Two cases of generalized pustular psoriasis complicated by IgG4-related disease

Miyazawa, Hajime; Fujita, Yasuyuki; Iwata, Hiroaki; Ishikawa, Yasunobu; Nishio, S.; Ishijima, Kan; Shinmei, Yasuhiro; Takeichi, Takuya; Goto, Kazuya; Oi, R.; Akiyama, Masashi; Shimizu, Hiroshi

doi:10.1111/bjd.16712

Cited by 3 publications

(3 citation statements)

References 7 publications

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“…T helper 17 skewing may explain the co-occurrence of pustular psoriasis and IgG4-RD. 31 Articles investigating the association between ACH and IgG4-RD are lacking.…”

Section: Discussionmentioning

confidence: 99%

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Deficiency of interleukin‐36 receptor antagonist (DITRA): An analysis of 58 Chinese patients in a tertiary hospital in Taiwan

Hsieh

Huang

et al. 2023

Experimental Dermatology

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DITRA, acronym for deficiency of interleukin‐36 receptor antagonist (IL36RN), leads to unopposed pro‐inflammatory signalling which typically manifests as pustular psoriasis. In Asian patients, c.115 + 6 T > C mutation is the most common and important single‐nucleotide variant in DITRA. We present the largest case series consisting of 58 DITRA patients carrying heterozygous or homozygous c.115 + 6 T > C mutation. The mean age of onset (±SD) was 20.74 (±20.86), and the median age of onset was 13 years old. Twelve patients (20.7%) had disease onset before the age of two. Twenty‐two patients (37.9%) had disease onset between the ages of 2–18. Main clinical phenotype was generalized pustular psoriasis (GPP) with systemic symptoms (33 patients, 56.9%), followed by acrodermatitis continua of Hallopeau (ACH) (16 patients, 27.6%). Nearly half of our patients (27 patients, 46.6%) ever had ACH, and only three of them are free of ACH currently, which indicates that the development of ACH is relatively persistent and irreversible. Thirty‐four patients (58.6%) had recurrent GPP and 29 patients (50%) have been admitted due to GPP flare. Compared to those with heterozygous (C/T) mutation, more patients carrying homozygous mutation (C/C) have recurrent episodes of GPP (C/T vs. C/C: 25.53 vs. 76.47%, p = 0.0367). Two patients with squamous cell carcinomas arising from the pustular psoriasis skin lesions were noted. Two patients had elevated serum IgG4 levels.

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“…T helper 17 skewing may explain the co-occurrence of pustular psoriasis and IgG4-RD. 31 Articles investigating the association between ACH and IgG4-RD are lacking.…”

Section: Discussionmentioning

confidence: 99%

“…GPP has been reported as a rare skin manifestation of IgG4‐RD, but the IL‐36 RN mutation status was not specified in the case report. T helper 17 skewing may explain the co‐occurrence of pustular psoriasis and IgG4‐RD 31 . Articles investigating the association between ACH and IgG4‐RD are lacking.…”

Section: Discussionmentioning

confidence: 99%

Deficiency of interleukin‐36 receptor antagonist (DITRA): An analysis of 58 Chinese patients in a tertiary hospital in Taiwan

Hsieh

Huang

et al. 2023

Experimental Dermatology

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“…In contrast, IgG4 is an immunoglobulin subtype associated with allergic responses related to T helper 2 (Th2), and it has been hypothesized that Th2 plays a role in the pathogenesis of IgG 4 ‐RD . Interestingly, in cases of generalized pustular psoriasis, IgG 4 ‐RDs were reported as complications . In light of this, the present case suggests that Th2 mechanisms might be involved in the pathogenesis of pustular psoriasis and related pustular skin diseases, although we were unable to find fully reasonable explanations for the correlation between the Th2 reaction and pathogenic mechanisms of the present subtype of pustular psoriasis.…”

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confidence: 57%

Acrodermatitis continua of Hallopeau with dense infiltration of IgG4‐positive cells in the lesional dermis

et al. 2018

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DEAR EDITOR, Acrodermatitis continua of Hallopeau (ACH) is a rare acropustular subtype of psoriasis. 1 The characteristic feature of ACH is chronic, painful and destructive disease affecting hands and feet, especially fingertips and toes. 2 The pathogenic mechanisms of ACH have not been fully elucidated. We report an individual with ACH whose skin lesions showed dense infiltration of IgG4-positive cells in the dermis.An 80-year-old Japanese man consulted our hospital complaining of chronic persistent pustules and erosions on fingers, toes and the right upper arm, and severe nail deformities

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