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CORRESPONDENCE
Year : 2016  |  Volume : 61  |  Issue : 6  |  Page : 693-694
A case of relapsing polychondritis with palmoplantar pustulosis


Department of Dermatology, Hallym University Sacred Heart Hospital, Anyang, Korea

Date of Web Publication9-Nov-2016

Correspondence Address:
Won Joo Kwon
Department of Dermatology, Hallym University Sacred Heart Hospital, Anyang
Korea
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0019-5154.193701

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How to cite this article:
Kwon WJ, Kim MS, Park JY, Cho EB, Park EJ, Kim KH, Kim KJ. A case of relapsing polychondritis with palmoplantar pustulosis. Indian J Dermatol 2016;61:693-4

How to cite this URL:
Kwon WJ, Kim MS, Park JY, Cho EB, Park EJ, Kim KH, Kim KJ. A case of relapsing polychondritis with palmoplantar pustulosis. Indian J Dermatol [serial online] 2016 [cited 2021 Mar 7];61:693-4. Available from: https://www.e-ijd.org/text.asp?2016/61/6/693/193701


Sir,

Relapsing polychondritis (RP) is a rare systematic autoimmune disease that is characterized by chronic inflammation and degeneration of cartilage, and it is often associated with other autoimmune inflammatory diseases. [1] Therefore, one could stress that RP should not be viewed as a unique disease but as a syndrome. [2] There have been a few cases of RP accompanying psoriasis vulgaris, among which there is a case of pustular psoriasis, but the disease has never been limited to hands and soles. [2],[3]

A 52-year-old Korean woman presented with skin lesions on both the palms and soles over the course of several weeks. She was diagnosed with RP 5 years ago with the symptoms of pain and swelling of proximal interphalangeal (PIP) joints of the hand, earlobes swelling and cartilage deformity, hoarseness, dyspnea, and fulfilled the empirical diagnostic criteria after excisional biopsy of the right auricular mass. Administration of oral steroid relieved symptoms, and she was taking methotrexate 12.5 mg/week as a maintenance therapy. The sequela of RP included swelling on her PIP joints with mild pain, deformity of both ear cartilages, and soft tissue thickening. As mentioned above, the patient had undergone biopsy in the right auricular mass at the time of diagnosis of RP, and histologic reading showed lymphohistiocytic infiltration and fibrosis around perichondrium, degeneration of cartilage and vacuolization of chondrocytes, and chronic active inflammation with foam cell collection [Figure 1]. Physical examination revealed localized erythematous crusted papules and sterile, purulent vesicles on both palms [Figure 2] and soles, more clearly noted on the former, with pruritus and tenderness. Biopsy results showed hyperkeratosis, parakeratosis, diminished granular layer, and subcorneal spongiform pustule filled with polymorphonuclear leukocytes [Figure 3]. She is now taking cyclosporine 100 mg/day and applying betamethasone/calcipotriol ointment in addition to methotrexate 12.5 mg/week for RP and her condition is improving.

Both RP and palmoplantar pustulosis (PPP) are immune-mediated diseases, yet their target antigens are still unclear, and the same is true for their exact pathogenesis. [4] Relation between RP and PPP is unclear in this case, but we assume that the underlying immune defect played an important role for both diseases. As in the past study involving five cases of psoriasis with RP, [5] there seems to little correlation between clinical course of both diseases, and there is a possibility that both diseases coincidentally occurred together.
Figure 1: Perichondrial lymphohistiocytic infiltration and degeneration of cartilage, vacuolization of chondrocytes and chronic active inflammation with foam cell collection (H and E, ×200)

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Figure 2: Localized erythematous pinhead-sized crusted papules and sterile, purulent vesicles on the both palms

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Figure 3: Subcorneal spongiform pustule and an intraepidermal cavity filled with polymorphonuclear leukocytes associated with spongiform changes within the surrounding epidermis (H and E, ×100)

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This case is significant in that RP co-occurred with PPP, a case not reported in the known literature. Globally, only five case reports demonstrate RP complicated with psoriasis vulgaris; only one of those was pustular psoriasis, and the lesion in question was not limited to the hands. Furthermore, PPP was used to be classified as a subtype of pustular psoriasis, but currently, PPP is recognized as an independent entity. The different clinical courses of RP and PPP imply that though the same immune defect may be behind the manifestations of both diseases, the pathogenesis regarding the evolution of disease from the beginning may take a different course, which needs further studies.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
   References Top

1.
Cohen PR, Rapini RP. Relapsing polychondritis. Int J Dermatol 1986;25:280-5.  Back to cited text no. 1
    
2.
Hager MH, Moore ME. Relapsing polychondritis syndrome associated with pustular psoriasis, spondylitis and arthritis mutilans. J Rheumatol 1987;14:162-4.  Back to cited text no. 2
    
3.
Trentham DE, Le CH. Relapsing polychondritis. Ann Intern Med 1998;129:114-22.  Back to cited text no. 3
    
4.
Arnaud L, Mathian A, Haroche J, Gorochov G, Amoura Z. Pathogenesis of relapsing polychondritis: A 2013 update. Autoimmun Rev 2014;13:90-5.  Back to cited text no. 4
    
5.
Borbujo J, Balsa A, Aguado P, Casado M. Relapsing polychondritis associated with psoriasis vulgaris. J Am Acad Dermatol 1989;20:130-2.  Back to cited text no. 5
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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