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PURPOSE OF REVIEW: This review summarizes latest developments in treatment of juvenile spondyloarthritis (JSpA), specifically enthesitis-related arthritis (ERA) and juvenile psoriatic arthritis (JPsA). RECENT FINDINGS: There has been addition of biologic disease modifying antirheumatic drugs (bDMARDs) beyond tumor necrosis factor inhibitors (TNFi) for JSpA such as IL-17 blockers, IL-23 blockers, and janus activating kinase inhibitors with favorable safety profile. Conducting robust clinical trials for this subpopulation of JIA remains a challenge; extrapolation studies are being used to obtain approval from regulatory agencies. SUMMARY: Newer drug therapies have expanded the scope of treatment for patients with JSpA. bDMARDs such as adalimumab, etanercept, infliximab, and secukinumab have demonstrated clinically significant treatment efficacy in ERA and JPsA. Based on extrapolation studies, intravenous golimumab, etanercept, abatacept, and ustekinumab have gained Food and Drug Administration (FDA) approval for JPsA. Long-term follow-up studies continue to demonstrate acceptable safety profiles. There is need for more real-world data on drug efficacy from Registry studies and research on effective de-escalation strategies.
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Antirreumáticos , Artrite Juvenil , Humanos , Antirreumáticos/uso terapêutico , Artrite Juvenil/tratamento farmacológico , Espondilartrite/tratamento farmacológico , Artrite Psoriásica/tratamento farmacológico , Produtos Biológicos/uso terapêutico , Resultado do Tratamento , CriançaRESUMO
The role of the microbiota in multiple autoimmune diseases, including juvenile idiopathic arthritis (JIA) has earned substantial attention in the last 10 years. Increasing evidence suggests that the microbiota's link to JIA begins in early childhood, as early life events that influence the nature of the microbiota also appear to influence disease risk. In this review, we discuss these early life events including mode of delivery, infant feeding practice, antibiotics exposure, and other events and their impacts on the microbiota and on disease risk; reported abnormalities of the microbiota in children with JIA; mechanisms by which an altered microbiota at birth and later on in childhood may influence disease risk; and the prospects for therapeutic alteration of the microbiota in children with JIA.
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Artrite Juvenil/microbiologia , Disbiose/imunologia , Microbiota/imunologia , Antibacterianos/efeitos adversos , Artrite Juvenil/imunologia , Criança , Dieta , Exposição Ambiental/efeitos adversos , Humanos , Recém-Nascido , RiscoRESUMO
OBJECTIVE: Several advanced therapies have been licensed across the related conditions of psoriatic arthritis (PsA), Crohn disease (CD), ulcerative colitis (UC), and noninfectious uveitis. We sought to summarize results from randomized controlled trials (RCTs) investigating the efficacy and safety of advanced therapies for these related conditions in patients with PsA. METHODS: We updated the previous systematic search conducted in 2013 with literature reviews of MEDLINE, Embase, and the Cochrane Library (from February 2013 to August 2020) on this subject; only those new studies are presented here. The quality of evidence was assessed using the Grading of Recommendations Assessment, Development, and Evaluation (GRADE) framework. RESULTS: The number of RCTs meeting eligibility criteria were 12 for CD, 15 for UC, and 5 for uveitis. The tumor necrosis factor inhibitor (TNFi) class appears to be efficacious and safe across CD, UC, and uveitis, with the exception of etanercept. Interleukin 12/23 inhibitors (IL-12/23i) are efficacious for CD and UC. Phase II and III RCTs of Janus kinase inhibitors (JAKi) and IL-23i in CD and UC are promising in terms of efficacy and safety. IL-17i must be used with great caution in patients with PsA at high risk of inflammatory bowel disease (IBD). RCTs in uveitis have mainly studied adalimumab. CONCLUSION: We have identified 32 recent RCTs in IBD and uveitis and updated recommendations for managing patients with PsA and these related conditions. A multispecialty approach is essential to effectively, safely, and holistically manage such patients. Advanced therapies are not equally efficacious across these related conditions, with dosing regimens and safety varying.
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Artrite Psoriásica , Colite Ulcerativa , Doença de Crohn , Doenças Inflamatórias Intestinais , Uveíte , Humanos , AdalimumabRESUMO
OBJECTIVES: Spondyloarthritis (SpA) results from the interplay between genetic and environmental factors. An emerging modifiable factor is the human intestinal microbiota, which multiple studies in children and adults have shown to be abnormal in SpA patients, including enthesitis related arthritis and ankylosing spondylitis (AS). However, HLA-B27 itself appears to impact the contents of the microbiota and is more common in SpA patients versus controls, thus serving as a confounding factor in most comparative studies. METHODS: This was a cross-sectional study that evaluated the contents of the faecal microbiota among 29 patients with HLA-B27+ AS and 43 healthy adults who underwent 16S sequencing and genotyping as part of the TwinsUK Programme. RESULTS: HLA-B27 positive+ patients and healthy controls demonstrated substantial clustering based upon diagnosis. Decreased richness was observed among the AS patients, although measures of evenness were similar. After correction for multiple comparisons, several taxa - including Faecalibacterium prausnitzii and Coprococcus - were elevated in AS patients compared to controls, even when restricted to female subjects, while Bacteroides fragilis, Ruminococcus, and Akkermansia muciniphila were depleted in AS patients. CONCLUSIONS: Consistent with some previous studies, our study demonstrates in patients with AS associations with Coprococcus, Bacteroides, and Ruminococcus. Other findings, including increased Faecalibacterium, are inconsistent with previous studies and thus potentially underscore the necessity of evaluating HLA-B27 positive controls in studies evaluating the impact of the intestinal microbiota on SpA.
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Microbiota , Espondilartrite , Espondilite Anquilosante , Adulto , Criança , Humanos , Feminino , Espondilite Anquilosante/complicações , Antígeno HLA-B27/genética , Estudos Transversais , Espondilartrite/complicaçõesRESUMO
OBJECTIVE: Many children with chronic musculoskeletal pain conditions experience stigma which can have negative downstream consequences. This study compares ratings of clinical pain (current pain intensity and pain interference), experimental pain (temporal summation, cold water tolerance, and cold pain intensity), and pain-related stigma among three groups of youth with rheumatic conditions. The relations among ratings of pain-related stigma and pain variables were explored. METHODS: Eighty-eight youth aged 8-17 years with a diagnosis of juvenile idiopathic arthritis (JIA = 32), juvenile fibromyalgia (JFM = 31), or non-specific chronic pain (NSCP = 25) completed measures of clinical pain ratings (average 7-day pain intensity, day of assessment pain (DoA), and pain interference), experimental pain (cold pain tolerance, cold pain intensity, and temporal summation of mechanical pain), and pain-related stigma. Data analysis compared pain-related stigma and pain ratings across the three groups and examined the relations among pain-related stigma and pain ratings. RESULTS: Youth with JFM reported higher ratings of clinical pain and pain-related stigma than their counterparts with NSCP or JIA. However, there were no differences in experimental pain. Pain-related stigma was associated with greater ratings of pain interference, particularly for those with JIA and NSCP. Pain-related stigma was also associated with greater average daily pain intensity but not DoA. CONCLUSION: Youth with medically unexplained pain report greater stigma and worse pain than their peers; thus, robust assessment of pain in this population is necessary. Future work should longitudinally explore the impact of pain-related stigma on pain outcomes and treatment responses.
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OBJECTIVES: To evaluate the current practices in management of patients with juvenile spondyloarthritis (JSpA) who failed anti-tumour necrosis factor agents (anti-TNF). METHODS: An online survey was distributed to Childhood Arthritis and Rheumatology Research Alliance (CARRA) members of the JIA workgroup. Data collection included estimated number of JSpA patients who have failed anti-TNF therapy over two-year period, reasons for discontinuing anti-TNF therapy and other medications used afterward. The JSpA population was de ned as the following subtypes: enthesitis-related arthritis, psoriatic arthritis, undifferentiated spondyloarthritis, juvenile ankylosing spondylitis (AS) i.e. meeting modi ed NY criteria for AS before age 16, and reactive arthritis. Findings were summarised using descriptive statistics. RESULTS: The survey response rate was 36% (n= 60/169). The majority of participants were paediatric rheumatologists (93%). Many physicians have JSpA patients who failed anti-TNF therapy (63%). The most common reason for changing anti-TNF therapy was secondary non-response (72%). Sacroiliitis was the most important factor considered when assessing response to an anti-TNF agent and the most common reason for primary non-response (45%). When assessing anti-TNF failure for sacroiliitis, many (65%) felt imaging of the sacroiliac joints was the most important aspect in their decision making. The majority try a second anti-TNF agent after initial anti-TNF failure (87%) and switch to another medication class after 2 anti-TNF agents have failed (62%). CONCLUSIONS: More than half of paediatric rheumatologists surveyed have at least one JSpA patient who failed anti-TNF therapy. The majority failed because of secondary non-response. Sacroiliitis is an important but challenging aspect to manage for patients with JSpA.
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Artrite Juvenil , Sacroileíte , Espondilartrite , Espondilite Anquilosante , Adolescente , Artrite Juvenil/complicações , Artrite Juvenil/diagnóstico , Artrite Juvenil/tratamento farmacológico , Criança , Humanos , Sacroileíte/tratamento farmacológico , Sacroileíte/patologia , Espondilartrite/complicações , Espondilartrite/diagnóstico , Espondilartrite/tratamento farmacológico , Espondilite Anquilosante/complicações , Inibidores do Fator de Necrose Tumoral/efeitos adversosRESUMO
PURPOSE: The purpose of this study was 1) to compare condyle - fossa relationships in the temporomandibular joint (TMJ), and 2) to score condylar resorption by using a TMJ indexing system in patients with JIA and without JIA. METHODS: The present retrospective cross-sectional study included cone-beam computed tomography (CBCT) images obtained from the sagittal, coronal, and axial slices. In the multidisciplinary Pediatric Rheumatology Outpatient Clinic at The University of Alabama at Birmingham (UAB) children with JIA are also examined by a group of orthodontists working in the same institute from October 2018 to July 2019. The predictor variable consists of patients with JIA and without JIA. The primary outcome variables are the depth of the mandibular fossa, joint spaces, axial angles, medio-lateral width, and condyle resorption. Other study variables were age and sex. In this study, the measurements obtained from 2 different groups (with JIA and without JIA) are compared using a t-test, where Tukey is utilized to adjust for multiple comparisons. The left and right joints are analyzed separately as the paired t test conducted showed a significant difference between the 2 joints (P < .05). RESULTS: The study was comprised of 34 patients diagnosed with JIA and 34 healthy subjects. The depth of the mandibular fossa, the anterior joint spaces, the axial angles, and the resorption index showed statistically significant differences between the JIA and healthy groups in both left and right sides (P < .05). However, there was no statistically significant difference in the posterior joint spaces and mediolateral width between JIA and healthy groups in both sides (P > .05). CONCLUSIONS: The results of our study presented the destructive potential of juvenile idiopathic arthritis by using CBCT. CBCT scanning is a helpful tool in the evaluation of the radiographic result of TMJ.
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Artrite Juvenil , Artrite Juvenil/complicações , Artrite Juvenil/diagnóstico por imagem , Osso e Ossos , Criança , Tomografia Computadorizada de Feixe Cônico , Estudos Transversais , Humanos , Côndilo Mandibular/diagnóstico por imagem , Estudos Retrospectivos , Articulação Temporomandibular/diagnóstico por imagemRESUMO
Approximately 5% of children with juvenile idiopathic arthritis (JIA) are diagnosed with the psoriatic form of the disease. In recent years, there has been substantial scholarship demonstrating both heterogeneity within the disease as well as similarities with other forms of JIA, culminating in a recent proposal for the categorization of JIA that excluded the psoriatic form altogether. The purpose of the review is to summarize the clinical, epidemiologic, and genetic features of psoriatic JIA (PsJIA), comparing it with other categories of JIA including spondyloarthritis. We conclude that there are sufficient unique clinical and genetic features within PsJIA as well as similarities with its adult counterpart that warrant including it within the JIA paradigm.
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Artrite Juvenil/classificação , Artrite Psoriásica/classificação , Adulto , Idade de Início , Artrite Juvenil/epidemiologia , Artrite Juvenil/genética , Artrite Juvenil/imunologia , Artrite Psoriásica/epidemiologia , Artrite Psoriásica/imunologia , Criança , Comorbidade , Humanos , Modelos Imunológicos , Espondilartrite/classificaçãoRESUMO
OBJECTIVES: Patients with Crohn's disease often produce antibodies against flagellated intestinal bacteria. There are mixed data as to whether such antibodies are present in patients with spondyloarthritis. Our objectives were to evaluate for the presence of antibodies against intestinal organisms in children with enthesitis related arthritis (ERA). METHODS: Children with ERA and healthy controls were recruited at three sites. Sera were plated on a nitrocellulose array and incubated with labelled antibodies to human IgA and IgG. RESULTS: At UAB, patients and controls had similar antibody levels against the majority of the bacteria selected, with the exception of increased IgA antibodies among ERA patients against Prevotella oralis (1231 [IQR 750, 2566] versus 706 [IQR 428, 1106], p = .007.) These findings were partially validated at a second but not at a third site. CONCLUSIONS: ERA patients may produce increased IgA antibodies against P. oralis. The possible significance of this finding bears further exploration.
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Antígenos de Bactérias/sangue , Antígenos de Bactérias/imunologia , Artrite Juvenil/sangue , Artrite Juvenil/imunologia , Prevotella/imunologia , Artrite Juvenil/microbiologia , Criança , Doença de Crohn/imunologia , Doença de Crohn/microbiologia , Feminino , Humanos , Imunoglobulina A/sangue , Imunoglobulina G/sangue , MasculinoRESUMO
Studies have identified abnormalities in the microbiota of patients with arthritis. To evaluate the pathogenicity of human microbiota, we performed fecal microbial transplantation from children with spondyloarthritis and controls to germ-free KRN/B6xNOD mice. Ankle swelling was equivalent in those that received patient vs. control microbiota. Principal coordinates analysis revealed incomplete uptake of the human microbiota with over-representation of two genera (Bacteroides and Akkermansia) among the transplanted mice. The microbiota predicted the extent of ankle swelling (R2 = 0.185, p = 0.018). The abundances of Bacteroides (r = -0.510, p = 0.010) inversely and Akkermansia (r = 0.367, p = 0.078) directly correlated with ankle swelling. Addition of Akkermansia muciniphila to Altered Schaedler's Flora (ASF) resulted in small but statistically significant increased ankle swelling as compared to mice that received ASF alone (4.0 mm, 3.9-4.1 vs. 3.9 mm, IQR 3.6-4.0, p = 0.041), as did addition of A. muciniphila cultures to transplanted human microbiota as compared to mice that received transplanted human microbiota alone (4.5 mm, IQR 4.3-5.5 vs. 4.1 mm, IQR 3.9-4.3, p = 0.019). This study supports previous findings of an association between A. muciniphila and arthritis.
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Artrite/microbiologia , Microbioma Gastrointestinal , Adolescente , Animais , Tornozelo/patologia , Bacteroides/isolamento & purificação , Bacteroides/patogenicidade , Criança , Feminino , Humanos , Masculino , Camundongos , Camundongos Endogâmicos NOD , Verrucomicrobia/isolamento & purificação , Verrucomicrobia/patogenicidadeRESUMO
When lichen planus involves the scalp, it is known as lichen planopilaris, and when it involves the eye, it is known as ocular lichen planus; both are rare. Early detection and targeted therapy are crucial in preventing hair loss and scarring conjunctivitis. Little is known regarding appropriate treatment for lichen planopilaris. The objective of this case study is to present a new case of pediatric ocular lichen planus and lichen planopilaris and to identify all reported cases of pediatric lichen planopilaris, highlighting disease involvement, treatment, and response to treatment.
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Líquen Plano/diagnóstico , Dermatoses do Couro Cabeludo/diagnóstico , Criança , Ciclosporina/uso terapêutico , Olho/patologia , Feminino , Glucocorticoides/uso terapêutico , Humanos , Imunossupressores/uso terapêutico , Líquen Plano/tratamento farmacológico , Metotrexato/uso terapêutico , Prednisolona/uso terapêutico , Couro Cabeludo/patologia , Dermatoses do Couro Cabeludo/tratamento farmacológicoRESUMO
Juvenile psoriatic arthritis (JPsA), a subtype of juvenile idiopathic arthritis (JIA), constitutes 5% of JIA. The literature is inconsistent regarding features of JPsA, and physicians debate whether it is a distinct entity within JIA. A biphasic age of onset distribution has been noted. Early-onset disease is characterized by female predominance, small joint involvement, dactylitis, and positive antinuclear antibodies. Late-onset JPsA resembles adult-onset psoriatic arthritis (PsA), with male predominance, psoriasis, enthesitis, and axial disease. Recent studies report improved outcomes, likely due to the widespread use of traditional and biologic disease-modifying antirheumatic drugs. Conflicting HLA associations have been reported in JPsA, but notably both HLA class I and II allele associations are suggested. Similar to PsA cohorts, subjects with JPsA have a lower frequency of a protective interleukin 23R allele than controls or other JIA subtypes. Data in the Childhood Arthritis and Rheumatology Research Alliance (CARRA) patient registry suggest the aggressive characteristics of JPsA: 24.6% of children have joint damage 4.6 years after symptom onset. Pediatric and adult PsA classification criteria define different JPsA cohorts within the registry and support a previous suggestion that the International League of Associations for Rheumatology criteria for JPsA may be overly stringent. Increased collaboration between pediatric and adult physicians and comparative research on these clinically related conditions are warranted.
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Antirreumáticos/uso terapêutico , Artrite Juvenil/diagnóstico , Artrite Juvenil/tratamento farmacológico , Criança , Humanos , Avaliação de SintomasRESUMO
PURPOSE OF REVIEW: There has been increasing interest in the contents and function of the microbiota, as it relates to pediatric inflammatory diseases. Here, we discuss the factors underlying the development of the microbiota, its role in juvenile idiopathic arthritis (JIA) and prospects for therapeutic interventions in the microbiota. RECENT FINDINGS: The human microbiota undergoes a succession of changes, until it reaches a mature form. A variety of early-life exposures, including mode of delivery and form of feeding, can affect the contents of the microbiota and possibly impact upon long-term risk of developing autoimmune diseases. The microbiota is altered in children with JIA, including elevated Bacteroides genus in JIA as a whole and decreased Faecalibacterium prausnitzii in pediatric spondyloarthritis. Although there are limited data so far indicating that microbiota-based therapies can result in therapeutic improvement of arthritis, most of the data are on adults and thus may not be applicable to children. SUMMARY: Perturbations of the microbiota during childhood may result in the development of a microbiota associated with increased risk of pediatric rheumatic illness. Whether the microbiota can be targeted is a focus of ongoing research.
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Artrite Juvenil/imunologia , Microbioma Gastrointestinal/imunologia , Espondiloartropatias/imunologia , Adolescente , Artrite Juvenil/microbiologia , Artrite Juvenil/terapia , Bacteroides , Criança , Dietoterapia , Faecalibacterium prausnitzii , Humanos , Probióticos/uso terapêutico , Doenças Reumáticas/imunologia , Doenças Reumáticas/microbiologia , Doenças Reumáticas/terapia , Fatores de Risco , Espondiloartropatias/microbiologia , Espondiloartropatias/terapiaRESUMO
The last decade has witnessed an explosion of studies evaluating the impact of the human microbiota on a variety of disease states. The microbiota can impact diseases in multiple ways, including through abnormalities in the diversity and contents of the microbiota, as well as by acting as targets of immunologic dysregulation. Herein, evidence that the microbiota in spondyloarthritis is both altered and abnormally targeted by the immune system will be presented.
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Microbioma Gastrointestinal/imunologia , Doenças Inflamatórias Intestinais/imunologia , Intestinos/imunologia , Espondiloartropatias/imunologia , Animais , Artrite Juvenil/imunologia , Artrite Juvenil/microbiologia , Artrite Psoriásica/imunologia , Artrite Psoriásica/microbiologia , Artrite Reativa/imunologia , Artrite Reativa/microbiologia , Humanos , Sistema Imunitário/imunologia , Inflamação , Doenças Inflamatórias Intestinais/microbiologia , Intestinos/microbiologia , Camundongos , Espondiloartropatias/microbiologia , Espondilite Anquilosante/imunologia , Espondilite Anquilosante/microbiologiaRESUMO
OBJECTIVE: Systemic juvenile idiopathic arthritis-associated lung disease (SJIA-LD) is a life-threatening disease complication. Key questions remain regarding clinical course and optimal treatment approaches. The objectives of the study were to detail management strategies after SJIA-LD detection, characterize overall disease courses, and measure long-term outcomes. METHODS: This was a prospective cohort study. Clinical data were abstracted from the electronic medical record, including current clinical status and changes since diagnosis. Serum biomarkers were determined and correlated with presence of LD. RESULTS: We enrolled 41 patients with SJIA-LD, 85% with at least one episode of macrophage activation syndrome and 41% with adverse reactions to a biologic. Although 93% of patients were alive at last follow-up (median 2.9 years), 37% progressed to requiring chronic oxygen or other ventilator support, and 65% of patients had abnormal overnight oximetry studies, which changed over time. Eighty-four percent of patients carried the HLA-DRB1*15 haplotype, significantly more than patients without LD. Patients with SJIA-LD also showed markedly elevated serum interleukin-18 (IL-18), variable C-X-C motif chemokine ligand 9 (CXCL9), and significantly elevated matrix metalloproteinase 7. Treatment strategies showed variable use of anti-IL-1/6 biologics and addition of other immunomodulatory treatments and lung-directed therapies. We found a broad range of current clinical status independent of time from diagnosis or continued biologic treatment. Multidomain measures of change showed imaging features were the least likely to improve with time. CONCLUSION: Patients with SJIA-LD had highly varied courses, with lower mortality than previously reported but frequent hypoxia and requirement for respiratory support. Treatment strategies were highly varied, highlighting an urgent need for focused clinical trials.
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Artrite Juvenil , Pneumopatias , Síndrome de Ativação Macrofágica , Criança , Humanos , Artrite Juvenil/complicações , Artrite Juvenil/diagnóstico , Artrite Juvenil/tratamento farmacológico , Estudos Prospectivos , Pulmão , Síndrome de Ativação Macrofágica/diagnóstico , Síndrome de Ativação Macrofágica/etiologia , Síndrome de Ativação Macrofágica/terapia , Progressão da DoençaRESUMO
OBJECTIVES: To develop and validate classification criteria for axial disease in youth with juvenile spondyloarthritis (SpA; AxJSpA). METHODS: This international initiative consisted of four phases: 1) Item generation; 2) Item reduction; 3) Criteria development; and 4) Validation of the AxJSpA criteria by an independent team of experts in an internationally representative Validation cohort. RESULTS: These criteria are intended to be used on youth with a physician diagnosis of juvenile SpA and for whom axial disease is suspected. Item generation consisted of a systematic literature review and a free-listing exercise using input from international physicians and collectively resulted in 108 items. After the item reduction exercise and expert panel input, 37 items remained for further consideration. The final AxJSpA criteria domains included: imaging: active inflammation, imaging: structural lesions, pain chronicity, pain pattern, pain location, stiffness, and genetics. The most heavily weighted domains were active inflammation and structural lesions on imaging. Imaging typical of sacroiliitis was deemed necessary, but not sufficient, to classify a youth with AxJSpA. The threshold for classification of AxJSpA was a score of ≥55 (out of 100). When tested in the validation data set, the final criteria had a specificity of 97.5% (95% CI: 91.4-99.7), sensitivity of 64.3% (95% CI: 54.9-73.1) and Area Under the Receiver Operating Characteristic (AUROC) curve of 0.81 (95% CI: 0.76-0.86). CONCLUSIONS: The new AxJSpA classification criteria require an entry criterion, physician diagnosis of juvenile SpA, and include seven weighted domains. The AxJSpA classification criteria are validated and designed to identify participants for research studies.
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OBJECTIVE: TM joint (TMJ) arthritis occurs in up to 80% of children with JIA and can result in substantial deformity. TMJ arthritis can be refractory to systemic immunosuppressive therapy and IA CS injections (IACIs). Multiple studies have shown the benefit of IA infliximab injections (IAIIs) in several different joints, so we used intra-articular infliximab injections (IAIIs) in JIA patients with TMJ arthritis refractory to IACIs. The objective of the study was to test the safety and efficacy of IAII therapy for TMJ arthritis. METHODS: Retrospective chart review was performed for all children with JIA treated at a single centre who received one or more IAIIs. Outcomes assessed were safety of the injections as well as efficacy as evidenced by maximal incisal opening (MIO) and MRI findings. RESULTS: Twenty-four children underwent bilateral IAIIs, all of whom had at least one follow-up visit after the final injection. All 24 tolerated the injections without any adverse events. MIOs were unchanged in patients before and after IAII. Findings of acute synovitis were present in 30/46 (65%) TMJs at baseline, 44/48 (92%) following completion of the IACI and 42/48 (88%) following completion of the IAII; findings of chronic synovitis at the three time points were 12/46 (26%), 29/48 (60%) and 38/48 (79%). Resolution of the arthritis was observed in six TMJs. CONCLUSION: IAII was safe and it reversed the progression of TMJ arthritis in some patients with refractory disease. Future studies will evaluate the efficacy of infliximab vs CS injections as initial therapy for TMJ arthritis.
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Anti-Inflamatórios não Esteroides/uso terapêutico , Anticorpos Monoclonais/uso terapêutico , Artrite Juvenil/tratamento farmacológico , Transtornos da Articulação Temporomandibular/tratamento farmacológico , Adolescente , Anti-Inflamatórios não Esteroides/efeitos adversos , Anticorpos Monoclonais/efeitos adversos , Criança , Pré-Escolar , Feminino , Humanos , Infliximab , Injeções Intra-Articulares , Masculino , Estudos Retrospectivos , Resultado do TratamentoRESUMO
Juvenile idiopathic arthritis is a common chronic childhood disease, with a prevalence of â¼1 per 1000 children. Arthritis can also be a manifestation of other inflammatory conditions, such as inflammatory bowel disease (IBD). Studies suggest a genetic influence in IBD, including mutations in CARD8. CARD8 is a negative regulator of the NLRP3 inflammasome, and mutations in this gene are hypothesized to induce gastrointestinal inflammation. However, few studies have evaluated this association and most have included a limited number of patients. We present a case of a pediatric patient with IBD-associated arthritis and a CARD8 mutation. Our patient is a 7-year-old female who was initially evaluated by rheumatology for right leg pain and an intermittent rash. She had clinically active arthritis on exam and was started on methotrexate with only slight improvement. Additional workup revealed sacroiliitis by imaging, elevated inflammatory markers, no anemia, and a variant of unknown significance in CARD8. Adalimumab was recommended but before medication initiation, our patient's symptoms progressed to worsening joint pain, fatigue, fevers, nausea, vomiting, diarrhea, and hematochezia. Infectious testing was negative. Fecal calprotectin was >8000 µg/g. A colonoscopy revealed IBD most consistent with Crohn's disease. Adalimumab was ultimately added, and she has responded well to combination therapy. This case report highlights the association between CARD8 mutations and IBD, especially in the setting of IBD-associated arthritis.
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Artrite Juvenil , Doença de Crohn , Doenças Inflamatórias Intestinais , Feminino , Humanos , Criança , Artrite Juvenil/complicações , Artrite Juvenil/diagnóstico , Artrite Juvenil/tratamento farmacológico , Adalimumab/genética , Doenças Inflamatórias Intestinais/complicações , Doenças Inflamatórias Intestinais/diagnóstico , Doenças Inflamatórias Intestinais/tratamento farmacológico , Doença de Crohn/complicações , Doença Crônica , Mutação , Proteínas de Neoplasias/genética , Proteínas Adaptadoras de Sinalização CARD/genéticaRESUMO
OBJECTIVE: Pediatric rheumatology faces a looming supply-demand crisis. While strategies have been proposed to address the supply shortfall, investigation into the increased demand for pediatric rheumatic care has been limited. Herein, we analyze new patient visits to a large tertiary care pediatric rheumatology center to identify emerging trends in referrals and areas for potential intervention to meet this increased demand. METHODS: All patients referred to and seen by the University of Alabama at Birmingham Pediatric Rheumatology Division between January 2019 and December 2021 for a new patient evaluation were identified. Patient data was retrospectively abstracted, de-identified, and analyzed to develop trends in referrals and frequency of rheumatic disease, non-rheumatic disease, and specific diagnoses. RESULTS: During the study period, 2638 patients were referred to and seen in by the pediatric rheumatology division. Six hundred and ten patients (23.1%) were diagnosed with rheumatic disease. The most common rheumatic disease was juvenile idiopathic arthritis (JIA) at 45.6%, followed by primary Raynaud phenomenon (7.4%), recurrent fever syndromes (6.9%), vasculitides (6.7%), and inflammatory eye disease (6.2%). Of the 2028 patients (76.9%) diagnosed with a non-rheumatic condition, benign musculoskeletal pain was the most common (61.8%), followed by a combination of somatic conditions (11.6%), and non-inflammatory rash (7.7%). CONCLUSION: In this analysis of new patient referrals to a large pediatric rheumatology center, the majority of patients were diagnosed with a non-rheumatic condition. As a worsening supply-demand gap threatens the field of pediatric rheumatology, increased emphasis should be placed on reducing non-rheumatic disease referrals.