# Severe, Resistant Generalized Pustular Psoriasis-Associated Inflammatory Syndrome Successfully Treated With Adalimumab: A Case Report

**Authors:** Evangelos Bourousis, Marina Triantafyllia Kotzamani, Despoina N Maritsi

PMC · DOI: 10.7759/cureus.98830 · Cureus · 2025-12-09

## TL;DR

An 8-year-old girl with severe, treatment-resistant pustular psoriasis was successfully treated with adalimumab and methotrexate.

## Contribution

This case demonstrates successful treatment of pediatric GPP with adalimumab and methotrexate after prior therapies failed.

## Key findings

- Genetic testing confirmed mutations in IL36RN and CARD14 in the patient.
- Adalimumab in combination with methotrexate led to sustained remission.
- Biologic agents may be effective in managing severe pediatric GPP.

## Abstract

Generalized pustular psoriasis (GPP) is a rare and severe variant of psoriasis, especially in pediatric patients, characterized by recurrent episodes of fever, malaise, and sterile pustules. We present the case of an eight-year-old girl with genetically confirmed GPP, carrying heterozygous mutations in IL36RN and CARD14, successfully treated with adalimumab in combination with methotrexate. Despite previous therapies, sustained remission was achieved only with this regimen. This case highlights the importance of genetic testing and the potential role of biologic agents in the management of pediatric GPP.

## Linked entities

- **Genes:** IL36RN (interleukin 36 receptor antagonist) [NCBI Gene 26525], CARD14 (caspase recruitment domain family member 14) [NCBI Gene 79092]
- **Diseases:** psoriasis (MONDO:0005083), generalized pustular psoriasis (MONDO:0100491)

## Full-text entities

- **Genes:** CARD14 (caspase recruitment domain family member 14) [NCBI Gene 79092] {aka BIMP2, CARMA2, PRP, PSORS2, PSS1}, IL36RN (interleukin 36 receptor antagonist) [NCBI Gene 26525] {aka FIL1, FIL1(DELTA), FIL1D, IL-36Ra, IL1F5, IL1HY1}
- **Diseases:** GPP (MESH:D011565), fever (MESH:D005334)
- **Chemicals:** Adalimumab (MESH:D000068879), methotrexate (MESH:D008727)
- **Species:** Homo sapiens (human, species) [taxon 9606]

## Full text

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## Figures

2 figures with captions in the complete paper: https://tomesphere.com/paper/PMC12782645/full.md

## References

10 references — full list in the complete paper: https://tomesphere.com/paper/PMC12782645/full.md

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Source: https://tomesphere.com/paper/PMC12782645