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LETTER TO EDITOR
Year : 2019  |  Volume : 20  |  Issue : 4  |  Page : 355-356

A case report of pediatric pyoderma gangrenosum: A diagnostic and treatment challenge


Department of Dermatology, Southern Philippines Medical Center, Davao, Philippines

Date of Web Publication30-Sep-2019

Correspondence Address:
Dr Margaret Stephanie Lagman Jimenez
Southern Philippines Medical Center, JP Laurel Street, Davao
Philippines
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijpd.IJPD_146_18

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How to cite this article:
Jimenez MS, Tangtatco JA, Crisostomo MV, Tabalon MM. A case report of pediatric pyoderma gangrenosum: A diagnostic and treatment challenge. Indian J Paediatr Dermatol 2019;20:355-6

How to cite this URL:
Jimenez MS, Tangtatco JA, Crisostomo MV, Tabalon MM. A case report of pediatric pyoderma gangrenosum: A diagnostic and treatment challenge. Indian J Paediatr Dermatol [serial online] 2019 [cited 2019 Nov 17];20:355-6. Available from: http://www.ijpd.in/text.asp?2019/20/4/355/268392



Sir,

Pyoderma gangrenosum (PG) is a rare neutrophilic disease. Its etiology remains unknown.[1] Incidence is estimated at 3–10 patients per million per year where it is rare among the pediatric population.[2],[3],[4]

We present a 14-year-old male who presented with 3-year history of recurrent pruritic exudative ulcers on the lower extremities despite treatment with oral antibiotics.

A month prior to consult, the patient noted recurrence of ulcers on the lateral region of the right leg and on the anterior region of the left leg, rapidly increasing in size with severe pruritus. Physical examination of the skin showed a solitary 5 cm × 3 cm ulcer on the lateral aspect of the right leg and a solitary 20 cm × 7 cm ulcer spanning the anterior, medial, and lateral region of the left leg. Both ulcers had serous discharge and with violaceous, well-defined, and undermined borders [Figure 1]. Wound culture was positive for Pseudomonas aeruginosa while a 4-mm skin punch biopsy revealed findings suggestive of cellulitis. Baseline laboratory results were normal. The initial assessment was PG with the secondary bacterial infection.
Figure 1: A solitary 5 cm × 3 cm ulcer with a well-defined, undermined violaceous border on the lateral region of the distal 1/3 of the right leg (a). A solitary 20 cm × 7 cm well-defined ulcer with undermined borders spanning the lateral to medial region of the distal 1/3 of the left leg (b)

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The patient was started on oral ciprofloxacin to treat the underlying infection and later started on mupirocin + betamethasone dipropionate ointment applied twice daily on the ulcers. After 5 months, there was note of complete resolution of the ulcer on the right leg and decrease in the size of the ulcer on the left leg (18 cm × 7 cm). There was also note of string-like projections of epithelium at the edges of healing ulcers representing the Gulliver's sign of PG [Figure 2]. A repeat skin punch biopsy was done which showed the neutrophilic dermatosis consistent with PG [Figure 3].
Figure 2: Cribriform scarring on the right leg (a). Edges show stringlike projections (yellow arrows) of epithelium between ulcer bed and surrounding skin on the left leg (b and c)

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Figure 3: Biopsy of the ulcer showed confluent parakeratosis overlying an acanthotic and spongiotic epidermis with intraepidermal collections of neutrophils (a) and dermal edema as well as diffuse predominantly neutrophilic inflammatory infiltrates. There are focal leukocytoclasia and blood vessel wall damage noted (b) (a: H and E, ×40, b: H and E, ×400)

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With this he was started on prednisone at 40 mg/day (0.5 mkd) for 3 weeks but with no improvement. Due to this, prednisone was increased to 60 mg/day (0.75 mkd) for 2 weeks where there was note of decrease in size of ulcer on the right leg (17 cm × 5 cm). Dapsone was then initiated at 50 mg/day (0.6 mkd) when G6PD level was found adequate. It was later increased to 100 mg/day (1.25 mkd), whereas prednisone was gradually tapered to nil. After 17 months, the ulcer on the left leg completely healed [Figure 4].
Figure 4: Complete resolution of ulcer with cribriform scarring or “cigarette paper-like” scarring (white arrows) on the left leg after treatment

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PG is a rare disease which poses as a diagnostic and therapeutic challenge. There are no accepted international diagnostic criteria to date. For the untrained eye, clinicians may unknowingly treat this case as a recurrent infection.

Further, there is also no universally accepted gold standard of treatment. Systemic steroids may be the line but pose numerous side effects.[4],[5] Steroid-sparing drugs, such as dapsone, may be given as adjunct therapy or an alternative monotherapy.[1] PG is rarely seen in children but must be highly suspected in the setting of the chronic nonhealing wound.[3] Early recognition is of utmost importance as it leads to proper treatment, prevents rapid progression, and improves disease outcomes.

Acknowledgment

The authors would like to thank Dr. Mary Jo Kristine S. Bunagan for guiding the clinical findings and therapeutic management and also Dr. Maricarr Pamela M. Lacuesta for teaching the histological findings.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understand that name and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Gameiro A, Pereira N, Cardoso JC, Gonçalo M. Pyoderma gangrenosum: Challenges and solutions. Clin Cosmet Investig Dermatol 2015;8:285-93.  Back to cited text no. 1
    
2.
Soleimani T, Sasor SE, Spera L, Eppley BE, Socas J, Chu MW, et al. Pediatric pyoderma gangrenosum: Is it just big wounds on little adults? J Surg Res 2016;206:113-7.  Back to cited text no. 2
    
3.
Ruocco E, Sangiuliano S, Gravina AG, Miranda A, Nicoletti G. Pyoderma gangrenosum: An updated review. J Eur Acad Dermatol Venereol 2009;23:1008-17.  Back to cited text no. 3
    
4.
Ehling A, Karrer S, Klebl F, Schäffler A, Müller-Ladner U. Therapeutic management of pyoderma gangrenosum. Arthritis Rheum 2004;50:3076-84.  Back to cited text no. 4
    
5.
Yasin F, Assad S, Zahid M, Malik SA. Extensive pyoderma gangrenosum: A challenging diagnosis and literature review of management. Cureus 2017;9:e1486.  Back to cited text no. 5
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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