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Surgical Treatment of Juvenile Idiopathic Arthritis A Review.

Juvenile idiopathic arthritis (JIA) is a heterogeneous group of conditions encompassing all forms of unknown origin arthritis before the age of 16 years that persist for more than 6 weeks. (16) Its incidence and prevalence in European and North American populations range from 2 to 20 and from 16 to 150 per 100,000, respectively. (2)

Current classification of JIA is based on the criteria created by the Pediatric Task Force of the International League of Associations for Rheumatology (ILAR), proposing seven categories based on clinical and laboratory features during the first 6 months of illness. (2,7-10)

Systemic juvenile idiopathic arthritis (sJIA), also known as Still's disease, is an acquired auto-inflammatory disease equivalent to adult-onset Still's disease (AOSD). It is defined as a systematic disease with the involvement of one or more joints and the presence or precedence of quotidian fever for at least 2 weeks, plus at least: 1. characteristic rash, 2. generalized symmetrical lymphadenopathy, and 3. enlargement of liver or spleen or serositis (pericarditis and pleural or pericardial effusion). (1,2,6,7,9,11) It accounts for 4% to 9% of JIA (11) and affects males and females equally. (12) Two sub-categories are described: 1. sJIA that responds to Interleukin-1 blockade and 2. sJIA resistant or with intermediate response to Interleukin-1 blockade. (1)

Systemic juvenile idiopathic arthritis may be associated with macrophage activation syndrome (MAS), a life-threatening condition requiring rapid treatment. (6,12) Macrophage activation syndrome associates with continuous fever or rash. Additional features are myocarditis, renal and hepatic dysfunction, coagulopathy, respiratory or central nervous system manifestations, and cardiopulmonary arrest. (12)

Other categories of JIA are:

1. Rheumatoid factor positive polyarthritis (RF+), present in 5% of patients with JIA, involving more than four joints during the first 6 months of disease (13); RF+ resembles that of RF+ adult rheumatoid arthritis, but the main difference is the retardation or acceleration of growth on the young skeleton;

2. Rheumatoid factor negative polyarthritis (RF-), present in 20% of patients with JIA, involving more than four joints during the first 6 months;

3. Persistent oligoarthritis, present in 40% of JIA patients, affecting four or fewer joints;

4. Extended oligoarthritis, present in 20% of JIA patients, affecting more than four joints, 6 months after the disease onset;

5. Enthesitis related arthritis, present in 5% of JIA patients older than 6 years, affecting usually the Achilles tendon and plantar fascia;

6. Psoriatic arthritis, present in 6% of JIA patients; and

7. Undifferentiated arthritis (15%), for patients that do not meet the criteria for any category or who meet the criteria for more than one.

Morning stiffness is a common occurrence in JIA patients. Severe manifestations usually lead to up to a 60% incidence in difficulty at daily activities and disability (10%).

Erythrocyte sedimentation rate (ESR) and c-reactive protein (CRP) are elevated, most markedly in sJIA, as well as white blood count (WBC), platelet count (PLT), and ferritin (usually > 500 ng/ml in sJIA). (11) Radiological findings include early erosions, limitation of joint space, bony fusion deformities, protrusi acetabuli, wear, and abnormal development of the affected joints. (13) Systemic illness is responsible for progressive erosions and poly-arthritis can cause early joint destruction. Ultrasound may be useful in detecting synovitis, (14,15) while magnetic resonance imaging (MRI) is the preferred screening method to detect sacroiliitis. (14,16)

The cause of JIA is thought to be multifactorial and is still elusive. A genetic background (HLA and non-HLA genes) along with several environmental factors might trigger the onset of this autoinflammatory (IL-1, IL-6 proinflammatory cytokines) rather than autoimmune disease. (1,6)

The most commonly affected joints are the hip (50% to 60%), the knee (30% to 50%), and the shoulder (33%), as well as the wrist, the ankle joint, and the spinal column, depending on the subcategory of JIA. Early diagnosis and appropriate treatment using drugs, such as nonsteroidal anti-inflammatory drugs (NSAIDs), corticosteroids, and disease-modifying anti-rheumatic drugs (DMARDs), conventional or biologic, could provide adequate suppression of the disease. In cases of severe joint degeneration and destruction with deformity, poor motion, and severe pain, the physicians' last options are minor surgeries and, finally, total joint arthroplasty (TJA).

The purpose of this review is to summarize the studies conducted on surgical treatment of JIA, up to those published in 2018 (performing a computerized search in Medline for specific terms), in order to provide a better understanding of the current therapeutic strategies, methods, and outcomes.

Materials and Methods

We conducted a search of the literature in the databases Medline and PubMed, using the terms: "juvenile idiopathic arthritis", "juvenile rheumatoid arthritis", "surgical treatment/management", "total joint arthroplasty/replacement", "total hip arthroplasty/replacement", "total knee arthroplasty/replacement", "ankle arthroplasty /replacement", "elbow arthroplasty /replacement", "shoulder arthroplasty/replacement", "wrist arthroplasty /replacement". To maximize the search, backward chaining of reference lists from retrieved papers was also undertaken. Preliminary assessment of only the titles and abstracts of the search results was initially performed (510 papers). A careful review of the full-text publications was conducted during the second stage (103 papers and 1 textbook). Inclusion criteria were studies focusing on treatment of juvenile idiopathic arthritis that included surgical interventions and the follow-up outcomes. We excluded all studies that dealt exclusively with adult rheumatoid arthritis, adult arthritis and its treatment, as well as papers not written in English.

Two presentations from the American College of Rheumatology/Association of Rheumatology Health Professionals 2018 Annual Meeting, October 20-24, Chicago, Illinois, focusing on treatment guidelines for juvenile idiopathic arthritis as well as a National Institute for Health and Care Excellence guideline for canakinumab, were included in this review.

Pharmaceutical Treatment of Juvenile Idiopathic Arthritis

Juvenile idiopathic arthritis requires intensive treatment from the beginning, especially in cases where poor prognostic factors are identified. Suppression of the disease to a state of low disease activity (LDA) is the main concern, whereas pain relief and alteration of the course of the inflammation are critical. The goal of therapy is to establish disease remission and maintain it. (8) Almost 60% of the patients, if left untreated, will have great difficulties in daily activities.

Recommendations for a therapeutic strategic plan were first introduced by the American College of Rheumatology (ACR) in 2011 and updated in 2013. New recommendations were recently developed by an ACR initiative and were introduced for discussion at the ACR's 2018 meeting. A new guideline is anticipated in the year following the discussion. (17)

The treatment strategy is based on a combination of drugs, physical therapy, psychological support, and social development. (12) A well-organized intervention team of pediatric rheumatologist, physical therapist, ophthalmologist, pediatric orthopedic surgeon, and pediatric psychiatrist specialized in this complex entity should collaborate in combining updated therapies in order to suppress the disease and improve the quality of life. (4)

Nonsteroidal anti-inflammatory drugs are usually used for symptomatic relief, playing a minor role in altering the disease process, in most subcategories of JIA. If the disease is still active, the use of NSAIDs as monotherapy is not suggested after 2 months. (29) Drugs such as naproxen, ibuprofen, and indomethacin and meloxicam (COX-1 and COX-2 inhibitors) are used as initial treatment, acting as analgesics and anti-inflammatory agents. (4,7,18)

The use of intra-articular corticosteroids (IAC) with a preference to triamcinolone hexacetonide or systemic corticosteroids, such as prednisone and methylprednisolone, is of importance. Intra-articular corticosteroids are an option for rapid pain and inflammation relief, especially in oligoarthritic cases as they yield better functional outcomes. (1,2,4,9,19) The use of IAC is also recommended for alleviating persistent arthritis in a single joint and can be used in intervals of 3 to 4 months in the same joint without compromising it. Systemic glucocorticosteroids serve as an add-on therapy for a short period of time, both in systemic JIA and the poly-arthritic subgroup. Nevertheless, side effects, such as osteoporosis, avascular necrosis of the bone, hypertension, hyperglycemia, growth suppression, infections, and inability to prevent joint destruction, should be taken into consideration. (6) Systemic administration of corticosteroids is also used for extra-articular signs and symptoms of JIA. Prednisone 0.5 to 2 mg/kg/day, at a maximum of 60 mg/day, can be given in a single dose or as divided doses. As far as methylprednisolone is concerned, a dose of 10 to 30 mg/kg/day, with a maximum of 1 g/day, for 1 to 3 days, is used in cases of pericarditis, myocarditis, central nervous system or pulmonary manifestations, as well as MAS (pulse iv dose, plus an IL-1 inhibitor or a calcineurin inhibitor). (2,4,12)

Many conventional (as in "non-biologic") DMARDs are widely used with relative safety in children with JIA and are considered maintenance therapy for the disease. This includes methotrexate (MTX), sulfasalazine (SSZ), leflunomide (LEF), and hydroxychloroquine (HCQ). (2-4,6,8,9,11,12,20)

Methotrexate (MTX) is the most widely prescribed conventional DMARD for JIA. It is safe and effective at achieving disease control, especially in oligoarthritic JIA and also in poly-arthritic or systematic JIA complicated by active arthritis. (9) It has a favorable dosage regimen once weekly, which is very important in children's compliance to treatment and can be administered parenterally (subcutaneous or intramuscular injection) and has a long half-life. With an immune-modulatory effect on low doses, by inhibition of interleukin-1 production and blockade of multiple cellular functions, with reduction of purines being the most prominent, MTX treated patients seem to have relatively good response in achieving remission. Serious side effects of MTX are related to liver toxicity and bone marrow suppression, (2-4,6,9) but the most common side effects are attributed to gastrointestinal disturbance. The usual dosage is 0.5 to 1 mg/kg/week orally or subcutaneously, with a maximum of 25 mg/week. (21)

Leflunomide seems to be also a good choice, but plays a secondary role on the therapeutic plan of the physicians, mostly in patients who cannot tolerate MTX. Immuno-modulatory action is attributed to inhibition of pyrimidine and T-cell proliferation. There are not enough studies to actually evaluate its true effect on JIA remission, and according to ACR recommendations, it should be initiated as an alternative to MTX after the later has failed or is inappropriate for treating an individual. The usual dosage regimen is 10 mg daily (orally) for children weighing less than 40 kg and 20 mg daily for those who are over 40 kg. (2,3,9,20)

Sulfasalazine is usually used in oligoarthritic disease and enthesitis-related arthritis, but its effect is still controversial since few studies have been initiated to evaluate response in a randomized control manner. (4) Immunomodulatory activity, which is still not well understood, is mainly accredited to cytokine inhibition and T-cell activity down-regulation. The usual dosage regimen consists of 50 mg/kg/day (maximum 2 g) orally, divided twice daily. Gastrointestinal disturbance and liver toxicity are the most commonly anticipated side effects.

Hydroxychloroquine (6 mg/kg/day) is usually recommended in combination with other DMARDs to enhance the possibility of remission and maintenance of therapeutic results. Monotherapy with HCQ is not advised. (22)

Biological Therapies: A New Area of Treatment of Juvenile Idiopathic Arthritis

In recent years, pharmacotherapy of JIA has changed significantly with the introduction of host immune response modifiers known as biologic agents that block and regulate the action of proinflammatory mediators or disrupt specific cell-to-cell communication or cell differentiation and proliferation. Patients with JIA who do not respond satisfactorily to traditional, conventional disease-modifying antirheumatic drugs and glucocorticoids are currently treated with biologic agents, such as TNFa, IL-6 or IL-1 inhibitors as well as B-cell-activation or T-cell-activation inhibitors, with the aim of suppressing inflammation. (17,23-25)

The IL-1 inhibitor Anakinra can be used for alleviating signs and symptoms of sJIA but more importantly critical complications, such as MAS. It can be used for children as early as 8 months of age and after NSAIDs and corticosteroids have failed. Children weighing less than 50 kg are dosed by body weight with a starting dose of 1 to 2 mg/kg/day, while patients weighing 50 kg or more are dosed with 100 mg/day. In children who exhibit an inadequate response, the dose can be escalated up to 4 mg/kg/day. (17)

Another IL-1 inhibitor, canakinumab, was recently approved for use in sJIA for children over 2 years of age. The recommended dose of canakinumab for patients with sJIA and body weight of 7.5 kg or more is 4 mg/kg (up to a maximum of 300 mg) administered every 1 week via subcutaneous injection. (26)

Both treatment options are strongly advised for sJIA complicated with MAS or active systemic disease, excluding active arthritis.

Another interleukin inhibitor, the humanized IgGl monoclonal antibody, tocilizumab, targets specifically the receptor of IL-6. This agent can be used both in systemic JIA and the poly-arthritic or extended oligoarthritic subgroups. It can serve as monotherapy if MTX is inappropriate for the individual and in children after the age of 2. The recommended dosage in patients above 2 years of age is 8 mg/kg once every 2 weeks for patients weighing greater than or equal to 30 kg and 12 mg/kg once every 2 weeks for patients weighing less than 30 kg. It can also be used subcutaneously with a dosage of 162 mg weekly if the patient weighs more than 30 kg, or every other week when body weight is lower than 30 kg. (17,27)

For polyarticular, oligoarticular, and ethesitis-related JIA, as well as sJIA with active arthritis and no systemic prevalent features, the group of anti-TNF[alpha] agents is strongly advised to initiate after MTX or other conventional DMARDs have failed to drive the disease into remission or maintain it after 3 to 6 months of continuous treatment.

Adalimumab, etanercept, and most recently golimumab have been approved for use in patients older than 2 years of age. Infliximab has been used with success in pediatric patients for other indications (psoriasis and inflammatory bowel disease) but has never been tested in patients with JIA in a randomized control trial. Nevertheless, its use for JIA was widespread owing to significant and sustainable results in treating uveitis and sacroiliitis in children. Certolizumab pegol has also been used with success, but only case series are reported in literature. (5,22)

Another biologic DMARD in use for pediatric patients with polyarticular JIA is abatacept, a fusion protein that consists of the extracellular domain of human cytotoxic T lymphocyte-associated antigen 4 (CTLA-4) linked to a modified Fc portion of human immunoglobulin G1 (IgGl). Abatacept selectively modulates a key costimulatory signal required for full activation of T lymphocytes expressing CD28 by specifically binding to CD80 and CD86. This agent has been approved for children over the age of 6 and is to be initiated after a DMARD, including a TNF[alpha] inhibitor, has failed to set the disease in remission or in patients where the use of a TNF[alpha] agent is inappropriate. The recommended dose forpatients 6 to 17 years of age with JIA who weigh less than75 kg is 10 mg/kg calculated based on the patient's body weight at each administration. Pediatric patients weighing 75 kg or more should be administered abatacept following the adult dosing regimenbut not to exceed a maximum dose of 1,000 mg. (5,17,25)

Rituximab is a chimeric monoclonal antibody that binds specifically to the transmembrane antigen CD20, a non-glycosylated phosphoprotein, located on pre-B and mature B lymphocytes, inhibiting further differentiation and proliferation to antibody producing plasma cells. Rituximab has not been evaluated for the pediatric population, but is recommended for the RF+ poly-arthritic subgroup of JIA if DMARDs and at least one other biologic, including a TNF[alpha] inhibitory agent, have been insufficient to produce remission of the disease. The recommended dosage is 750 mg/m2 (maximum 1,000 mg) by intravenous infusion followed by a second 1,000 mg intravenous infusion 2 weeks later. The need for further courses should be evaluated 24 weeks following the previous course. Retreatment should be given at that time if residual disease activity remains, otherwise retreatment should be delayed until disease activity returns. Co-treatment with methotrexate is strongly advised. (5,17,25)

The advance in biologic therapeutics resulted in marked improvement in JIA treatment and there is increasing evidence of the efficacy and the safety of distinct biologic agents while new drugs are being tested. (24) Despite the efficacy of these new agents, there are some major concerns about the long-term safety of biologic agents in the pediatric population because they have been associated with infusion and injection reactions, infections (especially tuberculosis and hepatitis), autoimmune diseases, cytopenia, and the development of malignancies (e.g., lymphomas and skin cancer for anti-TNF[alpha] inhibitors). The actual risk of these adverse outcomes is largely unknown in pedriatics.

Infusion reactions are frequently observed upon treatment with biologics due to the fact that they are in part xenoproteins and may lead to the development of antidrug antibodies. Moreover, autoimmune diseases (e.g., lupuslike reaction and sarcoidosis) were occasionally reported in clinical trials performed in JIA patients. Thus it is not clear yet if they develop upon treatment or not. Juvenile idiopathic arthritis patients frequently develop mild infections. The incidence of severe infections (bacterial, viral, and fungal infections) although rare, seems to be higher in biologic treated JIA patients compared to patients treated with other conventional disease-modifying antirheumatic drugs. Furthermore, cytopenias (decreased neutrophil values) are occasionally described in JIA upon treatment with TNF[alpha] inhibitors as well as IL-6 and IL-1 inhibitors. Finally, there are reports suggesting an elevated, although low, risk of developing malignancy, especially hematologic, in JIA patients treated with biologic therapies. (24)

Surgical Treatment of Juvenile Idiopathic Arthritis

Juvenile idiopathic arthritis is a disabling disease, in terms of pain and function, affecting the growing skeleton as well as the overall psychological status of the patient. Younger patients with JIA usually have more severe disease than adult RA patients. (28) The use of NSAIDs, corticosteroids, and conventional DMARDs is of great importance for suppressing the disease. Furthermore, new advanced biological therapies have opened a new pathway for disease modification and suppression. Despite all the advances in pharmaceutical therapy, up to 10% of patients in one earlier study suffered end-stage degenerative arthritis affecting primarily the hip and knee and required joint replacement. (29) In fact, as reported in the past, around 10% to 50% of the patients will develop severe active arthritis, and without the appropriate treatment 17% of them will use a walking aid during the first 10 years of the disease. (28-30)

Surgical management should be done thoughtfully to those who suffer from disease uncontrolled with drugs, pain, joint stiffness, deformities, and motion difficulties. (31) Minor surgeries should be considered first. Such operations are corrective osteotomies, arthroscopic synovectomies (mainly for pauciarticular types), (28,32,33) tenotomies, arthrodesis, soft tissue release, (34-36) and unicompartmental joint replacement. Total joint replacement is the last option for patients whose bones and joints have major degeneration and destruction.

The surgical management of these patients requires a multidisciplinary team approach due to the age and major differences of the growing, severely affected skeleton. Evaluation of the cervical spine stability and for mandibular hypoplasia is necessary for maintenance of a safe airway during anesthesia. (29) Pre-, peri-, and postoperative management of medications for JIA should be done thoughtfully because of the risk of delayed wound healing and infections (e.g., corticosteroids and MTX), which cause a relapse of the disease or compromise other joints. (37,38)

Total Hip and Knee Replacement (Table 1)

Juvenile idiopathic arthritis is the third most common indication for lower limb joint replacement in Northern Europe and North America, with poor prognosis after years of active disease. (39) The pharmaceutical treatment advances have managed to decrease the rates of hip and knee replacement by 50% compared to the highest rates seen in the 1980s and 1990s (40,41) before the era of biologics.

The hip joint is affected by JIA in around 50% to 60% of the cases. (14,42-44) Common clinical findings are pain, which may radiate, and limping that may be indicative of a flexion contracture. (14) Total hip athroplasty (THA), (Fig. 1), is a well-established technique and should be performed when non-operative management has failed. It provides pain relief and good functional outcome, and its benefits are well documented. (45-51) A femoral stem and a properly fixed acetabulum is the mainstay of treatment. Cemented (45,52,53) and uncemented techniques (54,55) have been used and reported. Fractures or dislocations, perforation of the femoral shaft, venous thromboembolism, infections, anemia, as well as early revision are some of the usual complications. (39,56)

Total hip arthroplasty is challenging for the surgeon. Long life span, small abnormally shaped bones (femur and acetabulum), thinner femoral cortices and smaller femoral canals, excessive femoral and acetabular anteversion, growth and re-modeling of bone, and osteoporosis from steroids and soft tissue contractures present the surgeon with a very complex challenge, requiring detailed preoperative planning. (29,57) Cemented techniques resulted in aseptic loosening rates of 19% to 57% at 5 to 10 years of follow-up, according to Williams et al. (51) and Chmell et al. (58) Whereas Kumar et al. (55) and Odent et al. (59) reported that cementless primary THAs showed a survivorship of 96% to 100% for the femoral stem and 88% and 90% for the acetabular cup at a follow-up of 5 to 13 years. On the other hand, Malviya et al. (44) and Li et al. (60) reported no clear difference in implant survival between cemented and uncemented techniques.

Kitsoulis et al. (29,47) reported 20 THAs in which 100% had uncemented acetabular cups and and 50% cemented femoral stems. Excellent pain relief was reported in 100% of the patients, and no femoral stems failures were reported over the 10-year follow-up period. Similarly, Lachiewicz et al. (46) reported 100% survival in 10 patients at 4.5 years, with a mean postoperative Harris hip score of 78, while Lehtimaki et al. (53) reviewed 186 cemented THAs in 116 patients with JIA and reported an overall survival of 91.9% at 10 years follow-up.

Restrepo et al. (61) noticed a significant improvement in function and relief of pain as measured by the Harris Hip score and SF-36 for 35 consecutive uncemented THAs in 25 patients who were between 13.5 to 20 years of age. Nine patients suffered from JIA. No complications or reoperations were reported except for one revision due to severe polyethylene wear. Uncemented total hip arthroplasty was found to provide a significant improvement in function with acceptable short-term outcome. In another study, Bilsel et al. (62) studied 37 hips (23 cemented, 14 hybrid) of 23 JIA patients with a mean age of 22 years who underwent THA. The Harris hip score increased during a mean follow-up of 135 months. In general, all patients were satisfied and were able to walk without support. Three hips required revision. Launay et al. (63) performed 17 uncemented THAs in 13 children suffering from JIA. There was one acetabular loosening and all patients were satisfied. Unsatisfactory function was found in only 17% postoperatively, while the Harris-hip score improved. In another study from Swamp et al., (43) implant survival and outcomes after THA in patient with JIA were reported. For 56 patients and a mean follow-up of 12 years, the 10-year implant survival was 85%. Hip disability and osteoarthritis outcome scores (HOOS) were better at follow-up, but significantly worse in women and patients with custom implants or history of revision THA.

De Ranieri et al. (64) reported the results of 37 primary THAs (26 cementless femoral components and cementless acetabular components with screws) in 24 patients with JIA (average age 22.6 years). Follow-up was up to 19.6 years. Twelve hips failed, six acetabular components were revised, and three cementless femoral stems failed. One cemented stem and two cementless stems loosened. In a study from Bessette et al., (65) a 67% survival rate was reported at 10 years for 16 THRs for JIA using cemented and uncemented components, while Torchia et al. (66) reported 73% survival of cemented THRs at the same period of follow-up.

Malviya et al. (44) studied the prosthesis survival for THAs due to JIA in correlation to the use of steroids and MTX. Survival rates were significantly worse (p = 0.001) inpatients taking corticosteroids and better for those taking MTX (p = 0.02). In a case study, Malizos et al. (67) described the protrusion of a ceramic femoral head through the acetabular metallic shell in a 24-year-old patient suffering from JIA. The patient underwent cementless left THA. After 14 years, extensive wear of the polyethylene liner of the cup, resulting in direct articulation and abrasion wear of the ceramic femoral head on the cup, was noticed. They claimed that it might was the first description of extensive metallosis.

As a whole, primary THAs have good implant survival rates. Males have better outcomes compared to females, and primary THAs show better outcomes compared to revision THAs. (8) Revision surgery is more challenging due to systematic disease, multiple affected joints, contractures, narrow canals, and thin walls leading to intraoperative fractures and infections. (29,68) The longevity of THAs is based on factors such as small and abnormal bones (excessive anteversion and hypoplasia), growth and remodeling of bone, steroids, osteoporosis, and soft tissue contractures.

Goodman et al. (69) recorded the need for 17 hip revisions after THA in a follow-up period of less than 12 years; revisions were performed for 10 femoral components, 15 acetabular components, 4 cemented cups, and 6 cementless cups. The reason for revision was loosening (two patients), worn out prostheses, and infections. All of the patients during this follow-up had been facing major kinematic problems and pain. In another study, Goodman et al. (68) reviewed 24 THAs in 15 patients suffering from JIA. The mean follow-up was 9 years. The Harris hip score improved, but complications included two proximal femoral fractures and one sciatic nerve palsy. Seven hips (29%) required reoperation or revision surgery (three for infection and four for mechanical loosening).

Wroblewski et al. (45) reported 292 Charnley low-friction arthroplasties in patients with JIA with a mean follow-up of 36 years. They reported that 45% of the surgeries improved hip function and 90% of the hips presented without pain. In another study, Wroblewski et al. (70) reported on 39 THRs in 28 patients with a mean age of 17.9 years. Of the remaining 35 THRs (four lost to follow-up), 16 (45%) required revision at a mean of 19.1 years.

Daurka et al. (57) reported the mid-term results of 52 uncemented THRs in 35 patients with JIA. The mean age at the time of surgery was 14.4 years. The mean follow-up was 10.5 years. Thirteen of the 52 THRs underwent revision surgery. With revision as an endpoint, 100% survival of the 23 ceramic-on-ceramic THRs and 55% of the metal or ceramic-on-polyethylene was reported (94% for the femoral component and 62% for the acetabular component). Revision of the acetabular component for wear and osteolysis was reported in 11 of the 13 revisions.

The knee is one of the most commonly affected joints in JIA and is involved in 30% to 50% of cases at the time of diagnosis (especially in the oligoarthritic subtype). Clinical features include pain, swelling, tenderness, and bursitis (especially suprapatellar). In some cases, quadriceps muscle atrophy may be present. Both knees are usually affected. (14)

The surgical treatment for a partially or totally destructed knee joint due to JIA include arthroscopic synovectomy, which can delay the erosive process, (71) uni-compartmental knee replacement, which improves motion range but do not last, (72) osteotomy, soft tissue release, reconstructive operations, cup arthroplasty, stapling, especially in genu valgum, and fusion of the knee, which is not recommended in children.

Total knee arthroplasty (Fig. 2) is a successful procedure for managing end-stage arthritis and has reported survival rates of almost 95% at 10 years and just below 90% at 20 years. (73-75) Challenges for TKA in JIA patients include poor bone quality, severe joint deformities and flexion contractures, and the immunosuppressive drugs that patients receive. In case of contractures in both knees, some authors prefer bilateral simultaneous TKAs. The mainstay is to operate on the most painful joint first. (29,76)

Heyse et al. (73) reported the results of 349 TKAs inpatients with JIA at a mean follow-up of 12 years. The average age at surgery was 28.9. The 10-year survivorship was 95%, decreasing to 82% by 20 years. At latest follow-up, 8.9% of TKAs had been revised for either polyethylene failure or loosening, infection, stiffness, and instability. In a retrospective study of 34 TKAs in 20 JIA patients from Malviya et al., (77) a 41.5% failure rate at 20 years was found. The median age was 35 years with median follow-up of 16 years. Total knee arthroplasty resulted in pain relief despite the poor survival rates.

Jolles et al. (78) reviewed 14 adult patients (22 knees) with severe JIA treated with primary TKA. They reported a major positive impact on quality of life after TKA (measured by the Patient-Specific Index). All patients were satisfied regarding pain relief. They found no revisions at a mean follow-up of 8 years. In their study, Thomas et al. (79) reported the functional outcomes and complications of a series of 17 cemented TKAs during a 10-year period. During a follow-up of 74 months, the Knee Society scores, range of motion, and ambulation scores improved. Complications included two transient regional pain syndromes and one patellofemoral subluxation.

Palmer et al. (80) studied 15 TKAs in 8 patients with JIA and a mean age of 16.8 years. The mean follow-up was 5.5 years. They stated a high improvement in pain and function in all knees. Failure occurred in three patients. Similarly, Parvizi et al. (81) performed 27 total knee arthroplasties in patients with JIA under the age of 20. Reduced pain and better function but no great difference in the range of motion were noted. Re-operation was done in four knees because of adhesions and knee misalignment.

Finally, Lyback et al. (82) assessed the influence of patellar resurfacing after knee replacement and the frequency of patella infera and its relation to the postoperative appearance of the knee pain in patients with JIA. They performed 77 TKAs in 52 patients and reported results at a follow-up of 7.3 years. Anterior knee pain was present in 47% of those with an unreplaced patella and in 11% with patella resurfacing. No connection between patella infera and anterior knee pain was found.

In cases of multiple joint involvement, such as hip and knee, it is suggested that THA should be performed before TKA in order to facilitate knee rehabilitationby first having a pain free, flexible hip joint that provides full knee extension in the absence of hip contractures. When THA is performed first, referred pain to the knee from hip pathology is also eliminated. (29)

Bilateral THAs or TKAs or simultaneous combined THAs-TKAs have been reported, especially in patients with flexion contractures, to facilitate rehabilitation. (28,29,39) Simultaneous TJA is associated with higher complication rates. Indications include the knee and hip contributing equally to gait inability and flexion contractures. Positive results reported in the literature, reduction in cost, and reduction in the time needed for rehabilitation are major factors for choosing this therapeutic approach, according to some authors. (28,39)

Taheriazam et al. (39) reported on concurrent one-stage TKA and THA in a 34-year-old patient with a history of JIA. After 2-year follow-up, the patient showed excellent clinical function and remained satisfied. No loosening or dislocation was reported. In another study, Mullhall et al. (28) evaluated the long-term outcomes for combined, bilateral THAs and TKAs (cemented and uncemented prostheses for the hips and uncemented prostheses for the knees) on a group of six young patients (14 to 16 years old) with JIA. Five of the patients were wheelchair dependent before the surgery. During a follow-up period of 13.8 years (for the hips) and 17.3 years (for the knees), four of the six patients were unlimited community ambulators, one a limited community ambulator, and the remaining patient a household ambulator. Revision occurred in five hips (three patients).

Finally, Kitsoulis et al. (83) reported results of 15 THAs and 8 TKAs in patients whose mean age was 17.8 years and suffered from JIA. All of them had lessened joint space, joint sclerosis, flattened femoral head, and inclination of femoral neck. Three of them had their joint ankylosed. During a follow-up of 9.7 years, only one acetabulum was revised while there was still a good function of the affected joint. No pain remained and the daily activities returned to normal for all patients.

Upper Limb (Shoulder and Elbow)--Table 2

Approximately 33% of JIA patients experience shoulder involvement after 5 years of active disease. (8485) Stiffness, contracture, and internal rotation and adduction are the main signs of shoulder involvement. Shoulder, elbow, and wrist involvement, especially in polyarticular types of the disease, cause major disability in everyday simple activities. When both upper limbs are involved, then the patient faces major problems. (86)

First line treatment options for shoulder pathologies include intensive physical therapy and pharmaceutical agents to suppress or modify the disease. Synovectomies can be utilized, but they are not a usual option, and arthrodesis is not proposed in cases of bilateral shoulder involvement. Arthroplasty, described by Neer, (87) can improve pain but not motion and function due to soft tissue contractures. Bony deformity and osteopenia make these procedures demanding and the use of uncemented fixation is usually indicated due to narrow humeral canals. (86)

Jolles et al. (86) published a series of 13 shoulders that underwent arthroplasty as first line treatment for JIA. All the patients were satisfied in terms of pain relief and range of motion at the 9-year follow-up. In another study from Thomas et al., (88) nine shoulder hemiarthroplasties in JIA patients were reported. During a follow-up of 6 years, no need of re-operation was reported, while there were improvement in the range of motion (34%), function (33%), and pain (78%). According to their suggestions, a shoulder hemiarthroplasty requires the use of custom-made implants for stability.

The elbow joint is not often involved in JIA, however when it is involved, there is pain, swelling, and stiffness. Joint destruction and soft tissue contractures are present in end-stage arthritis of the elbow. Several operations could be proposed, but the most frequently performed are synovectomy with or without radial head excision, interposition arthroplasty, and the total elbow replacement (TER). (89-90) Given that the elbow joint is damaged very early in JIA, all the relevant bones are usually small and have narrow canals.

Published literature on the outcomes of elbow arthroplasty inpatients with JIA is scant. Pedenetal. (91) treated one fused elbow suffering from JIA and two from RA. At the 12-year follow-up, they reported good to excellent results despite the high rate of complications (e.g., infection) and the need for reoperation in over half of their patients. In addition, Maenpaa et al. (92) completed a study of 24 elbow synovectomies in patients with JIA in which 72% of joints were free of pain postoperatively and only six of them needed re-operation. No improvement in the range of motion or the function was reported.

Connor et al. (93) performed 24 TERs in 24 patients with JIA. They recorded an improvement of flexion (30[degrees]) and pain relief in 96% of the patients. In general, 52% of the elbows at the 2-year follow-up had an excellent outcome. Postoperative complications were fracture of the olecranon, stiffness, and loosening of the stems. Similarly, Baghdadi et al. (94) reported on 29 TERs in 24 patients with JIA. The mean age was 37 years and mean follow-up was 10.5 years. They reported six implant revisions and satisfactory overall (76%) functional results. The rate of TER survival from any revision was 96.4% and 79.9% at 5 years and 10 years, respectively.

Ibrahim et al. (89) reported on 21 TERs in 14 JIA patients (14 unlinked, 7 linked). The mean age at surgery was 39.5 years with a mean follow-up of 11.7 years. Eight elbows (42.9%) required implant revision (six unlinked, two linked). At 2 years, 15 elbows were pain free, while the mean range of motion arc improved and 20 of 21 elbows had good or excellent function.

Hand and Wrist (Table 2)

The arthritic hand and wrist in young patients usually presents with swelling and tenderness leading to muscle weakness, reduced grip force, and decreased range of motion. The hamate, capitate, and third carpometacarpal joint are most frequently affected by cartilage damage and proximal interphalangeal joints (PIP) are more frequently affected than metacarpophalangeal joints (MCP). (95) Ultrasound is very useful in diagnosis, revealing subclinical synovitis, especially in MCP and PIP joints. (96) Magnetic resonance imaging is also a very useful diagnostic tool, revealing tenosynovitis and bone marrow lesions in carpal and metacarpal joints. (97)

Treatment options include pharmaceutical agents such as NSAIDs, DMARDs, corticosteroids, physical therapy, and splinting. Surgery is the last option and includes synovectomies, tendon repair, joint arthroplasty, and arthrodesis (mainly after skeletal maturity). (95) For the wrist joint, the biaxial total arthroplasty seems to be the dominant procedure given that bones and tendons can accommodate it. There are surgeons who suggest synovectomy, excision of the ulnar head, fusion of the ulna and radius, or arthrodesis.

Except for biaxial arthroplasty, there is also the triaxial procedure, however is not a preferred treatment. The literature for biaxial arthroplasty is sparse. Cobb et al. (98) in 1996 were the first to suggest the biaxial total wrist arthroplasty as an alternative to arthrodesis for patients with rheumatic diseases involving the wrist joint. They had good results and cited that the main reason for failure was implant loosening, but unfortunately for this summary, they operated on only one patient with JIA. Rossello et al., (99) in 1997, launched the use of silicone during arthroplasty, but the presence of synovitis and the shortening of carpal space overwhelmed the 69% of good results. Stegeman et al. (100) studied 16 biaxial total wrist arthroplasties without the use of cement. The main indication was pain. Good outcomes and improvement in the range of motion was seen in 69%. The main complication was the dislocation of three stems.

Ankle (Table 2)

An accepted surgical treatment option for end-stage arthritis of the ankle joint due to RA and JIA is ankle fusion. (101-103) Another treatment option is total ankle arthroplasty (TAR). First generations of TARs included two-parts implants, which showed a high rate of failure. Current TARs have three components with a polyethylene insert between the two parts that allows gliding and rotation. (104) Early synovectomy could be valuable as well. (105,106)

Van der Heide et al. (104) evaluated 58 TARs in 54 patients suffering from RA (53 prostheses) and JIA (5 prostheses). During a follow-up period of 2.7 years, of the 52 patients who were alive (56 prostheses), 51 implants were still in place and showed no signs of loosening on the most recent radiographs. The scores for pain, disability, and range of motion were good. The main reason for failure was infection.

Cervical Spine in JIA (Atlantoaxial Subluxation--Table 2)

Atlantoaxial subluxation (AAS) is a complication of JIA, with 33% prevalence, resulting in pain, reduced motion, and neurological problems. Cervical spine involvement is between the wide range of 25% to 80%, presenting with pain and stiffness, headaches, and ear and facial pain due to C2 nerve root compression. (107) It is considered to be a poor prognostic factor by the American College of Rheumatology. (25)

The diagnosis of AAS is based on an anterior atlanto-dental interval (AADI) greater than 3 mm. Treatment goals in JIA cervical instability include pain improvement and neural tissue protection. Cervical fusion is a reasonable choice, however it does have complications, such as pseudarthrosis, re-dislocation, neurological injury, and malalignment of the cervical spine. Salem et al. (107) reported on a 10-year-old male patient who suffered from AAS due to a history of JIA. He underwent fusion and at his final review 6-years postoperatively, he was asymptomatic with no functional limitation.

Discussion and Conclusion

Juvenile idiopathic arthritis is a complex entity requiring a multidisciplinary approach from a team of experts in order to achieve adequate disease remission and an improved quality of life. Treatment is based on the JIA subtype and disease severity. (8)

Early intervention is key to preventing severe disabilities and morbidity. (4) Pharmaceutical agents, such as NSAIDs, corticosteroids, conventional DMARDs, and biologics, are the first line treatment. (4,8,9) With the advances in biology and genetics and the introduction of biologic agents, physicians have been able to improve the quality of life of patients and significantly reduce the rates of surgical interventions. (40) Despite the fact that biologic agents are generally safe, larger scale and longer term studies should be conducted on their safety and side-effects, especially the possibility of carcinogenesis. (2) The pediatric patient has different pharmacokinetic, pharmacodynamic, and pharmacogenomic patterns from adults, so targeted studies should establish new safe and effective dosage regimens, and therapies should be constantly reevaluated. The ultimate goal will be individualized therapy based on the genetic background and course of each sub-type of the disease. (8)

Therapeutic advances in the field of JIA have witnessed an explosion. Despite this fact, there is still a considerable number of young patients who develop end-stage arthritis, especially in the hip and knee joint. (1439) In the past, these patients were wheelchair-dependent, but with advances in total joint replacement, patients manage to participate actively in daily and sports activities. Most of the studies in the literature showed acceptable to very good results in terms of pain relief, overall function, and rates of reoperation and revision. (43,47,61,62,65,66,73) Total joint arthroplasty as well as revision surgery are very challenging due to morphological variations in the young affected skeleton (29,57,68) as well as the use of drugs that can adversely affect the bones, such as steroids. (44) As far as the use of cement in THA is concerned, studies report debatable results. Comparing the survivorship, some authors report differences between cemented and uncemented prostheses, (51,55,58,59) while other report no clear differences. (44,60) This probably has to do with the study setup (number of THAs reviewed, experience of the surgeon, quality of prostheses, and so forth).

Bilateral THAs and TKAs or simultaneous combined THAs-TKAs have been reported. (13,29,39) Despite the higher complications rates that are expected, there were studies reporting good results for this aggressive approach. Along with reduction in cost and rehabilitation time, it is a recommended approach in selected patients.

Upper limb, ankle, and spine involvement occurs less often but is disabling when it exists. Literature on the surgical management of the shoulder, elbow, hand, wrist, ankle, and spine for JIA patients is limited. Nevertheless, encouraging results have been reported in several studies. (86,88,89,91-94,98,104,107)

Treatment of JIA is developing and encompasses genetics, biology, and demanding surgical procedures (Fig. 3). A multidisciplinary team of experts is required in order to achieve inactive disease, support the patient and his family, and enable the young patient to participate in daily activities normally. More studies should be conducted, especially in the field of biologic therapy and genetic manipulation, so the idea of non-surgical, cost-effective, and safe intervention might become feasible. Until then, surgery seems as the last dependable, effective, and affordable permanent solution for end-stage arthritis in young patients suffering from JIA.

Disclosure Statement

None of the authors have a financial or proprietary interest in the subject matter or materials discussed herein, including, but not limited to, employment, consultancies, stock ownership, honoraria, and paid expert testimony.


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Leonidas Mitrogiannis, MD, PhD, Alexandra Barbouti, PhD, Evangelos Theodorou, MD, MSc, Aikaterini Kitsouli, MD, Georgios Mitrogiannis, MD, Panagiotis Kanavaros, MD, PhD, loannis Mitrogiannis, and Panagiotis Kitsoulis, MD, PhD

Leonidas Mitrogiannis, MD, PhD, Alexandra Barbouti, PhD, Aikaterini Kitsouli, MD, Georgios Mtrogiannis, MD, Panagiotis Kanavaros, MD, PhD, loannis Mitrogiannis, and Panagiotis Kitsoulis, MD, PhD, Laboratory of Anatomy-Histology-Embryology, Faculty of Medicine, School of Health Science, University of Ioannina, Ioannina, Greece. Evangelos Theodorou, MD, MSc, 251 Hellenic Air Force Hospital, Department of Rheumatology, Athens, Greece. Correspondence: Leonidas Mitrogiannis MD, PhD, Trauma and Orthopedics Resident, Gravias 1, 45221, Ioannina, Greece;

Caption: Figure 1 A, Preoperative anteroposterior radiograph of the pelvis of a 13-year-old girl with juvenile idiopathic arthritis. B, Postoperative radiograph obtained 9 years after first surgery. This patient had revision of the acetabular implant 6 months later.

Caption: Figure 2 Preoperative anteroposterior radiographs of the knee joint of a 17-year-old girl with juvenile idiopathic arthritis (left). Postoperative radiograph obtained 6 years after surgery (right).

Caption: Figure 3 Treatment pathways for patients suffering from JIA.
Table 1 Total Hip and Knee Replacement Studies Summary

Study and Year of
Publication             Results and Notes
Williams et al. (46)    Cemented THA aseptic loosening rates: 19% to 57%
(1993)                  Follow-up: 5 to 10 years
Chmell et al. (53)      Cemented THA aseptic loosening rates: 19% to 57%
(1997)                  Follow-up: 5 to 10 years
Kumar et al. (50)       Cementless THA survivorship for the femoral
                        stem: 96% to 100%; and for the acetabular cup:
                        88% to 90%
(1998)                  Follow-up: 5 to 13 years
Odent et al. (54)       Cementless THA survivorship for the femoral
                        stem: 96% to 100%; and for the acetabular cup:
                        88% to 90%
(2005)                  Follow-up: 5 to 13 years
Malviya et al. (39)     No clear difference in implant survival between
                        cemented-uncemented techniques
Li et al. (55) (2012)   No clear difference in implant survival between
                        cemented-uncemented techniques
Kitsoulis et al. (42)   100% uncemented acetabular cups, 50% cemented
                        femoral stems
(2006)                  100% pain relief
                        No femoral stems failures
                        Follow-up: 10-years
Kitsoulis et al. (78)   15 THAs
(2006)                  Age: 17.8 years
                        Lessened joint space, joint sclerosis, flattened
                        femoral head, and inclination of femoral neck
                        3 patients with ankylosed joint
                        Follow-up: 9.7 years
                        1 acetabulum was revised
                        No pain, daily activities returned to normal
Lachiewicz et al. (41)  10 patients (4.5 years)
(1986)                  100% survival
                        Postoperative Harris hip score: 78
Lehtimaki et al. (48)   116 patients
(1997)                  186 cemented THAs
                        Survival: 91.9% (10 years)
Restrepo et al. (56)    35 uncemented THAs (25 patients)
(2008)                  13.5 to 20 years old
                        Significant improvement in function and pain
                        relief (Harris Hip score, SF-36)
                        No complications, 1 reoperations
                        Uncemented total hip arthroplasty: significant
                        improvement in function, acceptable short-term
Bilsel et al.
(57) (2008)             37 hips (23 cemented, 14 hybrid THAs)
                        23 patients
                        22 years old
                        Follow-up: 135 months
                        Harris hip score increased
                        All patients were satisfied and able to walk
                        without support
                        3 revisions
Launay et al. (58)      17 uncemented THAs (13 children)
(2002)                  1 acetabular loosening
                        All patients were satisfied
                        Unsatisfactory function: 17% postoperatively
                        Harris-hip score improved
Swarup et al. (38)      56 patients
(2015)                  Follow-up: 12 years
                        10-year implant survival: 85%
                        Hip disability and osteoarthritis outcome scores
                        (HOOS): better at follow-up (worse in women,
                        patients with
                        custom implants, history of revision)
De Ranieri et al. (59)  37 primary THAs (26 cementless femoral
                        components and cementless acetabular components
                        with screws)
(2011)                  24 patients
                        Age: 22.6 years
                        Follow-up: 19.6 years
                        12 hips failed (6 acetabular components revised,
                        3 cementless femoral stems failed, 1 cemented
                        stem and 2
                        cementless stems loosened)
Bessette et al. (60)    16 THRs (cemented and uncemented components)
(2003)                  67% survival rate (10 years)
Torchia et al. (61)     73% survival of cemented THRs (10 years)
Malviya et al. (39)     Survival rates for THAs due to JIA were worst (p
                        = 0.001) in patients taking corticosteroids and
                        better for those
(2011)                  taking MTX (p = 0.02)
Malizos et al. (62)     24-year-old patient suffering from JIA
                        (cementless THA)
(2009)                  Described the protrusion of a ceramic femoral
                        head through the acetabular metallic shell
                        (after 14 years)
                        Might was the first description of extensive
Goodman et al. (63)     24 THAs (15 patients)
(2014)                  Follow-up: 9 years
                        Harris hip score improved
                        Complications: 2 proximal femoral fractures, 1
                        sciatic nerve palsy
                        7 hips (29%) reoperation or revision (3
                        infections, 4 mechanical loosening)
Goodman et al. (64)     17 hip revisions (10 femoral components, 15
                        acetabular components)
(2006)                  4 cemented, 6 cementless
                        Follow-up: < 12 years
                        Reason for revision: loosening (2 patients),
                        worn out prosthesis, infection
Wroblewski et al. (40)  292 Charnley low-friction THAs
(2007)                  Follow-up: 36 years
                        41 revisions
                        45% surgeries improved hip function
                        90% hips with no pain
Wroblewski et al. (65)  39 THRs (28 patients); 35 THRs (4 lost to follow
(2010)                  Age: 17.9 years
                        45% revision (19.1 years)
Daurka et al. (52)      52 uncemented THRs (35 patients)
(2012)                  Age: 14.4 years
                        Follow-up: 10.5 years
                        13 THRs underwent revision
                        100% survival (23 ceramic-on-ceramic THRs), 55%
                        survival (metal or ceramic-on-polyethylene),
                        with revision
                        as an endpoint
                        11/13 revisions: acetabular component (wear,
Mullhall et al. (23)    Combined, bilateral (cemented and uncemented
(2008)                  6 patients (14 to 16 years of age)
                        5 patients were wheelchair dependent before the
                        Follow-up: 17.3 years
                        4/6 patients unlimited community ambulators, 1/6
                        limited community ambulator, 1/6 household
                        Revision: 5 hips (3 patients)
Heyse et al. (68)       349 TKAs
(2014)                  Follow-up: 12 years
                        10-year survivorship: 95% (82% at 20 years)
                        8.9% of TKAs revised
Malviya et al. (72)     34 TKAs (20 patients)
(2010)                  41.5% failure rate (20 years)
                        Follow-up: 16 years
                        Pain relief
                        Poor survival rates
Jolles et al.
(73) (2008)             22 knees (14 adult patients) treated with TKA
                        Major positive impact on quality of life after
                        TKA (measured by the Patient-Specific Index)
                        Pain relief
                        No revisions
                        Follow-up: 8 years
Thomas et al. (74)      17 cemented TKAs (10-year period)
(2005)                  Follow-up: 74 months
                        Knee Society scores, range of motion, ambulation
                        scores improved postoperatively
                        Complications: 2 transient regional pain
                        syndromes, 1 patellofemoral subluxation
Palmer et al. (75)      15 TKAs (8 patients)
(2005)                  Mean age: 16.8 years
                        Follow-up: 5.5 years
                        Improvement of pain, function
                        3 failures
Parvizi et al. (76)     27 TKAs (patients < 20 years of age)
(2003)                  Diminishing of pain, better function
                        No great difference in ROM
                        Re-operation: 4 knees (adhesions, misalignment)
Lyback et al. (77)      Influence of patellar resurfacing after TKA
(2004)                  Frequency of patella infera and its relation to
                        the postoperative appearance of the knee pain in
                        patients with JIA
                        77 TKAs (52 patients)
                        Follow-up: 7.3 years
                        Anterior knee pain: 47% (unreplaced patella),
                        11 % (patella resurfacing)
                        No connection between patella infera and
                        anterior knee pain
Mullhall et al. (23)    Combined, bilateral TKAs (uncemented prostheses)
(2008)                  6 patients (14 to 16 years of age)
                        5 patients were wheelchair dependent before the
                        Follow-up: 17.3 years
                        4/6 patients unlimited community ambulators, 1/6
                        limited community ambulator, 1/6 household
Kitsoulis et al. (78)   8 TKAs
(2006)                  Mean age: 17.8 years
                        Follow-up: 9.7 years
                        No pain
                        Daily activities returned to normal
Simultaneous THA-TKA
Taheriazam et al. (34)  Concurrent one-stage TKA and THA
(2017)                  34-years old patient with a history of JIA
                        Follow-up: 2 years
                        Excellent clinical function, patient satisfied,
                        no loosening or dislocation

Table 2 Upper and Lower Limb and Cervical Spine Studies Summary

                Study and Year of
                Publication                  Results and Notes

Shoulder        Jolles et al. (81) (2007)    13 shoulders
                                             Arthroplasty as first line
                                             treatment for JIA
                                             100% satisfaction rate
                                             (pain relief, ROM)
                                             Follow-up: 9 years
                Thomas et al. (83) (2005)    9 shoulders
                                             Follow-up: 6 years
                                             No need of re-operation
                                             Improvement in ROM (34%),
                                             function (33%), pain (78%)
                                             Suggestion: shoulder
                                             hemiarthroplasty requires
                                             custom-made implants for
Elbow           Peden et al. (86) (2008)     1 fused elbow suffering
                                             from JIA and 2 from RA
                                             Follow-up: 12 years
                                             Good to excellent results
                                             High rate of complications
                                             Need for reoperation in
                                             more than 50% of patients
                Maenpaa et al. (87) (2003)   24 elbows
                                             72% pain-free
                                             6 re-operations
                                             No improvement of ROM,
                Connor et al. (88) (1998)    24 TERs (24 patients)
                                             Improvement of flexion
                                             (30[degrees]) and pain
                                             relief: 96%
                                             52% of the elbows with
                                             excellent outcome
                                             Follow-up: 2 years
                                             Complications: fracture of
                                             the olecranon, stiffness,
                                             stem loosening
                Baghdadi et al. (89) (2014)  29 TERs (24 patients)
                                             Age: 37 years
                                             Follow-up: 10.5 years
                                             Functional results were
                                             satisfactory (76%)
                                             6 implant revisions
                                             TER survival from any
                                             revision was 96.4% and
                                             79.9% (5 and 10 years,
                Ibrahim et al. (84) (2017)   21 TERs (14 unlinked, 7
                                             14 patients
                                             Mean age: 39.5 years
                                             Follow-up 11.7 years
                                             Implant revision: 8 elbows
                                             (42.9%, 6 unlinked, 2
                                             15 elbows pain free (2
                                             ROM improved
                                             Good or excellent function:
                                             20 of 21 elbows
Hand and Wrist  Cobb et al. (93) (1996)      Biaxial total wrist
                                             arthroplasty (alternative
                                             to arthrodesis)
                                             Only 1 patient with JIA
                                             Reason for failure: implant
                Rossello et al. (94) (1997)  Use of silicone during
                                             Synovitis and shortening of
                                             carpal space overwhelmed
                                             the 69% of good results
                Stegeman et al. (95) (2005)  16 biaxial total wrist
                                             69% good outcome and
                                             improved ROM
                                             Main complication: 3
                                             dislocations of stems
Ankle           Van der Heide et al.99       58 TARs (53 prostheses for
                                             RA, 5 prostheses for JIA)
                (2009)                       54 patients
                                             Follow-up: 2.7 years
                                             52 patients alive (56
                                             51 implants still in place,
                                             no signs of loosening
                                             Scores for pain,
                                             disability, ROM were good
                                             Reason for failure:
Cervical Spine  Salem et al. (102) (2016)    10-year-old male patient
                                             (AAS due to JIA)
                                             Cervical fusion
                                             Asymptomatic, with no
                                             functional limitation (6
                                             years postoperatively)

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Author:Mitrogiannis, Leonidas; Barbouti, Alexandra; Theodorou, Evangelos; Kitsouli, Aikaterini; Mitrogianni
Publication:Bulletin of the NYU Hospital for Joint Diseases
Article Type:Disease/Disorder overview
Date:Apr 1, 2019
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