Successful treatment of a child's pityriasis rubra pilaris (<scp>PRP</scp>) with ustekinumab and acitretin

Medek, Katharina; Selhofer, Sylvia; Matthias, Buchner; Mrowietz, Ulrich; Laimer, Martin

doi:10.1111/pde.14994

Cited by 7 publications

(5 citation statements)

References 5 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…A recent systematic review identified that biologic treatment was successful in 81.1% of the patients when used as monotherapy or combination therapy with existing treatments [ 7 , 11 ]. Recent case reports demonstrated the effectiveness of the combination of ustekinumab, an IL-12 and IL-23 inhibitor, with acitretin, in clearing keratoderma within the classic adult, pediatric, and refractory PRP [ 5 , 10 ]. Another systematic review demonstrated that infliximab was associated with a more rapid response than etanercept.…”

Section: Discussionmentioning

confidence: 99%

“…Another systematic review demonstrated that infliximab was associated with a more rapid response than etanercept. On the other hand, ustekinumab monotherapy, or in combination with other regimens, has been proven to give long-term control in treating adult and juvenile PRP [ 5 , 10 ]. A few case reports have also shown clearance of palmoplantar keratoderma in types 1 and 2 PRP within two weeks of using secukinumab [ 9 ].…”

Section: Discussionmentioning

confidence: 99%

“…The pathophysiology remains unclear, nevertheless, studies reported that there may be an association with CARD 14 (caspase recruitment domain-containing protein 14) mutations, activating the Th17 pathway, as well as infections, autoimmune, and neoplastic triggers [ 4 , 5 ]. Wang et al highlighted the possibility of vitamin A's role in the pathogenesis as discussed later [ 3 ].…”

Section: Introductionmentioning

confidence: 99%

See 2 more Smart Citations

Misdiagnosed Pityriasis Rubra Pilaris Successfully Managed With Isotretinoin: A Case Series

Khalifa¹,

Alsabbagh²,

Raees³

et al. 2023

Cureus

View full text Add to dashboard Cite

Being a rare inflammatory, hyperproliferative dermatosis, diagnosing pityriasis rubra pilaris (PRP) can be a challenge to many clinicians. Our case reports aim to demonstrate that PRP is frequently diagnosed and managed as atopic dermatitis (AD) and that distinguishing features on dermoscopy, and biopsy can help diagnose this rare disease. The study also aims to show that PRP can be successfully treated with Isotretinoin. Our case series aims to describe two cases of PRP, initially diagnosed as AD and managed with topical corticosteroids. Being the first case series in the literature to describe the incidences of diagnosing PRP as AD portrays the significance of utilizing dermoscopy and biopsy as tools to confirm this diagnosis for appropriate management. Although PRP is a rare diagnosis, dermoscopy and biopsy can help confirm the disease. Management with isotretinoin will most likely have successful outcomes in those patients.

show abstract

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

See 1 more Smart Citation

Misdiagnosed Pityriasis Rubra Pilaris Successfully Managed With Isotretinoin: A Case Series

Khalifa¹,

Alsabbagh²,

Raees³

et al. 2023

Cureus

View full text Add to dashboard Cite

show abstract

“…All of these patients responded positively to this treatment. One patient, a 17-year-old boy previously treated with oral corticosteroids, phototherapy, acitretin and cyclosporine, did not respond to etanercept therapy but achieved disease remission with adalimumab [ 30 – 39 ]. Here, we have demonstrated a good efficacy and safety profile of risankizumab among pediatric patients with PRP.…”

Section: Discussionmentioning

confidence: 99%

Successful Treatment of Pityriasis Rubra Pilaris with Risankizumab in Children

Kołt-Kamińska,

Osińska,

Kaznowska

et al. 2023

Dermatol Ther (Heidelb)

View full text Add to dashboard Cite

Pityriasis rubra pilaris (PRP) is a rare inflammatory skin disease that affects men and women of all ages, including children. PRP is characterized by follicular and palmoplantar hyperkeratosis and salmon-colored scaling plaques. The exact pathogenesis of PRP is still unknown; most PRP cases are acquired, but some cases may show a familial occurrence, often associated with a mutation in the CARD14 gene. Due to the rarity of PRP, treatment recommendations are based mainly on case reports, small case series and expert opinions and still represent a major therapeutic challenge, especially in children. A growing number of reports on treatment with biologicals, particularly anti-TNFα, has been published. However, an involvement of the IL-23/Th17 axis in both psoriasis and PRP pathogenesis may suggest that this pathway may be a potential therapeutic target. Here, we present three pediatric patients with PRP successfully treated with risankizumab. All patients exhibited a severe course of PRP and lack of response to conventional therapy, including acitretin, cyclosporine and phototherapy. A single dose of 75 mg risankizumab resulted in almost complete clearance of skin lesions in case 1 and 2 at week 4. In patient 3, clear skin was achieved after the second administration of risankizumab (150 mg). All patients continue the treatment with risankizumab, and no adverse effects have been reported up to the present time. Our study demonstrates that risankizumab, an IL-23 blocker, shows good efficacy and safety among pediatric patients with PRP.

show abstract

“…Interleukin-17 inhibitors and ustekinumab, a combined interleukin-12 and interleukin-23 inhibitor, have shown promising results in several case reports. [1][2][3] In particular, ustekinumab is emerging as a potential first-line treatment for the subgroup of patients with CARD14 variants. 4 CARD14 variants are seen as a predisposing factor to autoinflammatory keratinization, as seen in juvenile PRP.…”

mentioning

confidence: 99%

Successful treatment of CARD14‐negative juvenile Pityriasis rubra pilaris with Ustekinumab

Reitmajer,

Forchhammer,

Silber

2023

Acad Dermatol Venereol

View full text Add to dashboard Cite

Dear Editor, Juvenile Pityriasis rubra pilaris (PRP) is an orphan disease without established efficient treatment options. Interleukin-17 inhibitors and ustekinumab, a combined interleukin-12 and interleukin-23 inhibitor, have shown promising results in several case reports. [1][2][3] In particular, ustekinumab is emerging as a potential first-line treatment for the subgroup of patients with CARD14 variants. 4 CARD14 variants are seen as a predisposing factor to autoinflammatory keratinization, as seen in juvenile PRP. [4][5][6] Here, we report the successful treatment with ustekinumab in a juvenile PRP patient without CARD14 mutation, proposing to overthink the potential range of indication.

show abstract

Successful treatment of a child's pityriasis rubra pilaris (PRP) with ustekinumab and acitretin

Cited by 7 publications

References 5 publications

Misdiagnosed Pityriasis Rubra Pilaris Successfully Managed With Isotretinoin: A Case Series

Misdiagnosed Pityriasis Rubra Pilaris Successfully Managed With Isotretinoin: A Case Series

Successful Treatment of Pityriasis Rubra Pilaris with Risankizumab in Children

Successful treatment of CARD14‐negative juvenile Pityriasis rubra pilaris with Ustekinumab

Contact Info

Product

Resources

About