrations of chromium and cobalt , 25,400 g / l and 26,500 g / l respectively ( fig .
whole blood chromium and cobalt concentrations were also significantly elevated , 31.8 g / l and 22.2 g / l respectively .
intraoperatively , gross local metallosis was found and large volumes of black fluid poured out from a trochanteric pseudotumor .
there was extensive osteolysis of the proximal femur but the stem remained fixed ( fig .
ceramic debris was also embedded in the articular surface of the polyethylene liner ( fig .
the morse taper of the stem was also damaged , allowing free rotation of the prosthetic head ( fig .
the acetabular shell , which remained well fixed , was left in place but the worn polyethylene liner was removed .
the femoral stem was exchanged with a long revision implant ( wagner sl revision uncemented stem ; zimmer gmbh , winterthur , switzerland ) . to prevent recurrence of the problem , a ceramic - on - ceramic articulation was inserted ( biolox delta ; zimmer gmbh ) ( fig .
, blood chromium and cobalt levels had returned to normal , 1.17 g / l and 0.19 g / l respectively .
one year later , the patient was able to mobilize painlessly , using one crutch .
to our knowledge , this is the first report , associating granulomatous lung disease with wear of metal implants , following the revision of a failed ceramic - onceramic tha .
granulomatous lung disease has been attributed to exposure to a variety of environmental and occupational agents , including mycobacteria , fungi , pollen , building materials and metallic dust and fumes34 ) .
sarcoidosis is a diagnosis of exclusion , suggesting an unrecognized causal factor , causing non - necrotic , non - infectious granulomas that are clinically and histologically indistinguishable from secondary granulomas3 ) .
since initially no causative agent had been identified , the overall presentation of the patient had been characterized as sarcoidosis .
the identification of the hip arthroplasty as a source of heavy metal ions and microparticles offered a reasonable specific etiology .
inhalation of metallic fumes and dust has been associated with pulmonary fibrosis and giant cell infiltration of pulmonary interstitial tissue4 ) .
metal nanoparticles are internalized by distant tissue cells , inducing chromosomal damage and oxidative stress2 ) .
macrophage and monocyte activation , following exposure to metal nanoparticles and ions , has been proven in vitro5 ) .
this event could be the first step in a cascade leading to granuloma formation6 ) .
it can be expected that similar tissue reactions occur with hematogenous spread of heavy metal particles .
et al.7 ) have reported granulomatous lesions in the spleen and liver after a metal - on - polythylene tha , with histological evidence of titanium and polyethylene infiltration of macrophages . in young patients ,
ceramic implants are considered more appropriate than the usual , metal - on - polyethylene bearings , because they produce less wear debris , allowing for increased longevity of the reconstruction . however , ceramic materials are associated with an up to 1.5% risk of fracture8 ) .
revision of a fractured ceramic implant , using a metal head and a polyethylene acetabular liner , may result in catastrophic wear of the new head1 ) . since complete removal of the ceramic debris from the periprosthetic tissues is impossible , embedding of remaining debris in the polyethylene liner acts as sandpaper , damaging the relatively softer new metallic head and producing metal nanoparticles and ions , causing local metallosis and systemic toxicity .
gessner et al.9 ) identified twenty five cases of systemic toxicity following metallosis from chrome / cobalt hip arthroplasty , with thyroid dysfunction , psychosocial manifestations , sensory and motor neuropathy , visual and auditory disturbances , or cardiomyopathy .
however , since five of these cases have been identified by the same team in alaska only , it is probable that systemic complications following metallosis from hip replacement are underreported9 ) .
it is therefore important , for both physicians and orthopedic surgeons , to suspect metallosis in patients who present with systemic disorders and have a history of a fractured ceramic bearing exchanged with a metal - on - polyethylene combination .
diagnostic difficulties arise with elderly patients , who , due to a lower activity level , have only mild symptoms from a potentially malfunctioning prosthetic joint , and whose medical problems can be easily attributed to comorbidities or medications .
guidelines , such as those of the uk medicines and health - related products regulatory agency10 ) , imposing frequent monitoring of blood ion levels and repeated imaging in arthroplasties with high risk for metal release , can lead to earlier diagnosis . with prompt synovectomy , lavage and revision to a ceramic - on - ceramic bearing , systemic complications of metallosis can be avoided .
a case of a female patient with local and systemic complications of metallosis , following catastrophic wear of a revised hip arthroplasty , is presented . 
 the patient had a history of a fractured ceramic - on - ceramic implant , exchanged with a metal - on - polyethylene prosthesis . 
 systemic complications included sarcoidosis - like reactions , presenting as granulomatous lung disease , along with chorioretinitis , erythema nodosum , and cardiomyopathy . 
 high local and circulating cobalt and chromium levels established the diagnosis . 
 the patient underwent extensive debridement and implant revision . 
 one year postoperatively , she had no respiratory symptoms or functional impairment 
 . local and systemic complications of metallosis after hip arthroplasty should be promptly recognized and treated operatively .