Recurrent palmoplantar pustulosis at the site of insertion of therapeutic titanium

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Recurrent palmoplantar pustulosis at the site
of insertion of therapeutic titanium

Fukumoto, Takeshi
Horita, Nobuyuki
Yoshioka, Ai
(Citation)
The Journal of Dermatology,50(7):e223-e225

(Issue Date)
2023-07

(Resource Type)
journal article

(Version)
Accepted Manuscript

(Rights)
This is the peer reviewed version of the following article: [Fukumoto, T., Horita, N.
and Yoshioka, A. (2023), Recurrent palmoplantar pustulosis at the site of insertion of
therapeutic titanium. J Dermatol, 50: e223-e225.], which has been published in final
form at [https://doi.org/10.1111/1346-8138.16750]. This article may be used for non-…
commercial
purposes in accordance with Wiley Terms and Conditions for Use of Self(URL)
Archived Versions. This article may not be enhanced, enriched or otherwise transformed
https://hdl.handle.net/20.500.14094/0100482873
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Recurrent palmoplantar pustulosis at the site of insertion of therapeutic titanium

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Running Title: PPP and titanium

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Word Count: 488, References: 5, Figures and Tables: 1 figure

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Takeshi Fukumoto1*, Nobuyuki Horita2, Ai Yoshioka1

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School of Medicine, 7-5-1 Kusunoki-cho, Chuo-ku, Kobe 6500017, Japan

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Division of Dermatology, Department of Internal Related, Kobe University Graduate

Chemotherapy Center, Yokohama City University Hospital, Yokohama, Japan

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*Corresponding Author: Takeshi Fukumoto, M.D., Ph.D.

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Division of Dermatology, Department of Internal Related, Kobe University Graduate

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School of Medicine, 7-5-1 Kusunoki-cho, Chuo-ku, Kobe 650-0017, Japan

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Tel.: +81-78-382-6134, Fax: +81-78-382-6149, E-mail: fuku@med.kobe-u.ac.jp

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FUNDING: None

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CONFLICTS OF INTEREST: None

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INFORMED CONSENT:

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The patients in this manuscript have given written informed consent.

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AUTHORS’ CONTRIBUTIONS： T.F., N.H., and A.Y. designed the study and drafted

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the manuscript. T.F. and A.Y. contributed to data collection and interpretation of the

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results. All authors have read and approved the final manuscript.

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KEYWORDS: metal allergy, titanium, palmoplantar pustulosis (PPP), dermcidin

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Dear Editor,

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Palmoplantar pustulosis (PPP) is a refractory inflammatory disease characterized

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by pustular eruptions on the palms and soles.1-3 The etiology of PPP is unknown, but it

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may be caused by metal allergy, tonsilitis, and smoking.1-4 The mechanism by which

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metal allergy causes PPP has not been elucidated yet. A systematic review of 519

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patients with PPP revealed that metal allergens triggered PPP.3 Conversely, another

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retrospective analysis suggested that dental metal removal did not improve PPP

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symptoms.1 Herein, we report a case with PPP presenting with recurrence of

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non-bacterial pustular eruptions only at the site of insertion of therapeutic titanium for

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bone fracture healing.

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A 62-year-old man had PPP on the palms and soles for 15 years and had been

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in remission at the time of the present case report. The patient had a bone fracture in his

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left lower leg, which was treated with a therapeutic titanium implant. After 5 months, he

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presented with non-bacterial pustular eruptions with erythematous scaling only at the

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site of insertion of therapeutic titanium (Figure 1a–e). The skin biopsy from the pustule

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on the left lower leg showed PPP manifestations (Figure 1f). First, we hypothesized that

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the operation led to an eccrine sweat glands disorder resulting in sweat leakage in the

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dermis of the affected area. However, a thermoregulatory sweat test using the

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starch-iodine method with sweating revealed neither hypohidrosis nor hyperhidrosis of

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the affected area (Figure 1g–h).5 Moreover, immunohistochemical staining for

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dermcidin, the major sweat antimicrobial peptide, did not show sweat leakage (Figure

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1i–j).5 The patch test with metal allergens revealed the erythema result for titanium and

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iron, both of which are included in the insertion although the iron content is low (<1%)

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(Figure 1k). Patient’s symptoms responded to the difluprednate ointment, but when the

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treatment was discontinued the symptoms recurred.

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PPP may be caused by various factors.1,3,4 In the present study, we showed that

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the potential contribution of metal allergy to the etiology of PPP is in line with the

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systematic review by Brunasso et al.3 However, no similar cases have been reported and

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it is difficult to confirm whether a metal allergy is the main cause, because therapeutic

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titanium cannot be removed from the bone. Also, it is challenging to propose a

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mechanism explaining how it would lead to the symptoms of PPP. Although titanium

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allergy is rare, titanium used for orthopedic surgery for left distal radius fracture

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reportedly led to PPP.4 Murakami et al. performed the immunohistochemical staining of

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dermcidin as a maker for sweat secretion and revealed the contribution of the abnormal

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eccrine sweating to PPP.2 However, the sweat secretion, as well as sweat function, were

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normal in this case. Further studies are needed to elucidate the mechanism through

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which metal allergies cause PPP, and whether the Koebner phenomenon by bone

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fracture and/or operation may induce a PPP lesion. Nevertheless, our results show the

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potential contribution of metal allergy to PPP etiology.

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References:

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1.

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palmoplantar pustulosis after dental infection control and dental metal removal. J Dermatol. Jun

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2017;44(6):695-698. doi:10.1111/1346-8138.13751

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2.

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formation

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doi:10.1038/jid.2010.87

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3.

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Systematic Review. Dermatol Pract Concept. Apr 2019;9(2):105-110. doi:10.5826/dpc.0902a05

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4.

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Pustulosis after On-Pump Coronary Artery Bypass Grafting in a Patient with Titanium Allergy. Ann

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Thorac Cardiovasc Surg. Jun 20 2020;26(3):170-173. doi:10.5761/atcs.cr.18-00031

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5.

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factors for unilateral lichen planus. J Dermatol. Aug 2017;44(8):963-966. doi:10.1111/1346-8138.13836

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Kouno M, Nishiyama A, Minabe M, et al. Retrospective analysis of the clinical response of

Murakami M, Ohtake T, Horibe Y, et al. Acrosyringium is the main site of the vesicle/pustule
in

palmoplantar

pustulosis.

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Dermatol.

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2010;130(8):2010-6.

Brunasso Vernetti AMG, Puntoni M, Massone C. Palmoplantar Pustulosis and Allergies: A
Kono T, Oda T, Akaiwa K, Nakamura K, Sasaoka K, Tanaka H. Remission of Palmoplantar

Fukumoto T, Tsuchiyama S, Fukunaga A, Nishigori C. Hypohidrosis and metal allergy: Trigger

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Figure 1. Clinical and histopathological findings of the patient with palmoplantar

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pustulosis at the site of insertion of therapeutic titanium in the left lower leg

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(a–e) Clinical appearance of palmoplantar pustulosis at the site of insertion of

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therapeutic titanium in the left lower leg, showing pustular eruptions with erythematous

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scaling.

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(f) Hematoxylin and eosin staining of the skin biopsy from the left lower leg, showing a

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micro abscess in the epidermis with inflammatory cell infiltration. [×20, scale bar = 500

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μm]

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(g, h) No significant difference was observed in the number of black dots as perspiration

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points between the soles of the feet.

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(i, j) Immunohistochemical staining of dermcidin (mouse monoclonal antibody to

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dermcidin [G-81]; Santa Cruz Biotechnology, Dallas, TX) showing no significant sweat

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leakage in the affected area. [i: ×40, scale bar = 500 μm, j: ×200, scale bar = 100 μm]

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(k) The patch test reaction using titanium (10.0 % Titanium in petrolatum, Cat. T-042,

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CHEMOTECHNIQUE DIAGNOSIS, Sweden) showed the slightly edematous

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erythema. ...

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