Efficacy of Adalimumab on Severe Sapho Syndrome Likely Triggered by Isotretinoin

2020 ◽  
Author(s):  
Michele Luzzati ◽  
Cesare Filippeschi ◽  
Teresa Giani ◽  
Sandra Trapani

Abstract Background SAPHO (synovitis, acne, pustolosis, hyperostosis and osteitis) syndrome is a rare autoinflammatory chronic disorder, presenting with non-infectious inflammatory osteitis, sterile joint inflammation and skin manifestations including palmoplantar pustolosis and severe acne. It could be often misdiagnosed for its heterogeneous clinical presentation. Isotretinoin, which is commonly used for severe acne treatment, has been rarely described as possible trigger of osteo-articular manifestations, in particular sacroiliitis. Various biological treatment have been proposed in refractory patients.Case presentation The case of an adolescent male, affected by acne fulminans and depression, who presented with sacroiliitis after a 10-week treatment with isotretinoin is presented and discussed. After SAPHO diagnosis, the boy started NSAIDs therapy but the onset of bilateral gluteal hidradenitis suppurativa required the switch to a tumour necrosis factor (TNF)-α antagonist (adalimumab). Despite specific therapy with sertraline, the patient continued to complain severe depression, this symptom has been widely reported in patient with SAPHO.Conclusions Our case strengthens the hypothesis that isotretinoin could be a trigger of musculoskeletal involvement in SAPHO. The occurrence of hidradenitis suppurativa as additional clinical feature of SAPHO, already described in literature, supported the TNF-α blocker’s commencement in our patient. Furthermore, the good outcome of our case confirms the efficacy of ADA treatment in obtaining persistent clinical remission of cutaneous and osteoarticular symptoms in SAPHO syndrome.

2020 ◽  
Vol 46 (1) ◽  
Author(s):  
Michele Luzzati ◽  
Gabriele Simonini ◽  
Cesare Filippeschi ◽  
Teresa Giani ◽  
Sandra Trapani

Abstract Background SAPHO (synovitis, acne, pustolosis, hyperostosis and osteitis) syndrome is a rare autoinflammatory chronic disorder, presenting with non-infectious osteitis, sterile joint inflammation and skin manifestations including palmoplantar pustolosis and severe acne. It could be often misdiagnosed for its heterogeneous clinical presentation. Treatment is challenging and, due to the rarity of this syndrome, no randomized controlled clinical trials have been conducted. Empirical treatments, including non-steroidal anti-inflammatory drugs (NSAIDs), corticosteroids, antibiotics and bisphosphonates and disease-modifying anti-rheumatic drugs (DMARDs) could be quite effective. Anti-tumor necrosis factor-alpha (anti-TNF-α) agents and interleukin-1 (IL-1) antagonists have shown promising results in refractory patients. Isotretinoin, commonly used for severe acne, has been rarely described as possible trigger of osteo-articular manifestations, in particular sacroiliitis. Case presentation The case of a boy, affected by acne fulminans and depression, who presented with sacroiliitis after a 10-week treatment with isotretinoin is presented. After SAPHO diagnosis, NSAIDs therapy was started but the onset of bilateral gluteal hidradenitis suppurativa required the switch to a TNF-α antagonist (Adalimumab) with the achievement of a good control of the disease. Despite specific therapy with sertraline, the patient continued to complains severe depression. Conclusions Our case reports a temporal association between the onset of osteo-articular symptoms and the introduction of isotretinoin, as previously described. However, this timeline is not sufficient to establish a causal role of this drug into the pathogenesis of sacroiliitis. At this regard, further studies are required. The occurrence of hidradenitis suppurativa during SAPHO course supported the introduction of TNF-α blockers with a favourable result, as reported in a few cases in literature. The association between SAPHO syndrome and depressive mood disorders is already reported. Our patient experienced severe depression whose trend seems to be independent from the course of the main disease. Currently, it is not clarified if depression could be considered reactive to the underling disease or if it forms an integral part of the autoinflammatory disorder.


2021 ◽  
pp. 1-4
Author(s):  
George Kontochristopoulos ◽  
Efthymia Agiasofitou ◽  
Eftychia Platsidaki ◽  
Anastasia Kapsiocha ◽  
Stamatios Gregoriou ◽  
...  

The coexistence of hidradenitis suppurativa (HS) and acne fulminans (AF) has only recently been reported in the literature. We present a case of a 17-year-old man who presented with a 2 years history of severe acne and HS. He was initially started on oral clindamycin and rifampicin for 3 months with no clinical improvement. Acne lesions became worse with the presence of nodules and necrotic ulcers, while weight loss, low-grade fever, back and knee pain, and psychological distress were noted. We prescribed adalimumab in its standard dosing regimen. Remission of AF was achieved in 3 months, whereas adalimumab has not been as effective in treating the HS lesions. Its dosage was increased to 80 mg weekly and more than 80% clinical improvement of HS lesions was obtained in 2 months. The patient maintained on this dosage till this day and efficacy is sustained. TNF-α inhibitors are considered an effective option in the treatment of HS, while it has been also suggested as a treatment option in AF. Our patient was successfully treated with adalimumab. Since the coexistence of HS and AF has a devastating emotional effect on the patient, there is an urgent need to implement therapeutic approaches.


2020 ◽  
Vol 16 ◽  
Author(s):  
Dalia S. Saif ◽  
Nagwa N. Hegazy ◽  
Enas S. Zahran

Background: Among rheumatoid arthritis patients (RA), general disease activity is well regulated by diseasemodifying anti-rheumatic medications (DMARDS), but sometimes local inflammation still persists among a few joints. Adjuvant modern molecular interventions as Platelet Rich Plasma (PRP) with a suggested down regulating effect on inflammatory mediators has a proven effect in management of RA. We aim to evaluate the therapeutic effect of intra-articular PRP versus steroid in RA patients and their impact on inflammatory cytokines IL1B , TNF α, local joint inflammation, disease activity and quality of life (QL). Methods: Open labeled parallel randomized control clinical trial was carried out on 60 RA patients randomly divided into 2 groups, Group 1: included 30 patients received 3 intra-articular injections of PRP at monthly interval, Group 2: included 30 patients received single intra-articular injection of steroid. They were subjected to clinical, laboratory, serum IL1B and TNF α assessment at baseline and at 3, 6 months post injection. Results: Patients of both groups showed improvements in their scores of evaluating tools at 3months post injection and this improvement was persistent in the PRP group up to 6 months post injection while it was continued only for 3 months in the steroid group. Conclusions: PRP is a safe, effective and useful therapy in treating RA patients who had insufficient response and persistent pain and inflammation in just one or two joints through its down regulating effect on inflammatory cytokines IL1B, TNF α with subsequent improvement of local joint inflammation, disease activity and QL.


2021 ◽  
Vol 22 (3) ◽  
pp. 1022
Author(s):  
Tatyana P. Makalish ◽  
Ilya O. Golovkin ◽  
Volodymyr V. Oberemok ◽  
Kateryna V. Laikova ◽  
Zenure Z. Temirova ◽  
...  

The urgency of the search for inexpensive and effective drugs with localized action for the treatment of rheumatoid arthritis continues unabated. In this study, for the first time we investigated the Cytos-11 antisense oligonucleotide suppression of TNF-α gene expression in a rat model of rheumatoid arthritis induced by complete Freund’s adjuvant. Cytos-11 has been shown to effectively reduce peripheral blood concentrations of TNF-α, reduce joint inflammation, and reduce pannus development. The results achieved following treatment with the antisense oligonucleotide Cytos-11 were similar to those of adalimumab (Humira®); they also compared favorably with those results, which provides evidence of the promise of drugs based on antisense technologies in the treatment of this disease.


2021 ◽  
Vol 8 (1) ◽  
pp. 37-39
Author(s):  
Alexia Maillard ◽  
Damien Pastor ◽  
Rastine Merat

Mucocutaneous adverse events are commonly observed under immune checkpoint inhibitors (ICIs) therapy. Here, we report the case of a 43-year-old male patient with a stage IIIC melanoma disease who developed hidradenitis suppurativa (HS) three months after the beginning of an anti-PD-1 (nivolumab) adjuvant therapy. The patient had no comorbidities other than obesity and severe acne during adolescence. After an unsuccessful course of lymecycline while he was still treated with nivolumab, he gradually improved under zinc gluconate therapy and, more importantly, after nivolumab cessation. HS is a recurrent follicular inflammatory disease in the apocrine gland-bearing areas of the body often associated with obesity, metabolic syndrome, tobacco smoking, inflammatory bowel diseases, psoriasis, and arthritis. In our patient, the latency period between drug initiation and onset of HS symptoms and the improvement after immunotherapy discontinuation, argued strongly in favor of an anti-PD-1-induced HS. Anti-PD-1 therapies often trigger T cells-mediated adverse events that mimic Th17-mediated inflammatory and neutrophilic diseases. We suggest that HS, as other pustular skin reactions and ICIs-induced neutrophilic colitis, can be part of the anti-PD-1 mucocutaneous adverse event spectrum.


2018 ◽  
Vol 38 (1) ◽  
pp. 53-61 ◽  
Author(s):  
Chen Li ◽  
Xia Wu ◽  
Yihan Cao ◽  
Yueping Zeng ◽  
Weihong Zhang ◽  
...  
Keyword(s):  

2020 ◽  
Vol 12 ◽  
pp. 1759720X2091286
Author(s):  
Shuang Liu ◽  
Mingwei Tang ◽  
Yihan Cao ◽  
Chen Li

Synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome is a spectrum of heterogeneous diseases characterized by osteoarticular and dermatological manifestations. Osteitis and hyperostosis are core clinical manifestations in SAPHO syndrome, typically affecting multiple areas and possibly progressing to irreversible osteoarticular damage. Most patients with SAPHO have cutaneous involvement, mainly manifested as palmoplantar pustulosis and severe acne. Systemic manifestations are uncommon but occasionally reported. Epidemiological studies suggest the annual prevalence of SAPHO syndrome varies from 0.00144 in 100,000 in Japanese individuals to fewer than 1 in 10,000 in White individuals. The precise etiopathogenesis of SAPHO remains unclear, but it is generally considered an autoinflammatory syndrome that may be related to various etiologies, such as immune dysfunction, infection and genetic predisposition. Owing to the relapsing–remitting disease course, the goal of management is to improve clinical symptoms and prevent disease progression. Various treatments, including nonsteroidal anti-inflammatory drugs, conventional disease-modifying antirheumatic drugs, bisphosphonates, biologics, and antibiotics, are promising options for alleviating the disease.


2019 ◽  
Vol 3 (1) ◽  
Author(s):  
Kevin Phan ◽  
Olivia Charlton ◽  
Saxon D. Smith

Abstract Background Hidradenitis suppurativa (HS) is a chronic inflammatory skin disorder which involves painful nodules and draining abscesses in flexural areas. Acne vulgaris and its more severe variants including acne conglobata and acne fulminans are also disorders involving the follicular unit. Given that follicular obstruction, dilatation and inflammation feature in both HS and acne vulgaris/conglobata, it has been suggested that HS is associated with acne vulgaris/conglobata. Methods The present systematic review and meta-analysis was performed according to recommended PRISMA guidelines. All eligible case-control studies comparing patients with HS vs non-HS were included in the present review. All studies must have included either the proportion of patients with acne vulgaris/conglobata in each group, or the summary effect size for association between HS and acne vulgaris/conglobata. The odds ratio (OR) was used as a summary statistic. Results From pooled unadjusted meta-analysis, we found a significantly higher proportion of patients with acne vulgaris/conglobata in HS cases compared to controls (OR 3.44, 95% CI 1.95–6.07, P < 0.0001, I2 = 100%). Pooled meta-analysis was also performed with adjusted effect sizes. This demonstrated that HS was significantly associated with acne vulgaris/conglobata after adjustment for potential confounders (OR 3.44, 95% CI 2.43–4.87, P < 0.00001, I2 = 99%). Conclusions In summary, a significant association was found between HS and acne vulgaris/conglobata. This has implications in terms of understanding the burden of disease on patient quality of life as well as consideration of optimal management strategies to target both disorders. Physicians taking care of patients with HS should be aware of this association.


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