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CASE REPORT
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A challenging case of pyoderma gangrenosum following a routine cesarean section


 Department of Dermatology, Venerology and Leprosy, Dr. D. Y. Patil Medical College and Hospital and Research Centre, Pune, Maharashtra, India

Date of Submission02-Mar-2020
Date of Decision03-Jul-2020
Date of Acceptance07-Jul-2020

Correspondence Address:
Shreya Deoghare,
Department of Dermatology, Venerology and Leprosy, Dr. D. Y. Patil Medical College and Hospital and Research Centre, Sant Tukaram Nagar, Pimpri, Pune - 411 018, Maharashtra
India
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/mjdrdypu.mjdrdypu_115_20

  Abstract 


Pyoderma gangrenosum (PG) is a rare neutrophilic inflammatory skin condition which affects up to ten cases per million people per year. It mostly presents in the second to sixth decades of life with female preponderance. There are only a few reports of this condition following obstetric and gynecological procedures. The current article aims to highlight that PG should be kept as a differential in case of a nonhealing surgical site wound, misdiagnosis of which results in physical and psychological morbidity.

Keywords: Lower segment cesarean section, nonhealing ulcer, pyoderma gangrenosum



How to cite this URL:
Deo K, Deshmukh KM, Deoghare S, Kothari R. A challenging case of pyoderma gangrenosum following a routine cesarean section. Med J DY Patil Vidyapeeth [Epub ahead of print] [cited 2021 Jun 12]. Available from: https://www.mjdrdypv.org/preprintarticle.asp?id=317487




  Introduction Top


Pyoderma gangrenosum (PG) is a rare neutrophilic inflammatory skin condition.[1] It was first described in 1930 by Brunsting et al.[2] as an acute neutrophilic dermatosis. PG affects up to ten cases per million people per year. It is more common in females and may occur anywhere on the body and at any age, but mostly presents in the second to sixth decades of life.[1],[3],[4] Less than 15 cases of postsurgery PG have been reported in literature, worldwide.[5] Very few cases of PG following lower abdomen Cesarean section have been reported worldwide,[5],[6],[7],[8] and only two from India.[5],[6] In this article, we present a case of PG following Cesarean section which was managed by regular collagen dressing and daily prednisolone.


  Case Report Top


A 25-year-old female was referred to the department of dermatology from the department of obstetrics and gynecology on her 10th postoperative day in view of a large nonhealing ulcer at the site of lower segment Cesarean section (LSCS), which showed no growth of organisms on wound swab culture and failed to respond to debridement and antibiotics.

History revealed that the patient was a full-term primi who was diagnosed with abruptio placenta and underwent emergency LSCS to deliver a healthy female baby 10 days back. Recovery was satisfactory for the initial 3 days. On the 4th postoperative day, the patient developed fever of 38.5°F and increase in the intensity of pain at the site of LSCS. On the 6th postoperative day, the dressing was soaked with serous discharge. On the 7th postoperative day, the dressing was removed to reveal the gaping of wound. Routine laboratory investigations were within normal limits. There was no evidence of neutrophilic leukocytosis. Other tests including dengue NS1 antigen, Widal test, and rapid malaria test were sent in view of fever, and all were found to be normal. Repeated cultures of the wound site and blood were sent, which revealed no growth of organisms.

On examination, a large ulcer was noted at the site of LSCS measuring approximately 12 cm in length, 4 cm at the greatest width, and 5–6 cm in depth (one finger deep). The margin of the ulcer was irregular with violaceous undermined edge and granulation tissue at the base. There was a surrounding zone of erythema. The lesion was very tender [Figure 1].
Figure 1: Ulcer at the site of lower segment cesarean section measuring approximately 12 cm × 4 cm × 6 cm

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On further examination, a large scar measuring approximately 14 cm × 10 cm was noted over the right gluteal region. The scar was firm to touch with hard hyperpigmented raised borders. It was nontender [Figure 2].
Figure 2: A cribriform scar on the right gluteal region measuring approximately 14 cm × 10 cm

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On further inquiry, the patient gave a history of a similar episode of nonhealing wound on the right gluteal region following an intramuscular injection 2 years back, which took a long time to heal. Incision and drainage were done followed by oral antibiotics and anti-inflammatory drugs and with regular dressings.

On the basis of the typical history and examination findings, a provisional diagnosis of PG was made. A punch biopsy was taken from the margin of the lesion.

Local treatment included biweekly cleaning of the wound with normal saline and dressing with collagen. She was started on once-daily doses of tablet prednisolone 40 mg and tablet dapsone 100 mg along with analgesics and antihistaminics.

Histopathological finding of the biopsy showed subcorneal pustules with the underlying dermis having perivascular lymphocytic and neutrophilic infiltrate with leukocytoclasia [Figure 3]. Foci of vasculitis were seen.
Figure 3: (a) Low-power and (b) high-power views showing perivascular lymphocytic and neutrophilic infiltrates with leukocytoclasia

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The patient reported improvement in symptoms, and the size of the ulcer decreased gradually following regular collagen dressings and daily oral prednisolone and dapsone [Figure 4].
Figure 4: Sequential decrease in the size of ulcer with oral prednisolone and oral dapsone and regular collagen dressing. (a) POD 18, (b) POD 51, (c) POD 68, and (d) POD 86. (POD: postoperative day)

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The ulcer healed completely by postoperative day 86 [Figure 4]d.

The dose of prednisolone was then tapered, and dapsone was continued. The patient was given detailed information about her condition and was asked to consult a dermatologist before undergoing any surgical procedure in future.


  Discussion Top


PG is a rare neutrophilic inflammatory skin condition. The ulcerative variant is the most common. Other variants are bullous, pustular, and vegetative. PG presents as a painful nodule, plaque, or pustule. It then enlarges and breaks down to form an ulcer that progressively enlarges and has raised, undermined, violaceous borders and a surrounding zone of erythema. On healing, the lesions of PG lesions develop a cribriform appearance.[1]

Inflammatory mediators and aberrant neutrophilic activity play an important role in the pathogenesis of PG.[1]

PG most often occurs in association with systemic inflammation such as inflammatory bowel disease (IBD).[1] Our case was unique because there were no symptoms and signs suggestive of IBD. Furthermore, ultrasonography of the abdomen and pelvis was done, which was normal.

PG shows pathergy. It tends to occur at the site of trauma or irritation.[1] Hence, debridement should be avoided. In our case, the wound was cleaned with sterile saline, collagen was applied over the raw areas, and dressing was done with mildly adhesive micropore on alternate days.

PG is no longer considered a diagnosis of exclusion.[9] In our case, the diagnosis of PG was made based on “A Delphi Consensus of International Experts-Diagnostic Criteria of Ulcerative Pyoderma Gangrenosum.”[9] The presence of classical dermatological and histological findings in the absence of infection, a typical history suggestive of pathergy, and response to immunosuppressive medication were suggestive of the diagnosis of PG.

Mild PG responds to topical or intralesional corticosteroids.[1] For more severe or extensive PG, systemic glucocorticoids are given.[1] For very aggressive disease, intravenous pulse methylprednisolone is the initial therapy, followed by daily oral glucocorticoids.[1] Response to systemic glucocorticoids is rapid and halts progression within 1–2 weeks.

We emphasize the importance of careful evaluation of any postsurgical, nonhealing wound to identify PG at the earliest. Local wound management with oral steroids is the mainstay in the treatment of PG.

Declaration of patient consent

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

Acknowledgment

We would like to thank Dr. M. S. Deora and Dr. Neeta Gokhale for their constant support and motivation throughout the management of this case and while writing this case report. We would also like to thank the Department of Obstetrics and Gynaecology, Dr. D. Y. Patil Medical College and Hospital, Pimpri, Pune for their valuable suggestions and guidance while managing this case.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Jourabchi N, Lazarus GS. Pyoderma gangrenosum. In: Kang S, Amagai M, Bruckner AL, Enk AH, Margolis DJ, Mcmichael AJ, et al., editors. Fitzpatrick's Dermatology. 9th ed. New York: Mcgraw-Hill Education; 2019. p. 605-16.  Back to cited text no. 1
    
2.
Brunsting LA, Goeckerman WH, O'Leary PA. Pyoderma (ecthyma) gangrenosum: Clinical and experimental observations in five cases occurring in adults. Arch Dermatol Syphilol 1930;22:655-80.  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.
Körber A, Klode J, Al-Benna S, Wax C, Schadendorf D, Steinstraesser L, et al. Etiology of chronic leg ulcers in 31,619 patients in Germany analyzed by an expert survey. J Dtsch Dermatol Ges 2011;9:116-21.  Back to cited text no. 4
    
5.
Radhika AG, Singal A, Radhakrishnan G, Singh S. Pyoderma gangrenosum following a routine caesarean section: Pseudo-infection in a caesarean wound. Qatar Med J 2015;2015:1.  Back to cited text no. 5
    
6.
Amin SV, Bajapai N, Pai A, Bharatnur S, Hebbar S. Pyoderma gangrenosum in two successive pregnancies complicating caesarean wound. Case Rep Obstet Gynecol 2014;2014:654843.  Back to cited text no. 6
    
7.
Banga F, Schuitemaker N, Meijer P. Pyoderma gangrenosum after caesarean section: A case report. Reprod Health 2006;3:9.  Back to cited text no. 7
    
8.
Murata T, Kyozuka H, Fukuda T, Hiraiwa T, Yamaguchi A, Fujimori K. Incisional pyoderma gangrenosum after caesarean section: Two case reports. Case Rep Womens Health 2019;23:e00128.  Back to cited text no. 8
    
9.
Maverakis E, Ma C, Shinkai K, Fiorentino D, Callen JP, Wollina U, et al. Diagnostic criteria of ulcerative pyoderma gangrenosum: A Delphi consensus of international experts. JAMA Dermatol 2018;154:461-6.  Back to cited text no. 9
    


    Figures

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



 

 
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