Pyoderma gangrenosum in pregnancy

ARTICLE

Auteur(s) : Canan Gorpelioglu, Evren Sarifakioglu, Aylin Ayrim

Department of Dermatology, Fatih University Faculty of Medicine, Hosdere Cad. No: 145-147 06540/Y. Ayranci/Ankara, Turkey

Pyoderma gangrenosum (PG) is a painful, ulcerating skin disease of unknown aetiology. It can be associated with systemic diseases such as inflammatory bowel disease, inflammatory polyartritis and haematological disorders or after minor trauma and surgery [1]. We report a pregnant patient with PG that formed on a capillary hemangioma, but with no associated systemic disease.

A 26-year-old woman at 24 weeks’ gestation presented with a 3-week history of a painful ulcer on the right big toe. She recalled a capillary hemangioma beneath the lesion in the past. She was treated with topical mupirocin ointment and consecutive week-long courses of amoxicillin-clavulanate, but the lesion continued to enlarge, became ulcerated and developed a purulent discharge. Examination revealed a tender 3 × 5 cm crusted ulceration, surrounded by a rolled, raised border (figure 1). No microbial growth was detected on wound culture. A biopsy of the lesion showed numerous neutrophils within the dermis and epidermis. In the histopathological differential diagnosis, ulcerated pyogenic granuloma was excluded, because no lobular capillary hemangioma was present. The findings led us to suspect the diagnosis of PG, which was confirmed by the clinical features, repeatedly negative bacteriological cultures and lack of response to antibiotics. The following investigations were negative or within normal limits: complete blood count; erythrocyte sedimentation rate; biochemical, complement, immunoglobulin and autoantibody profiles, especially anticardiolipin antibodies; urinary examination, syphilis serology; chest radiograph; throat swabs; multiple ulcer swabs; and examination and culture of biopsy tissue for bacteria, fungi and acid-fast bacilli. She denied a history or clinical signs of inflammatory bowel disease. Radiological investigations of the alimentary tract were not performed.

The lesion was injected with intralesional corticosteroids. The concentration of triamcinalone acetonide was 5 mg/mL (by dilution, triamcinalone acetonide 40 mg/mL with water). A total 8 mL of mixture was injected at the lesion border. The injections were given weekly over a 2 month period until her delivery. The patient was seen in irregular follow-up during her delivery period. The lesion healed completely with residual erythema and scarring, two months after her delivery. During a follow up visit at 6 months, no recurrences appeared.

A few cases of PG have been described in pregnancy, following caesarean section and during the puerperium [1-3]. Most of these cases were associated with concomitant diseases and conditions, while our case had no significant medical history and all her investigations were in normal ranges. The effects of pregnancy on other dermatoses reveal that some can be healed or exacerbated [4]. No specific data exist for PG. Pregnancy is associated with immunosuppression, including IL-2 and IL-1 inhibition and depressed polymorphonuclear leukocyte chemotaxis and adherence functions. It is suggested that these alterations in the immune functions of pregnant women could play a part in the development of PG in pregnancy [1].

Functional modifications and focal hyperplasia of cutaneous blood vessels are particularly common during pregnancy and result in the enlargement of hemangioma. There was no finding relating to this type of vascular changes in our patient. Despite its rare association with pregnancy, PG in pregnancy poses an important therapeutic challenge. Successful systemic therapies for PG have included glucocorticoids, azothioprine, cyclosporine, acitretin and antibiotics [5, 6]. We could not safely use these treatment regimens because our patient was pregnant. Local therapies, such as intralesional steroid injections, as used in our patient, help to clear and halt progression of the lesion, without systemic therapies.

The true pathogenesis and first-line therapy of PG in pregnant women will only come with further sequential studies of this unusual disease.

Acknowledgements

References

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3 Steadman UA, Brennan TE, Daman LA, Curry SL. Pyoderma gangrenosum following cesarean delivery. Obstet Gynecol 1998; 91: 834-6.

4 Winton GB, Lewis CW. Dermatoses of pregnancy. J Am Acad Dermatol 1982; 6: 977-98.

5 Callen JP, Jackson JM. Pyoderma gangrenosum: an update. Rheum Dis Clin North Am 2007; 33: 787-802.

6 Lu XL, Zhao W, Xia YK, et al. Good response of a combined treatment of acitretin and antibiotics in blastomycosis-like pyoderma. Eur J Dermatol 2009 Feb 12 [Epub ahead of print].