10.5826/dpc.1302a103
text/xml
2160-9381
2740
en
Alessandra Narcici
https://creativecommons.org/licenses/by-nc/4.0
Dermatology Practical & Conceptual
biologics
Biologics for the Treatment of Severe Acrodermatitis Continua of Hallopeau: Report of Two Cases Successfully Treated with Ixekizumab and Ustekinumab 
Text.Serial.Journal
https://dpcj.org/index.php/dpc
Research Letter
2023-04-29
2022-08-02
2023-04-29
2023-06-05
e2023103
Dermatol Pract Concept
Alessandra Narcici
2023/04/29
10.5826/dpc.1302a103
e2023103
Dermatology Unit, IRCCS Humanitas Research Hospital, Rozzano (MI); Department of Biomedical Sciences, Humanitas University, Pieve Emanuele (MI), Italy
2160-9381
biologics
en
e2023103
Gargiulo L, Pavia G, Ibba L, et al. Generalized Pustular Psoriasis and Plaque Psoriasis Successfully Treated With Ixekizumab. Clin Exp Dermatol Ther. 2022; 7: 183. doi: 10.29011/2575-8268.100183. Published online ahead of print.
Biologics for the Treatment of Severe Acrodermatitis Continua of Hallopeau: Report of Two Cases Successfully Treated with Ixekizumab and Ustekinumab 
https://dpcj.org/index.php/dpc/article/download/2740/2076
           Biologics for the Treatment of Severe
          Acrodermatitis Continua of Hallopeau:
       Report of Two Cases Successfully Treated with
               Ixekizumab and Ustekinumab
                 Luigi Gargiulo1,2, Francesco Toso1,2, Luciano Ibba1,2, Mario Valenti1,2,
                              Antonio Costanzo1,2, Alessandra Narcisi1,2


1 Dermatology Unit, IRCCS Humanitas Research Hospital, Rozzano, Milano, Italy
2 Department of Biomedical Sciences, Humanitas University, Pieve Emanuele, Milano, Italy



Key words: psoriasis, acrodermatitis continua of Hallopeau, ixekizumab, ustekinumab, biologics
Citation: Gargiulo L, Toso F, Ibba L, Valenti M, Costanzo A, Narcisi A. Biologics for the Treatment of Severe Acrodermatitis Continua of
Hallopeau: Report of Two Cases Successfully Treated with Ixekizumab and Ustekinumab. Dermatol Pract Concept. 2023;13(2):e2023103.
DOI: https://doi.org/10.5826/dpc.1302a103
Accepted: September 26, 2023; Published: April 2023
Copyright: ©2023 Gargiulo et al. This is an open-access article distributed under the terms of the Creative Commons Attribution-
NonCommercial License (BY-NC-4.0), https://creativecommons.org/licenses/by-nc/4.0/, which permits unrestricted noncommercial use,
distribution, and reproduction in any medium, provided the original authors and source are credited.
Funding: None.
Competing Interests: Antonio Costanzo has been a consultant and/or speaker for Abb-Vie, Almirall, Amgen, Janssen, Leo Pharma, Eli Lilly,
Galderma, Boehringer, Novartis, Pfizer, Sandoz, and UCB. Alessandra Narcisi has been a consultant and/or speaker for Abb-Vie, Almirall,
Amgen, Janssen, Leo Pharma, Eli Lilly, Boehringer, Novartis, Pfizer and UCB. Mario Valenti has been a consultant and/or speaker for, Leo
Pharma, Eli Lilly, Boehringer and Sanofi. Luigi Gargiulo, Francesco Toso and Luciano Ibba have nothing to disclose.
Authorship: All authors have contributed significantly to this publication.
Corresponding Author: Dr. Francesco Toso, MD, Dermatology Unit, Department of Biomedical Sciences IRCCS Humanitas Research
Hospital, Humanitas University, Rozzano (MI), Italy. Tel: +39 0282244050 Email: francesco.toso@humanitas.it




Introduction                                                             antibody targeting IL-17A, and the second was given usteki-
                                                                         numab, an inhibitor of IL-12/23.
Acrodermatitis continua of Hallopeau (ACH) is a rare form
of pustular psoriasis, which involves the distal portion of the
                                                                         Case Presentation
digits of hands and feet [1]. The exact pathogenesis of this dis-
ease is still not completely understood, and a role of interleukin       A 56-year-old patient presented to our Dermatology de-
­(IL)-36 and IL-17 related cytokines (IL-17A/F, IL23Receptor)            partment with a history of psoriasis and psoriatic arthritis
has been proposed [1]. ACH has a chronic-relapsing course,               (PsA), with a Dermatology Life Quality Index (DLQI) score
and it can lead to severe nail dystrophies [1]. Given the rarity of      of 28. In the past, she was treated with oral acitretin 10 mg,
this condition, no data are available from clinical trials regard-       one capsule daily for 3 months, ineffectively. On clinical
ing the use of biological drugs in these patients [1,2]. We pres-        examination, we observed intense red erythema on the dis-
ent two cases of severe ACH successfully treated with biologics:         tal surface of the five fingers of the right foot, along with
one patient received ixekizumab, a humanized monoclonal                  intense pustulation and severe onychopathy (Figure 1A),



Research Letter | Dermatol Pract Concept. 2023;13(2):e2023103                                                                          1
    A                                                                     B




Figure 1. (A) Severe acrodermatitis continua of Hallopeau affecting the digits of the right foot of a 56-year-old patient, with intense pustula-
tion on erythematous skin along with severe psoriatic onychopathy. (B) Clinical appearance of the left foot.




and moderate erythema and onychopathy on the left foot                    Conclusions
­(Figure 1B). A skin biopsy was not performed as the clinical
picture was strongly suggestive for ACH. Given the severity               The treatment of ACH is challenging, due to the lack of
of the clinical picture, as screening exams were all in the nor-          randomized clinical trials on biological drugs, given the rar-
mal ranges, we prescribed therapy with ixekizumab 80 mg,                  ity of this condition [2]. There are only a few case reports
two injections at baseline followed by one injection every                on the efficacy of ixekizumab in the treatment of pustular
two weeks until week 12, and then every four weeks. At                    variants of psoriasis [3]. In our first case, we prescribed
week 36, the patient came back to our department showing                  ­ixekizumab for several reason: the rapid onset of action, the
complete skin clearance, with the persistence of only slight              efficacy on PsA and our favorable experience on this drug
onychopathy (Figure 2, A and B) and a DLQI of 1.                          on difficult-to-treat areas [4,5]. On the other hand, more
    The second patient is a 32-year-old female, who pre-                  data are available on ustekinumab in patients affected by
sented to our Institute in 2016 with a history of flares of               ACH [6]. In a multicenter retrospective study, ustekinumab
ACH (with fever and elevated neutrophils count), previously               showed improvement in 75.0% of patients [6]. In our case,
ineffectively treated with oral cyclosporine. On physical                 we decided to prescribe ustekinumab because at the time
examination, several pustules on erythematous skin were                   it was the most recent and most effective biological drug
observed on the distal surface of the fingers of both hands.              approved for psoriasis.
Subsequently, after screening exams returned all in the nor-                  Further studies, with longitudinal design and larger
mal ranges, we prescribed ustekinumab 45 mg, 1 injection at               ­cohorts of patients, are needed to establish the exact role
weeks 0, 4, and then every 12 weeks. At week 40, the patient              of biologics, including ustekinumab and ixekizumab, in the
showed complete skin clearance. She is still on treatment to              management of ACH.
date, without any relapse of the disease.




2                                                                             Research Letter | Dermatol Pract Concept. 2023;13(2):e2023103
Figure 2. (A,B) Clinical picture at week 36, showing resolution of the cutaneous lesions. Only slight onychopathy is observed.




References                                                               4. Narcisi A, Valenti M, Cortese A, et al. Anti-IL17 and anti-IL23
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   ­Gooderham MJ. The Use of Biologic Therapy in the Treatment of          ­Venereol. 2020;34(10):2330-2338. DOI:10.1111/jdv.16265 PMID:
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    PMID: 30938189.                                                         and Plaque Psoriasis Successfully Treated With I­xekizumab.
3. Miller AC, Holland TE, Cohen DJ. Treatment of acrodermati-               Clin Exp Dermatol Ther. 2022;7:183. DOI: 10.29011/2575
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Research Letter | Dermatol Pract Concept. 2023;13(2):e2023103                                                                               3
