Home About us Editorial board Search Ahead of print Current issue Archives Submit article Instructions Subscribe Contacts 230


 
 Table of Contents  
CASE REPORT
Year : 2017  |  Volume : 8  |  Issue : 1  |  Page : 55-57

Childhood pyoderma gangrenosum


Department of Skin and VD, MGM Medical College, Navi Mumbai, Mumbai, Maharashtra, India

Date of Web Publication2-Feb-2017

Correspondence Address:
Mayur Ranu Bhobe
House No. 613/E/D/C, Cupangal, Behind Chowgule College, Fatorda, Salcete - 403 602, Goa
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0975-9727.199365

Rights and Permissions
  Abstract 

Pyoderma gangrenosum (PG) is rare in children. We report a case of PG in a 4-year-old male who presented with ulcers over face, axillae, and gluteal area. The ulcers healed with cribriform scarring after starting systemic steroids and azathioprine. The patient, however, was unable to follow-up regularly. He had a recurrence of lesions developed sepsis and septic shock and unfortunately expired.

Keywords: Child, cribriform scars, pyoderma gangrenosum, ulcer


How to cite this article:
Bhobe MR, Someshwar S, Jerajani HR, Ami D. Childhood pyoderma gangrenosum. Muller J Med Sci Res 2017;8:55-7

How to cite this URL:
Bhobe MR, Someshwar S, Jerajani HR, Ami D. Childhood pyoderma gangrenosum. Muller J Med Sci Res [serial online] 2017 [cited 2022 Jun 26];8:55-7. Available from: https://www.mjmsr.net/text.asp?2017/8/1/55/199365


  Introduction Top


First described in 1930 by Brunsting et al.,[1] pyoderma gangrenosum (PG) is an idiopathic neutrophilic dermatosis that was once considered pathognomonic of idiopathic ulcerative colitis. The histopathologic distinction of PG from other ulcerative processes with dermal neutrophilia is challenging and at times impossible.


  Case Report Top


A 4-year-old male child presented with ulcers over axillae, face, gluteal area, and legs for 15 days. Lesions reportedly began as blisters over both axillae which ruptured oozing clear fluid leaving ulcers that gradually increased to the current size. Similar lesions developed over buttocks, cheeks, and back of knees 5 days later. The ulcers were punched out with slough over the floor and violaceous rim [Figure 1],[Figure 2],[Figure 3]. The base was formed by muscle. There was no history of altered bowel habits or abdominal pain. There was no history of joint pains. There was no history of tiredness, spontaneous bleeding. The patient was investigated with a differential diagnosis of ecthyma gangrenosum, ecthyma, PG, pyoderma vegetans, chancriform pyoderma, and tuberculous gumma. Biopsy taken from the edge of the ulcer revealed focal loss in continuity of the epidermis with the center showing homogeneous eosinophilic material with neutrophilic microabscesses [Figure 4] and [Figure 5]. After clinicopathological correlation, diagnosis of PG was made.
Figure 1: Ulcers over axilla with punched out edges and slough over floor

Click here to view
Figure 2: Ulcers over face

Click here to view
Figure 3: Ulcers over gluteal area

Click here to view
Figure 4: Biopsy from the edge of ulcer showing loss in continuity of epidermis with homogeneous eosinophilic material (H and E, ×10)

Click here to view
Figure 5: Neutrophilic infiltrates in the floor of the ulcer (H and E, ×40)

Click here to view


There was an increase in total count with neutrophilia (absolute neutrophil count - 15,960). Tuberculin test was negative. Pathergy test was also negative. Culture from pus grew Escherichia coli and Pseudomonas. Stool examination was normal.

The patient was put on intravenous antibiotics and dressings with colloidal silver. However, there was no significant improvement. He was later started on systemic steroids at 1 mg/kg and azathioprine 20 mg at 1 mg/kg. Lesions healed with cribriform scars but would recur on attempting to taper steroids. The patient had started on pulse methylprednisolone 500 mg on 3 days/month. Unfortunately, the patient did not follow-up and developed a flare and died of septic shock.


  Conclusion Top


PG is a rare dermatosis. It usually occurs between 25 and 54 years of age and occurs rarely in children. In a study carried out at the Mayo Clinic, in a review of 180 cases diagnosed between 1930 and 1982, it was seen that only eight cases (4%) involved children younger than 15 years.[1] In a separate study of 86 patients seen at the Mayo clinic from 1970 to 1983, four cases (4.6%) were aged <14 years of age.[2] In adults, the lesions are commonly seen on the lower extremities; whereas in children, the lesions are also seen on the head and face, buttocks, and perianal and genital regions.[3] The above distribution of lesions was also seen in our case. In children, lesions begin as pustules rather than macules or papules.[1],[3],[4] Unlike in adults where the legs are more commonly involved, in children, the distribution is more generalized and the general prognosis is better than in adults.[5]

PG is known to be associated with arthritis, inflammatory bowel disease, hematologic malignancy, monoclonal gammopathy, and myeloma. In adults, PG is most frequently associated with arthritis; whereas in children, it is associated with ulcerative colitis,[1],[3],[4] followed by leukemia [5] and Crohn's disease.[3] We could not find any associated systemic disease despite thorough evaluation.

Systemic steroids are considered the drug of choice. Sulfa drugs such as sulfasalazine, sulfapyridine, and sulfamethoxypyridine have also been used. Refractory lesions have been successfully treated with the addition of clofazimine, dapsone, rifampicin, azathioprine, cyclophosphamide, and cyclosporine to the steroid regimen.[6],[7],[8] Although pediatric PG has a good prognosis, this patient ultimately died of septic shock despite initial response to immunosuppressive treatment.

Declaration of patient consent

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

Financial Support and Sponsorship

Nil.

Conflicts of Interest

There are no conflicts of interest.

 
  References Top

1.
Brunsting LA, Goeckermann WH, O'Leary PA. Pyoderma (ecthyma) Gangrenosum: Clinical and experimental observation in five cases occurring in adults. Arch Dermtol 1930;22:655-80.  Back to cited text no. 1
    
2.
Powell FC, Schroeter AL, Su WP, Perry HO. Pyoderma gangrenosum: A review of 86 patients. Q J Med 1985;55:173-86.  Back to cited text no. 2
    
3.
Graham JA, Hansen KK, Rabinowitz LG, Esterly NB. Pyoderma gangrenosum in infants and children. Pediatr Dermatol 1994;11:10-7.  Back to cited text no. 3
    
4.
Bhat RM. Pyoderma gangrenosum: An update. Indian Dermatol Online J 2012;3:7-13.  Back to cited text no. 4
[PUBMED]  Medknow Journal  
5.
Hayani A, Steuber CP, Mahoney DH Jr., Levy ML. Pyoderma gangrenosum in childhood leukemia. Pediatr Dermatol 1990;7:296-8.  Back to cited text no. 5
    
6.
Schwaegerle SM, Bergfeld WF, Senitzer D, Tidrick RT. Pyoderma gangrenosum: A review. J Am Acad Dermatol 1988;18:559-68.  Back to cited text no. 6
    
7.
Chow RK, Ho VC. Treatment of pyoderma gangrenosum. J Am Acad Dermatol 1996;34:1047-60.  Back to cited text no. 7
    
8.
Bhat RM, Shetty SS, Kamath GH. Pyoderma Gangrenosum in childhood. Int J Dermatol 2004;43:205-7.  Back to cited text no. 8
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]



 

Top
 
 
  Search
 
Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
 Related articles
Access Statistics
Email Alert *
Add to My List *
* Registration required (free)

 
  In this article
Abstract
Introduction
Case Report
Conclusion
References
Article Figures

 Article Access Statistics
    Viewed3082    
    Printed130    
    Emailed0    
    PDF Downloaded241    
    Comments [Add]    

Recommend this journal


[TAG2]
[TAG3]
[TAG4]