Successful Treatment of Coexistent Acne Fulminans and Severe Hidradenitis Suppurativa with Adalimumab

2021 ◽  
pp. 1-4
Author(s):  
George Kontochristopoulos ◽  
Efthymia Agiasofitou ◽  
Eftychia Platsidaki ◽  
Anastasia Kapsiocha ◽  
Stamatios Gregoriou ◽  
...  
Keyword(s):  
Clinical Improvement ◽  
Hidradenitis Suppurativa ◽  
Low Grade ◽  
Therapeutic Approaches ◽  
Dosing Regimen ◽  
Severe Acne ◽  
Tnf Α ◽  
Effective Option ◽  
Acne Fulminans ◽  
History Of

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.

Efficacy of Adalimumab on Severe Sapho Syndrome Likely Triggered by Isotretinoin

2020 ◽  
Author(s):  
Michele Luzzati ◽  
Cesare Filippeschi ◽  
Teresa Giani ◽  
Sandra Trapani
Keyword(s):  
Hidradenitis Suppurativa ◽  
Severe Depression ◽  
Joint Inflammation ◽  
Sapho Syndrome ◽  
Adolescent Male ◽  
Clinical Feature ◽  
Severe Acne ◽  
Chronic Disorder ◽  
Tnf Α ◽  
Acne Fulminans

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.

scholarly journals Case Report: PsAPSASH syndrome: an alternative phenotype of syndromic hidradenitis suppurativa treated with the IL-17A inhibitor secukinumab

F1000Research ◽  
2021 ◽  
Vol 10 ◽  
pp. 381
Author(s):  
Georgios Nikolakis ◽  
Katja Kreibich ◽  
Aristeidis Vaiopoulos ◽  
Katarzyna Kaleta ◽  
Joud Talas ◽  
...  
Keyword(s):  
Case Report ◽  
Hidradenitis Suppurativa ◽  
Therapeutic Approaches ◽  
Autoinflammatory Syndromes ◽  
Skin Symptoms ◽  
Tnf Α ◽  
Pyogenic Arthritis ◽  
Mixed Phenotype ◽  
Genetic Form

Syndromic hidradenitis suppurativa (HS) is a form of symptom constellations, which differs from the familial and genetic form and comprises predominantly osteoarticular manifestations. Many forms include pyoderma gangrenosum and acne (PASH), pyogenic arthritis (PAPASH), spondyloarthritis (PASS) and psoriatic arthritis (PsAPASH) and are categorized in the autoinflammatory syndromes. Anti-TNF-α and anti-IL-1a blockade are between the therapeutic approaches that improve skin symptoms and prevent permanent osteoarticular damage. This case report refers to the successful treatment of a mixed phenotype of the aforementioned symptoms using the IL-17A inhibitor secukinumab after initial treatment with adalimumab. The therapy improved both cutaneous and reported osteoarticular symptoms. Different approaches for these recalcitrant HS syndromes are essential in order to achieve long-term remission for those patients.

scholarly journals SAPHO syndrome: the supposed trigger by isotretinoin, the efficacy of adalimumab and the specter of depressive disorder: a case report

2020 ◽  
Vol 46 (1) ◽  
Author(s):  
Michele Luzzati ◽  
Gabriele Simonini ◽  
Cesare Filippeschi ◽  
Teresa Giani ◽  
Sandra Trapani
Keyword(s):  
Hidradenitis Suppurativa ◽  
Case Reports ◽  
Interleukin 1 ◽  
Severe Depression ◽  
Sapho Syndrome ◽  
Temporal Association ◽  
Favourable Result ◽  
Necrosis Factor Alpha ◽  
Severe Acne ◽  
Tnf Α

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.

Adalimumab in Treatment-Resistant Hidradenitis Suppurativa following Recurrence after Extensive Affected Area Excision: A Review of Biologics Therapy

2013 ◽  
Vol 17 (4_suppl) ◽  
pp. S23-S32 ◽  
Author(s):  
Michael Samycia ◽  
Alain Brassard
Keyword(s):  
Clinical Judgment ◽  
Marked Improvement ◽  
Hidradenitis Suppurativa ◽  
Biologic Agents ◽  
Low Grade ◽  
Treatment Resistant ◽  
Perianal Region ◽  
Effective Treatment Modality ◽  
History Of ◽  
Traditional Therapies

Background: Hidradenitis suppurativa (HS) is characterized by chronic, suppurative abscesses, sinus tracts, and fistulas affecting the axilla, groin, and perianal region resulting from hyperkeratosis and occlusion of the terminal hair follicle. Objective: This report highlights the use of biologic agents for the treatment of recalcitrant HS. Method: We report on a 48-year-old male with a 15-year history of refractory perianal-inguinal-buttock HS who, despite receiving numerous surgical drainages and traditional medical treatment for HS, still had severe pain. After trialing etanercept and infliximab with methotrexate, the patient had marked improvement with adalimumab. A literature review of biologics therapy was also performed. Results: After trialing many traditional therapies, we found that adalimumab appears to be the most effective treatment modality for our patient. A literature search revealed 53 articles on biologics therapy in HS. These articles are summarized. Discussion: Biologic agents have been shown to have variable results in the treatment of refractory HS. Enough low-grade evidence has been accumulated to make the use of these agents suitable in HS. Until more clinical trials are performed on this topic, physicians should use clinical judgment when treating HS with biologic agents and be cautious by watching for significant adverse effects.

scholarly journals Case Report: PsAPSASH syndrome: an alternative phenotype of syndromic hidradenitis suppurativa treated with the IL-17A inhibitor secukinumab

F1000Research ◽  
2021 ◽  
Vol 10 ◽  
pp. 381
Author(s):  
Georgios Nikolakis ◽  
Katja Kreibich ◽  
Aristeidis Vaiopoulos ◽  
Katarzyna Kaleta ◽  
Joud Talas ◽  
...  
Keyword(s):  
Case Report ◽  
Hidradenitis Suppurativa ◽  
Therapeutic Approaches ◽  
Autoinflammatory Syndromes ◽  
Skin Symptoms ◽  
Tnf Α ◽  
Pyogenic Arthritis ◽  
Mixed Phenotype ◽  
Genetic Form

Syndromic hidradenitis suppurativa (HS) is a form of symptom constellations, which differs from the familial and genetic form and comprises predominantly osteoarticular manifestations. Many forms include pyoderma gangrenosum and acne (PASH), pyogenic arthritis (PAPASH), spondyloarthritis (PASS) and psoriatic arthritis (PsaPASH) and are categorized in the autoinflammatory syndromes. anti-TNF-α and anti-IL-1a blockade are between the therapeutic approaches that improve skin symptoms and prevent permanent osteoarticular damage. This case report refers to the successful treatment of a mixed phenotype of the aforementioned symptoms using the IL-17A inhibitor secukinumab after initial treatment with adalimumab. The therapy improved both cutaneous and reported osteoarticular symptoms. Different approaches for these recalcitrant HS syndromes are essential in order to achieve long-term remission for those patients.

Primary acquired melanosis (PAM) without atypia/WHO low-grade conjunctival melanocytic intraepithelial lesion over areas of oculodermal melanocytosis

2020 ◽  
Vol 13 (10) ◽  
pp. e236741
Author(s):  
Bashar M Bata ◽  
Sachin M Salvi ◽  
Hardeep Singh Mudhar
Keyword(s):  
Bulbar Conjunctiva ◽  
Low Grade ◽  
Primary Acquired Melanosis ◽  
Conjunctival Biopsy ◽  
History Of ◽  
Intraepithelial Lesion ◽  
The Relationship ◽  
Oculodermal Melanocytosis ◽  
Substantia Propria ◽  
New Onset

An elderly white man with a history of left oculodermal melanocytosis presented with new onset brown pigmentation of the left bulbar and inferior tarsal conjunctiva. The bulbar conjunctival pigmentation was at the level of the conjunctival epithelium and was overlying areas of typical slate-grey scleral pigmentation characteristic of oculodermal melanocytosis. Both areas of new pigmentation were biopsied. The bulbar conjunctiva revealed primary acquired melanosis (PAM) without atypia with increased melanin production and the tarsal conjunctival biopsy showed PAM without atypia sine pigmentio overlying areas of substantia propria spindle-shaped heavily pigmented melanocytes of oculodermal melanocytosis. The case report examines the relationship between the epithelial and substantia propria melanocytes and correlates the findings with what is known about this association from the dermatopathology literature.

scholarly journals History of IgA Nephropathy Mouse Models

2021 ◽  
Vol 10 (14) ◽  
pp. 3142
Author(s):  
Batoul Wehbi ◽  
Virginie Pascal ◽  
Lina Zawil ◽  
Michel Cogné ◽  
Jean-Claude Aldigier
Keyword(s):  
Iga Nephropathy ◽  
Small Animal ◽  
Experimental Models ◽  
Murine Models ◽  
Therapeutic Approaches ◽  
Complement Components ◽  
Simple Description ◽  
Primary Glomerulonephritis ◽  
History Of ◽  
Small Animal Models

IgA nephropathy (IgAN) is the most common primary glomerulonephritis in the world. It was first described in 1968 by Jean Berger and Nicole Hinglais as the presence of intercapillary deposits of IgA. Despite this simple description, patients with IgAN may present very broad clinical features ranging from the isolated presence of IgA in the mesangium without clinical or biological manifestations to rapidly progressive kidney failure. These features are associated with a variety of histological lesions, from the discrete thickening of the mesangial matrix to diffuse cell proliferation. Immunofluorescence on IgAN kidney specimens shows the isolated presence of IgA or its inconsistent association with IgG and complement components. This clinical heterogeneity of IgAN clearly echoes its complex and multifactorial pathophysiology in humans, inviting further analyses of its various aspects through the use of experimental models. Small-animal models of IgAN provide the most pertinent strategies for studying the multifactorial aspects of IgAN pathogenesis and progression. Although only primates have the IgA1 subclass, several murine models have been developed in which various aspects of immune responses are deregulated and which are useful in the understanding of IgAN physiopathology as well as in the assessment of IgAN therapeutic approaches. In this manuscript, we review all murine IgAN models developed since 1968 and discuss their remarkable contribution to understanding the disease.

scholarly journals Anti-PD-1-Induced Hidradenitis Suppurativa

2021 ◽  
Vol 8 (1) ◽  
pp. 37-39
Author(s):  
Alexia Maillard ◽  
Damien Pastor ◽  
Rastine Merat
Keyword(s):  
Adverse Events ◽  
Hidradenitis Suppurativa ◽  
Checkpoint Inhibitors ◽  
Latency Period ◽  
The Body ◽  
Skin Reactions ◽  
Zinc Gluconate ◽  
Severe Acne ◽  
Bowel Diseases ◽  
Inflammatory Bowel

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.

scholarly journals Fourteen days of smoking cessation improves muscle fatigue resistance and reverses markers of systemic inflammation

2021 ◽  
Vol 11 (1) ◽  
Author(s):  
Mohammad Z. Darabseh ◽  
Thomas M. Maden-Wilkinson ◽  
George Welbourne ◽  
Rob C. I. Wüst ◽  
Nessar Ahmed ◽  
...  
Keyword(s):  
Oxidative Stress ◽  
Skeletal Muscle ◽  
Smoking Cessation ◽  
Muscle Fatigue ◽  
Fatigue Resistance ◽  
Systemic Inflammation ◽  
Muscle Function ◽  
Low Grade ◽  
Skeletal Muscle Function ◽  
Tnf Α

AbstractCigarette smoking has a negative effect on respiratory and skeletal muscle function and is a risk factor for various chronic diseases. To assess the effects of 14 days of smoking cessation on respiratory and skeletal muscle function, markers of inflammation and oxidative stress in humans. Spirometry, skeletal muscle function, circulating carboxyhaemoglobin levels, advanced glycation end products (AGEs), markers of oxidative stress and serum cytokines were measured in 38 non-smokers, and in 48 cigarette smokers at baseline and after 14 days of smoking cessation. Peak expiratory flow (p = 0.004) and forced expiratory volume in 1 s/forced vital capacity (p = 0.037) were lower in smokers compared to non-smokers but did not change significantly after smoking cessation. Smoking cessation increased skeletal muscle fatigue resistance (p < 0.001). Haemoglobin content, haematocrit, carboxyhaemoglobin, total AGEs, malondialdehyde, TNF-α, IL-2, IL-4, IL-6 and IL-10 (p < 0.05) levels were higher, and total antioxidant status (TAS), IL-12p70 and eosinophil numbers were lower (p < 0.05) in smokers. IL-4, IL-6, IL-10 and IL-12p70 had returned towards levels seen in non-smokers after 14 days smoking cessation (p < 0.05), and IL-2 and TNF-α showed a similar pattern but had not yet fully returned to levels seen in non-smokers. Haemoglobin, haematocrit, eosinophil count, AGEs, MDA and TAS did not significantly change with smoking cessation. Two weeks of smoking cessation was accompanied with an improved muscle fatigue resistance and a reduction in low-grade systemic inflammation in smokers.

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