Case of pityriasis rubra pilaris progressed to generalized erythroderma following blockade of interleukin‐17A, but improved after blockade of interleukin‐12/23 p40

Matsuda, Tomoko; Yamazaki, Fumikazu; Ueda‐Hayakawa, Ikuko; Kambe, Naotomo; Okamoto, Hiroyuki

doi:10.1111/1346-8138.14709

Cited by 14 publications

(18 citation statements)

References 11 publications

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“…Secukinumab is the first IL-17 inhibitor approved for the treatment of moderate-to-severe plaque psoriasis, and has now been extensively used for this disorder in adults. 5 However, we reviewed the literature and found that only nine studies included 12 adult patients with PRP (nine cases of type I, two cases of type II, and one case of type III) treated with secukinumab, [6][7][8][9][10][11][12][13][14] as shown in Table 1…”

Section: Discussionmentioning

confidence: 99%

“…There were no any adverse effects and relapse during or after secukinumab treatment. Notably, two cases discontinuing treatment are due to unexpectedly worsening symptoms of skin lesions after the second and fifth injection,10,13 respectively; however, one case not only did not show further worsening of disease but also excellent response when retreatment with secukinumab was performed. 10 Type V PRP often occurs during the first few years of life, has a longer course of disease in general, and is the primary form of familial PRP.…”

mentioning

confidence: 99%

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Secukinumab monotherapy successfully treated severe refractory type V (atypical juvenile) pityriasis rubra pilaris: A case report and literature review

Liang

Xiang

et al. 2020

Dermatologic Therapy

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Pityriasis rubra pilaris (PRP) is a rare heterogeneous group of papulosquamous inflammatory disorders with unknown etiology. PRP is often resistant to many conventional therapies which has made more challenging on treatment. More recently, several studies have shown encouraging clinical results of secukinumab in the treatment of PRP in adult, but no studies have explored its effects in children. We herein report a 7-year-old boy with severe type V PRP responded rapidly to secukinumab monotherapy (150 mg once weekly) when conventional therapies have failed. The patient showed rapid and dramatic improvement of erythema, palmoplantar hyperkeratosis, scaling, and itching within only 5 weeks, with no adverse effects. Secukinumab could be considered as a treatment option for refractory PRP in children, as recently reported in adult. K E Y W O R D S monotherapy, pityriasis rubra pilaris, secukinumab 1 | INTRODUCTION Pityriasis rubra pilaris (PRP) is an uncommon papulosquamous inflammatory disorder characterized by red-orange scaling erythema, follicular keratotic papules, and palmoplantar hyperkeratosis. 1 According to age at onset, clinical features, and prognosis, PRP can been subdivided into five clinical types: adult-onset PRP (classical, type I; atypical, type II) and juvenile-onset PRP (classical, type III; circumscribed, type IV; atypical, type V). 2 Type V PRP is clinically similar to type II PRP except for different onset ages, which are both typified by ichthyosiform features and usually chronic intractable, accounting, respectively, for about 5% of all cases. 1 The therapy in children is sometimes based on research and clinical experience from adults. More recently, secukinumab, an anti-IL-17A monoclonal antibody, has been showed promising results in small series and case reports in adult PRP. In this study, we present a 7-year-old boy with severe atypical juvenile PRP (type V) successfully treated with secukinumab alone who had an unsatisfactory response to first-and second-line treatment, and review all relevant cases reported in the English literature. 2 | CASE REPORT A 7-year-old Chinese boy presented with generalized erythematous plaques and scaly eruption which started on head and trunk during first month after the birth and progressed to involve his entire body. He was diagnosed as atopic dermatitis and generalized pustular psoriasis by other hospitals, and has tried different treatments such as topical corticosteroids, calcineurin inhibitors, systemic methotrexate, and acitretin without any significant benefit. Physical examination revealed confluent ichthyosiform erythematous eruption all over his body with a large amount of scales, both upper eyelids welling and ectropion with incomplete closure, conjunctiva congestion, radial wrinkles around his month, thick palmoplantar Jing-yao Liang and Rui-xian Ye have contributed equally to this study.

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Section: Discussionmentioning

confidence: 99%

mentioning

confidence: 99%

Secukinumab monotherapy successfully treated severe refractory type V (atypical juvenile) pityriasis rubra pilaris: A case report and literature review

Liang

Xiang

et al. 2020

Dermatologic Therapy

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“…As regards anti-IL-12/23 agents, ustekinumab was used in 18.8% of examined PRP cases, showing a high effectiveness (CR of 80.7%) with low relapse rates at the withdrawal. 8,[12][13][14][15] In most cases, it was employed as rescue treatment for recalcitrant forms (88.5%), but first-line use was also reported (11.5%). 8 In addition, ustekinumab was shown to be effective not only in sporadic PRP but also in familial forms and in patients unresponsive to anti-IL17A…”

Section: Discussionmentioning

confidence: 99%

“…and TNF-α. 8,12 Regarding our patients, the man was affected by a moderate form of PRP without systemic symptoms and noteworthy comorbidities for which, in line with commonly shared therapeutic indications, oral retinoid and nb-UVB were prescribed. A successful and long-lasting (even if slow) response was observed at 5 month-follow-up.…”

Section: Discussionmentioning

confidence: 99%

Erythrodermic pityriasis rubra pilaris treatment: Two case reports and literature review

Camela

Miano

Caterino

et al. 2020

Dermatologic Therapy

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Pityriasis rubra pilaris (PRP) is a rare inflammatory dermatosis characterized by hyperkeratotic follicular papules and erythematous-desquamative plaques that tend to progressively evolve into erythroderma. Treatment is challenging given that international guidelines are not available and large-scale trials do not exist. Traditionally, many topical and systemic drugs had been used as consolidated agents; recently, biologicals are gaining increasing importance, promisingly dominating the therapeutic scenario ahead. Herein, we present a case series showing the "past" and the "future" therapeutic approaches to erythrodermic PRP, one case treated with acitretin and nb-UVB phototherapy combination, while the other with ustekinumab, performing also a throughout literature review.

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“…Nonetheless, treatment response is difficult to predict due to the heterogeneity of the disease (73). Both cases with progression using IL-17 inhibition that subsequently improved using ustekinumab (74) as a patient with limited response to ustekinumab who benefited from secukinumab, have been published (73).…”

Section: Psoriasiform Disorders: Pityriasis Rubra Pilarismentioning

confidence: 99%

Learning From Success and Failure: Biologics for Non-approved Skin Diseases

2019

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The impressive potential of biologics has been demonstrated in psoriasis, hidradenitis suppurativa, and urticaria. Numerous biologicals are entering the field for a restricted number of skin disorders. Off-label use of biologics in other recalcitrant skin diseases has increased. Mounting data point to the potential of already existing biologics acting on the IL-17/IL-23 pathway in skin disorders with epidermal hyperkeratosis (e.g., pityriasis rubra pilaris), acneiform inflammation (e.g., hidradenitis suppurativa), and loss of mucosal integrity (e.g., aphthosis). TNF-α blockers are also effective in the latter conditions but seem of particular value in granulomatous (e.g., granuloma annulare) and neutrophilic disorders (e.g., pyoderma gangrenosum). Failure of IL-17 blockade in skin diseases resulting from immune-mediated cell destruction (e.g., alopecia areata and vitiligo) illustrates its limited involvement in Th1-dependent skin immunology. Overall, disappointing results of TNF-α blockers in alopecia areata and vitiligo point to the same conclusion although promising results in toxic epidermal necrolysis suggest TNF-α exerts at least some in vivo Th1-related activities. Acting on both the Th1 and Th17 pathway, ustekinumab has a rather broad potential with interesting results in lupus and alopecia areata. The efficacy of omalizumab in bullous pemphigoid has revealed an IgE-mediated recruitment of eosinophils leading to bullae formation. Reconsidering reimbursement criteria for less common but severe diseases seems appropriate if substantial evidence is available (e.g., pityriasis rubra pilaris). For other disorders, investigator-and industry-initiated randomized clinical trials should be stimulated. They are likely to improve patient outcome and advance our understanding of challenging skin disorders.

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Case of pityriasis rubra pilaris progressed to generalized erythroderma following blockade of interleukin‐17A, but improved after blockade of interleukin‐12/23 p40

Cited by 14 publications

References 11 publications

Secukinumab monotherapy successfully treated severe refractory type V (atypical juvenile) pityriasis rubra pilaris: A case report and literature review

Secukinumab monotherapy successfully treated severe refractory type V (atypical juvenile) pityriasis rubra pilaris: A case report and literature review

Erythrodermic pityriasis rubra pilaris treatment: Two case reports and literature review

Learning From Success and Failure: Biologics for Non-approved Skin Diseases

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