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Table of Contents
CASE REPORT
Year : 2017  |  Volume : 4  |  Issue : 1  |  Page : 62-65

Pyoderma gangrenosum following breast reconstruction: successful treatment with steroids


1 Department of Gynaecologic Oncology Unit, Gynaecology and Obstetrics, University Hospital La Paz, Madrid-, Spain
2 Department of Plastic Surgery, University Hospital La Paz, Madrid-, Spain
3 Department of Breast Pathology Unit, Gynaecology and Obstetrics, University Hospital La Paz, Madrid-, Spain

Date of Web Publication6-Jul-2017

Correspondence Address:
B Diaz-de la Noval
Department of Gynaecologic Oncology Unit, Gynaecology and Obstetrics, University Hospital La Paz
Spain
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Source of Support: None, Conflict of Interest: None


DOI: 10.5530/ami.2017.4.12

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  Abstract 


Introduction: Pyoderma gangrenosum is arare cause of skin necrosis after surgery. It often appears with extensive ulcerations and scaring with significant aesthetic aftermaths. The pathogenesis is poorly understood but often associated with inflammatory diseases, however post-surgical idiopathic pyoderma gangrenosum can occur. Case Report: We report a case of pyoderma gangrenosum following a bilateral breast reconstruction in a 33-year-old woman, successfully treated with steroids. A week after the surgery wounds started to break down and extensive necrotic ulceration appeared quickly covering both breasts. After dermatopathology evaluation she received antibiotics and prednisone and three months later wounds were completely healed and surgery was not necessary.

Keywords: Pyoderma gangrenosum, Breast reconstruction, Steroids


How to cite this article:
Noval B D, Sánchez C C, García J D, la Muela M H. Pyoderma gangrenosum following breast reconstruction: successful treatment with steroids. Acta Med Int 2017;4:62-5

How to cite this URL:
Noval B D, Sánchez C C, García J D, la Muela M H. Pyoderma gangrenosum following breast reconstruction: successful treatment with steroids. Acta Med Int [serial online] 2017 [cited 2017 Sep 22];4:62-5. Available from: http://www.actamedicainternational.com/text.asp?2017/4/1/62/209823




  Introduction Top


Pyoderma gangrenosum (PG) is an infrequent ulcerative skin disease.[1] PG is characterized by a quickly progressive necrosis. It is usually a manifestation of underlying systemic disease. PG is also known as Cullen's postoperative progressive gangrene (PPG). Several cases have been published,[2] but only some of them developed after breast surgery.[3],[4]

Our aim is to update the management of Pyoderma gangrenosum, reviewing its diagnosis, treatment and prognosis, apropos of a case and according to current literature.


  Materials and Methods Top


We report a case of pyoderma gangrenosum following breast reconstruction which was successfully treated with steroids. The etiology and therapy are discussed considering the current literature. Literature review was made searching in PubMed (http://www. ncbi.nlm.nih.gov/pubmed) with the terms “Pyoderma gangrenosum”, “breast reconstruction” and “steroids”.


  Results Top


We report a case of a 33-year-old Caucasian woman, carrying a BRCA-2 gene mutation, who quickly developed an inflammatoryinfectious process on the right breast, following bilateral mastectomies with breast reconstruction in November 2014. Rokitansky syndrome treated surgically at 19-years-old. She was in good health with no history of systemic illness or autoimmune disease.

The surgery consisted of both subcutaneous mastectomies with preservation of nippleareola-complex and immediate reconstruction (in the same surgical procedure) by bilateral sub-pectoral breast implant. The immediate postoperative period was going well. A week later she complained of pain from the wound on both breasts and lymphadenopathies on the right armpit, but was not valuable. The following week skin lesions appeared on both breasts with extensive necrotic ulceration rapidly covering the right breast. The inflammation increased and the scars started to break down [Figure 1]. With clinical suspicion of PG or an infection, assessment was conducted by dermatology, which took a sample for biopsy and microbiology.
Figure 1: Appearance of the right breast after reconstruction at the time of the diagnosis of Pyoderma gangrenosum. Extensive necrotic ulceration, inflammation and wound dehiscence along scars and nipple areola complex.

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We got normal results from the blood count, coagulation, biochemistry, serology, immune study, calprotectin and serum protein. Biopsy from the breast ulceration showed an intensive mixed acute inflammatory infiltrate, mainly neutrophils, with granulation tissue and granulomatous reaction, vessel destruction and necrosis of the epithelium [Figure 2]. The injury associated super infection and foreign body reaction.
Figure 2: Breast skin biopsy (Hematoxylin and Eosin stain (H&E) 40x).Intensive mixed acute inflammatory infiltrate, mainly neutrophils with granulation tissue and granulomatous reaction, vessel destruction and necrosis of the epithelium.

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Dermatopathological and pathological evaluations revealed PG. The patient was appropriately treated with a high-dose of corticosteroids (prednisone 50 mg/daily orally) and antibiotics (doxycycline 100 mg/daily 14 days), continuing with local cures. Pseudomonas aeruginosa was detected in culture and treated with ciprofloxacin 500mg / 12 hrs orally for 7 days.

Within a week of starting corticosteroids slight improvement of ulcers was observed. By the second week there was a clear improvement in the area of inflammation and some reepithelialisation of the skin with granulation tissue and signs of healing.

The same pattern of corticosteroids was maintained for a month, and then decreased weekly maintaining 5 mg/day on alternate days until the disappearance of the lesions. At the same time, she was treated conservatively with local measures, proteolytic agents to remove the necrotic tissue.

Two months later, the ulcers and inflammation had disappeared. At 3 months they were completely healed [Figure 3]. Finally, an aesthetic intervention or breast implant removal was not necessary. Pyoderma gangrenosum represents a rare dermatosis[5] that quickly evolves causing severe disruption of wound healing following surgical management.[6] The legs are the most common location of PG, but the disease may also involve other regions[7],[8] such as the breasts.[9] Even in typical cases PG is often misdiagnosed or not readily recognized, therefore wrongly treated.
Figure 3: Three months after starting steroids treatment wounds were completely healed with a really good aesthetic result.

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PG starts as an erythematous area that breaks down to form a painless skin irregular and deep ulcer with necrosis and hemorrhagic background. The ulcers may grow fast and result in extensive skin damage; although in rare cases they may remain unchanged for months.[7]

PG is idiopathic but can occur post-surgically.[3] The unknown aetiology and poorly understood pathogenesis often makes it difficult to detect and treat.[10] For about 50% of patients PG is usually a manifestation of an underlying systemic disease,[1] associated with monoclonal gammapathy, inflammatory bowel disease, myeloproliferative disorders, rheumatologic diseases and paraneoplastic syndrome. However post-surgical idiopathic PG can occur in healthy patients.[11] Our patient had an incident with intestinal discomfort years before, bloating and altered bowel habit, compatible with intestinal inflammatory process, but this could not be assured after digestive evaluation.

The pathogenesis remains unclear since there are cases without a preceding surgical intervention associated with a immunological disorder.[9] In most reported cases, the injuries were related to surgical interventions, as the result of “a pathergy phenomenon”. A common hapten (immunogenic molecule) between tumour and skin may explain the origin of this inflammatory disease.[5] Biopsies show chronic inflammatory infiltrate and skin necrosis but there was no subcutaneous damage, except in advanced disease.[4] Another theory is based on neutrophil and monocyte dysfunctions (phagocytosis or chemotaxis impaired function).[8]

PG is diagnosed after other possibilities are ruled out, but it can be suspected by clinical appearance and the lack of response to standard initial care.[12] The main differential diagnoses were skin and soft tissue infections including necrotizing fasciitis, Sweet's syndrome and malignant neoplasms.[13] PG can be difficult to distinguish from infection[1] but PG is a neutrophilicdermatosis with areas of thrombosis, haemorrhage, necrosis or re-epithelisation and absence of inflammatory lymphangitis or lymphadenopathy.[10] Pseudomonas infection detected in our case was considered secondary infection from wounds. Sweet's syndrome is a neutrophilicdermatosis usually associated with autoimmune disorders and malignant hematological neoplasm.[8]

Often, multiple therapies have been employed without success. The treatment used for PG includes systemic corticosteroids, azathioprine, mercaptopurine, sulphasalazine, sulphapyridine,[9] cyclophosphamidem,[13] isotretinoin, immunoglobulins and cyclosporine.[2] The recommended first-line therapy for PG are decreasing dose corticosteroids, between 20 to 30mg/d orally for 3 to 4 months and, if necessary, associated with other immunosuppressive drugs.[8] In our case the associated treatment was a systemic low-dose of corticosteroid therapy with local wound care. Our patient reacted well to systemic steroids and the ulcer completely healed after 3 months. Surgery or radiotherapy are contraindicated, injuries appear again,[8] but Baruch et al preferred surgical excision and systemic corticosteroids.[11],[13] Also, it should be remembered that treatment includes the management of the underlying disease if required.

Pyoderma gangrenosum has a recurrent nature and may be reactivated by various causes within a period of several years. Duchnowska et al[8] reported a case about of recurring PG as amanifestation of locally advanced breast cancer by exacerbation of rheumatoid arthritis. Long et al[14] suggested that avoiding epidermal sutures would prevent precipitation of PG at suture sites.

In conclusion, post-surgical idiopathic PG is very rare, but can occur after breast surgery. Early diagnosis and therapy are important to prevent serious clinical consequences. The recommended first-line therapy is medical management with local wound care and antibiotics.


  Conclusion Top


Conclusion is previous and last written parragraph. see above. In conclusion, post-surgical idiopathic PG is very rare, but can occur a er breast surgery. Early diagnosis and therapy are important to prevent serious clinical consequences. The recommended first-line therapy is medical management with local wound care and antibiotics.


  Conflict of Interest Top


The authors declare that they have no competing interests.


  Acknowledgement Top


None


  Abbreviations Used Top


PG: Pyoderma grangrenosum; PPG: Postoperative progressive gangrene.



 
  References Top

1.
Viard R, Scevola A, Veber M, Toussoun G, Delay E. Pyoderma gangrenosum: spontaneous healing after early diagnosis. Three cases and general review. Ann Chir Plast Esthet. 2013;58(2):146- 51. https://doi.org/10.1016/j.anplas.2011.01.001 PMid:21316137.  Back to cited text no. 1
    
2.
Schöfer H, Baur S. Successfusl treatment of postoperative Pyoderma gangrenosum with cyclosporin. J Eur Acad Dermatol Venereol. 2002;16(2):148-51. https://doi.org/10.1046/j.1468- 3083.2002.00387.x PMid:12046819.  Back to cited text no. 2
    
3.
Rajapakse Y, Bunker CB, Ghattaura A, Jallali N, Henton J, James SE, Harris PA, Searle AE. Case report: pyoderma gangrenosum following Deep Inferior Epigastric Perforator free flap breast reconstruction. J Plast ReconstrAesthet Surg. 2010;63(4):e395- 6. https://doi.org/10.1016/j.bjps.2009.10.017 PMid:19926352.  Back to cited text no. 3
    
4.
Baruch J, Julien M, Touraine R, Slaoui SE, Auffret P. Postoperative pyoderma gangrenosum and cancer of the breast. Apropos of a case. Chirurgie. 1989;115(2):142-5. PMid:2805923.  Back to cited text no. 4
    
5.
Labat JP, Simon H, Metges JP, Lucas B, Malhaire JP. Pyoderma gangrenosum and breast cancer: a new case. Ann Med Interne. 2000;151(4):314-5.  Back to cited text no. 5
    
6.
Henseler H, Menke H, Olbrisch RR. Pyoderma gangrenosum: a rare cause of unexpected wound healing delay. Chirurg. 2001;72(10):1196-200. https://doi.org/10.1007/s001040170060 PMid:11715624.  Back to cited text no. 6
    
7.
Davis MD, Alexander JL, Prawer SE. Pyoderma gangrenosum of the breasts precipitated by breast surgery. J Am Acad Dermatol. 2006;55(2):317-20. https://doi.org/10.1016/j.jaad.2006.02.066 PMid:16844520.  Back to cited text no. 7
    
8.
Long CC, Jessop J, Young M, Holt PJA. Minimizing the risk of post-operative pyoderma gangrenosum. Br J Dermatol. 1992;127:45-https://doi.org/10.1111/j.1365-2133.1992. tb14826.x PMid:1637694.  Back to cited text no. 8
    
9.
Mansur AT, Balaban D, Göktay F, Takmaz S. Pyoderma gangrenosum on the breast: A case presentation and review of the published work. J Dermatol. 2010;37(1):107-10. https://doi. org/10.1111/j.1346-8138.2009.00756.x PMid:20175832.  Back to cited text no. 9
    
10.
Baruch J, Julien M, Touraine R, Auffret P. Postoperative pyoderma gangrenosum and cancer of the breast. Apropos of a case. Ann Chir Plast Esthet. 1990;35(1):73-5. PMid:1693834.  Back to cited text no. 10
    
11.
Duchnowska R, Ziajka E, Góralska A, Grala B. Recurrent pyoderma gangrenosum precipitated by breast cancer: a case report and review of the literature. J Med Case Rep. 2014;25(8):https://doi.org/10.1186/1752-1947-8-226 PMid:24965126 PMCid:PMC4090656.  Back to cited text no. 11
    
12.
Maigre M, Bouachour G, Varache N, Alquier P, Verret JL. Pseudo-septicemic pyoderma gangrenosum and breast cancer. Apropos of a case caused by an intramuscular injection. Rev Med Interne. 1991;12(6):452-4. https://doi.org/10.1016/S0248- 8663(05)83195-9.  Back to cited text no. 12
    
13.
MacKenzie D, Moiemen N, Frame JD. Pyoderma gangrenosum following breast reconstruction. Br J Plast Surg. 2000;53(5):441-3. https://doi.org/10.1054/bjps.2000.3349 PMid:10876288.  Back to cited text no. 13
    
14.
Callen JR Pyoderma gangrenosum. Lancet 1998;351:581-5. https://doi.org/10.1016/S0140-6736(97)10187-8.  Back to cited text no. 14
    

 
  Authors Top





Díaz de la Noval, B. MD, a Gynaecologist. She obtained European Society of Gynaecology Oncology (ESGO) Fellowship Program of sub-specialization in Gynaecology Oncology Trainee. Currently working at Gynaecologic Oncology and breast pathology Unit, Department of Gynaecology and Obstetrics, LaPaz University Hospital, IdiPAZ, Madrid - Spain. Clinical Associate Professor in Gynecology and Obstetrics, Autonomous University of Madrid, Madrid - Spain. She is a Member of Spanish Gynecology and Obstetrics Society (SEGO) and Gynaecology- Oncology Section.








Casado Sanchez, C. MD, Ph.D. is a Plastic Surgeon and working in a department of Plastic, Reconstructive and Aesthetic Surgery, La Paz University Hospital,Madrid. Also working as Associate Professor of Surgery, Autonomous University of Madrid, Madrid - Spain. He is a Secretary General of the Spanish Society of Plastic, Reconstructive and Aesthetic Surgery. (SECPRE).








De Santiago García, J. MD, Ph.D. is a Gynaecologist. working in a Gynaecologic Oncology and breast pathology Unit, Department of Gynaecology and Obstetrics, La Paz University Hospital, IdiPAZ, Madrid - Spain.Head of Gynaecology and Obstetrics Department, La Paz University Hospital, Madrid - Spain.Medicine and Surgery Ph.D. Autonomous University of Madrid, Madrid -Spain.Associate Professor of Obstetrics and Gynaecology, Autonomous University of Madrid, Madrid -Spain. He is acting as a President of the Gynecologic Oncology and Breast Pathology section of the Spanish Gynecology and Obstetrics Society (SEGO).








Herrera de la Muela M, MD, Ph.D. is a Gynaecologist. Multidisciplinary Unit of Breast Disease, Department of Gyne- cology and Obstetrics. La Paz University Hospital Madrid - Spain. Medicine and Surgery Ph.D. Autonomous University of Madrid, Madrid - Spain. Also as Quality Coordinator for Gynaecology and Obstetrics Department. La Paz University Hospital,Madrid - Spain. She is a Member of La Paz Biomedicine Research Institute-IdiPAZ, Madrid - Spain. Associate Professor, Autonomous University of Madrid, Madrid - Spain.Being Member of the European Society of Surgical Oncology (ESSO).Member of the Spanish Society of Surgical Oncology (SEGO). She is a Member of the Spanish Society of Senology and Breast Pathology (SESPM). She is a Member of Spanish Gynecology and Obstetrics Society (SEGO) and Gynaecology-Oncology and Breast Pathology Section.






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