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CASE REPORT
Year : 2022  |  Volume : 23  |  Issue : 2  |  Page : 170-171

Early-onset, severe prurigo nodularis in a young child with atopic dermatitis treated successfully with dupilumab


Department of Paediatrics, Paediatric Dermatology Unit, Women and Children Hospital Kuala Lumpur, Kuala Lumpur, Malaysia

Date of Submission14-Dec-2021
Date of Decision27-Jan-2022
Date of Acceptance28-Jan-2022
Date of Web Publication30-Mar-2022

Correspondence Address:
Dr. Henrietta Albela
Department of Paediatrics, Paediatric Dermatology Unit, Women and Children Hospital Kuala Lumpur, Jalan Pahang, 50586, Kuala Lumpur
Malaysia
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijpd.ijpd_175_21

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  Abstract 


Prurigo nodularis is an extremely pruritic skin condition as a result of chronic and vicious cycle of itching and scratching. It is rare in children. We report a case of a 4-year-old child with a background of poorly controlled atopic dermatitis with early-onset, severe prurigo nodularis, who was treated successfully with off-label use of dupilumab injection. There was a significant reduction in itchiness by week 2 of treatment, and achieved 80% reduction in Eczema Area and Severity Index with almost complete resolution of prurigo nodules by week 8. Dupilumab can be considered a treatment option for young pediatric patients with early-onset prurigo nodularis.

Keywords: Atopic dermatitis, biologics, pediatric dermatology


How to cite this article:
Albela H, Leong KF. Early-onset, severe prurigo nodularis in a young child with atopic dermatitis treated successfully with dupilumab. Indian J Paediatr Dermatol 2022;23:170-1

How to cite this URL:
Albela H, Leong KF. Early-onset, severe prurigo nodularis in a young child with atopic dermatitis treated successfully with dupilumab. Indian J Paediatr Dermatol [serial online] 2022 [cited 2022 May 17];23:170-1. Available from: https://www.ijpd.in/text.asp?2022/23/2/170/341468




  Introduction Top


Prurigo nodularis is an extremely pruritic skin condition as a result of chronic, vicious cycle of itching and scratching. Treatment for prurigo nodularis can be challenging especially in the paediatric population due to lack of standardized treatment. Recent reports have demonstrated efficacy of dupilumab in treating prurigo nodularis.


  Case Report Top


A 4-year-old Malay boy presented to us with atopic dermatitis and prurigo nodularis. His symptoms started since he was 6 months old. He was poorly controlled due to noncompliance and frequently missed clinic visits due to his poor social background. He developed highly pruritic nodular lesions on his trunk and limbs since the age of 2 years. On examination, he had multiple erythematous and excoriated plaques and nodules over his limbs and back with generalized dry skin and flexural lichenification [Figure 1] and [Figure 2]. Baseline Eczema Area and Severity Index (EASI) score was 33.

First-line treatment included ultra-potent topical steroids and wet wrap therapy for 3 months, without significant sustained improvement. Parents were not keen on oral immunosupressants, fearing the potential side effects and the need for frequent blood examinations, while phototherapy was not feasible due to logistical issues. Dupilumab was then offered as a treatment option, and parents consented to its off-label usage. Dose-adjusted syringe of 100 mg (7 mg/kg) of dupilumab, prepared from prefilled 300 mg syringe, was administered subcutaneously every 4 weeks. There was a significant reduction in pruritus by week 2 and achieved 80% reduction in EASI score (from baseline of 33–14.2 at week 4 and 5.3 at week 8) with almost complete resolution of prurigo nodules [Figure 1] and [Figure 2]. The child has completed 6 months of dupilumab therapy till date and is currently on the monthly injections, without any adverse events reported so far.
Figure 1: Prurigo nodules on patient's back, before and after treatment with dupilumab

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Figure 2: Prurigo nodules on patient's lower limbs, before and after treatment with dupilumab

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  Discussion Top


Prurigo nodularis is an extremely pruritic skin condition as a result of chronic and vicious cycle of itching and scratching. Nearly half of patients with prurigo nodularis have either atopic predisposition or atopic dermatitis.[1] It is rare in children, with a reported lower prevalence rate compared to the adult population (21.6 compared to 72, respectively, per 100,000).[2]

Treatment for prurigo nodularis can be challenging, especially in the pediatric population due to the lack of standardized treatment. Treatment options for this difficult-to-treat condition include topical agents such corticosteroids, calcineurin inhibitors, calcipotriol, and capsaicin; phototherapy, thalidomide, cyclosporine, methotrexate, and certain antidepressants.[3]

Dupilumab is a monoclonal antibody which blocks Interleukin (IL)-4 receptor alpha subunit activity, causing IL-4 and IL-13 inhibitory activity. It has been successfully used in the treatment of atopic dermatitis, asthma, and chronic rhinosinusitis with nasal polyposis. In 2020, dupilumab was approved was the U.S. Food and Drug Administration (FDA) for children aged 6 to 11 years with moderate-to-severe atopic dermatitis, with a recommended dosing regimen of either every two weeks (200 mg) or four weeks (300 mg), based on weight, following an initial loading dose. Recent reports have also demonstrated the efficacy of dupilumab in treating prurigo nodularis,[4],[5] with ongoing pivotal phase 3 clinical trial showing positive results to date.[6] Given that the most unbearable symptom of prurigo nodularis is pruritus, aim of treatment was to reduce itchiness significantly. Nearly half of adult patients with prurigo nodularis treated with dupilumab have complete resolution of pruritus, although the time taken to achieve this effect is at least 2 months from the start of treatment.[5]

In our patient, there is significant response in pruritus by 2 weeks of treatment and visible improvement in the nodular lesions after just 3 doses of dupilumab by week 8 of treatment. In conclusion, dupilumab can be considered a treatment option for young patients with early-onset prurigo nodules. Future research is needed to analyze the efficacy and safety of dupilumab for pediatric patients with prurigo nodules.

Declaration of consent

The authors certify that they have obtained all appropriate consent forms, duly signed by the parent(s) of the patient. In the form the parent(s) has/have given his/her/their consent for the images and other clinical information of their child to be reported in the journal. The parents understand that the names and initials of their child will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Acknowledgment

The authors would like to thank the Director General of Health, Malaysia, for permission to publish this article.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Iking A, Grundmann S, Chatzigeorgakidis E, Phan NQ, Klein D, Ständer S. Prurigo as a symptom of atopic and non-atopic diseases: Aetiological survey in a consecutive cohort of 108 patients. J Eur Acad Dermatol Venereol 2013;27:550-7.  Back to cited text no. 1
    
2.
Huang AH, Roh YS, Sutaria N, et al. Real-world disease burden and comorbidities of pediatric prurigo nodularis. J Am Acad Dermatol. 2022;86:5-7  Back to cited text no. 2
    
3.
Qureshi AA, Abate LE, Yosipovitch G, Friedman AJ. A systematic review of evidence-based treatments for prurigo nodularis. J Am Acad Dermatol 2019;80:756-64.  Back to cited text no. 3
    
4.
Napolitano M, Fabbrocini G, Scalvenzi M, Nisticò SP, Dastoli S, Patruno C. Effectiveness of dupilumab for the treatment of generalized prurigo nodularis phenotype of adult atopic dermatitis. Dermatitis 2020;31:81-4.  Back to cited text no. 4
    
5.
Husein-ElAhmed H, Steinhoff M. Dupilumab in prurigo nodularis: A systematic review of current evidence and analysis of predictive factors to response. J Dermatolog Treat 2020. p. 1-7.  Back to cited text no. 5
    
6.
Sanofi. Dupixent® (Dupilumab) is the First Biologic to Significantly Reduce Itch and Skin Lesions in Phase 3 Trial for Prurigo Nodularis, Demonstrating the Role of Type 2 Inflammation in this Disease; October 22, 2021. Available from: https://www.sanofi.com/en/media-room/press-releases/2021/2021-10-22-07-00-00-2318876. [Last accessed on 2021 Dec 01].  Back to cited text no. 6
    


    Figures

  [Figure 1], [Figure 2]



 

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