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Pyoderma gangrenosum after stapedotomy - First report in otolaryngology literature
Otolaryngology Case Reports 6 (2018) 22–24
Contents lists available at ScienceDirect
Otolaryngology Case Reports journal homepage: www.elsevier.com/locate/xocr
Pyoderma gangrenosum after stapedotomy - First report in otolaryngology literature
T
Luiz C.A. Sousaa,b, Aldo J. Bellodia,b, Daiane O. Bragaa,b, Henrique F. Paunac,∗ a
Clínica Paparella, Ribeirão Preto, São Paulo, Brazil Department of Otorhinolaryngology, University of Ribeirão Preto, Ribeirão Preto, São Paulo, Brazil c Department of Otorhinolaryngology, Head and Neck Surgery, University of Campinas, Campinas, São Paulo, Brazil b
A R T I C L E I N F O
A B S T R A C T
Keywords: Pyoderma gangrenosum Stapedotomy Inflammatory diseases Titanium prostheses
Introduction: Pyoderma gangrenosum is an inflammatory disease of unknown etiology characterized by a rapidly progressive necrotizing fasciitis. Our objective is to report a case of pyoderma gangrenosum after stapedotomy. Case report: A 46 years-old female, presented an ulcerated skin lesion above the left pinna after stapedotomy, diagnosed as pyoderma gangrenosum. Clinical treatments failed, and surgical removal of the prosthesis lead to completed healing of the lesion. Discussion: Pyoderma gangrenosum has been associated as a complication of many surgical procedures. However, to the best of our knowledge, this is the first study to describe this disease as complication of a stapedotomy. Conclusion: The healing of pyoderma gangrenosum after the prosthesis removal suggests its participation on the genesis of the disease. This fact had not yet been reported in the literature.
Introduction Pyoderma gangrenosum (PG) is an inflammatory disease of unknown etiology, rarely seen in surgical disciplines, characterized by a rapidly progressive necrotizing fasciitis of the wound edges with typical morphologic features [1,2]. Here, we present a case of PG after stapedotomy. Case presentation A 46 years-old, female Caucasian, was diagnosed with bilateral conductive hearing loss due to otosclerosis. She underwent to stapedotomy on her left in October 2006, and on the right in June 2007. It was used a titanium prosthesis for both ears, with good hearing outcomes after the surgeries. One year after the second procedure, she started to develop an ulcerative skin lesion over the left pinna and left temporal area, with no other systemic symptoms (Fig. 1). Biopsy of the affected area showed irregular epithelium hyperplasia, serous-neutrophilic crusts and dermal fibrosis with foci of perivascular lympho-histiocytic infiltration, and neutrophil exudate (Figs. 2 and 3). Additionally, no foci of malignancy were found within the samples.
Culture was not performed from the sample. A dermatologic evaluation resulted in the diagnosis of pyoderma gangrenosum, and the experienced team of dermatologists recommended the removal of the prosthesis from the left ear. Otoscopy, audiogram, and CT scan of the temporal bone were normal, during this period. However, few months later, the patient started to complain about tinnitus in her left ear. A new audiogram was performed, and a severe sensorineural hearing loss was observed on her left ear. Another CT scan was obtained, and it was again, normal. After discussion with the Otology team, the patient agreed to remove the prosthesis. During the surgery, middle ear looked clear, the prosthesis was normally vibrating, and no drainage was observed from the vestibule. After three months of the procedure, the patient was in good shape, with excellent healing aspect. There was no recurrence of the dermatologic lesion in seven years of follow-up. Discussion Pyoderma gangrenosum is considered a highly destructive neutrophilic dermatosis, non-infectious, mainly located in lower body (77%) whereas 7.8% occurs on the head [3]. It is usually presented as superficial ulcers with mucopurulent exudate, eventually hemorrhagic
∗ Corresponding author. Department of Otorhinolaryngology, Head and Neck Surgery, 126, Tessália Vieira de Camargo St, University of Campinas – UNICAMP, Campinas, 13083-887, SP, Brazil. E-mail address: [email protected] (H.F. Pauna).
https://doi.org/10.1016/j.xocr.2017.12.001 Received 24 September 2017; Received in revised form 28 November 2017; Accepted 4 December 2017 Available online 06 December 2017 2468-5488/ © 2017 Published by Elsevier Inc. This is an open access article under the CC BY license (http://creativecommons.org/licenses/BY/4.0/).
Otolaryngology Case Reports 6 (2018) 22–24
L.C.A. Sousa et al.
Fig. 1. Image of the patient after one year of the second procedure. Note the ulcerative lesion at the left temporal region of the head. No bleeding or active inflammatory reaction was seen (A). A closer picture of the ulcerative lesion above the left pinna (B).
Fig. 3. Histopathology of the biopsy: skin with irregular epithelial hyperplasia, serousneutrophilic (arrows) and fibrotic foci with perivascular lymphocytic infiltrate and neutrophilic exudate (asterisk) (H&E, 20×, A; H& E, 60×, B).
It is important to note that the incidence peak of PG occurs in older patients (> 50 years, 44.4%), and predominantly in women [5]. Additionally, many patients present early lesions spreading despite preventive antibiotics and no bacterial growth from wound swabs [2,5]. Our patient presented the signals and symptoms with no association to other systemic diseases and was not responsive to clinical treatment. Interestingly, no single medical treatment is effective in PG patients [5]. Systemic steroids with or without immunosuppressive therapy are the gold standard, but other agents have been successfully reported, such as azathioprine, sulfasalazine, dapsone, thalidomide, colchicine, tacrolimus, methotrexate, intravenous immunoglobulin, systemic antibiotics (like tetracyclines and rifampicin), anti-TNF-α (tumor necrosis factor-alfa) – in cases of resistant PG – negative pressure therapy, and hyperbaric oxygen [1,4,5]. No previous reports of PG after stapedotomy have been described. In this case, the disease developed at the side of the second surgery. The lesion was initially managed as infections with antibiotics and removal of necrotic tissues, which made the ulcerative skin disease worse. Finally, after the prosthesis removal, patient evolved with good healing of the lesion.
Fig. 2. Histopathology of the biopsy: irregular epithelial hyperplasia, neutrophilic (arrows) and fibrotic foci with perivascular invasion (asterisk) (H&E, 2×).
[3]. Pathology is typically an ulcerative lesion with polynuclear neutrophil infiltrate and associated capillary network disorder (mainly, venous thrombosis), but it helps a little [2]. It has been associated with leukemia, inflammatory bowel disease, and induction by a trauma may be considered one of the pathogenic processes, as well as the surgical trauma of our patient [4]. PG has been associated as a complication of many surgical procedures. However, to the best of our knowledge, this is the first study to describe PG as complication of a stapedotomy. Interestingly, this fact happened after two years of the first surgery.
Conclusion Pyoderma gangrenosum is known to be initiated at the site of surgical trauma. Early diagnosis of PG is vital to prevent secondary 23
Otolaryngology Case Reports 6 (2018) 22–24
L.C.A. Sousa et al.
infection of the wound, and to avoid more aggressive treatment.
Appendix A. Supplementary data
Consent
Supplementary data related to this article can be found at http://dx. doi.org/10.1016/j.xocr.2017.12.001.
An informed consent was obtained previously to all procedures.
References
Conflict of interest
[1] Rothenburger M, Tjan TD, Schmid C, Schmidt C, Schwarz T, Scheld HH. Pyoderma gangrenosum after aortic valve replacement. Ann Thorac Surg 2001;71(1):349–51. [2] Kasper K, Manger B, Junger A, Reichert B, Sievers R, Herdtle S. Pyoderma gangrenosum following AICD implantation: differential diagnosis to necrotizing fasciitis. Anaesthesist 2012;61(1):47–51. [3] Kurniadi I, Imanishi H, Furukawa H, Sowa-Osako J, Tsuruta D. Case of facial pyoderma gangrenosum. J Dermatol 2016;43(11):1373–4. [4] Adšen E, Erduran F, Gürer MA. Pyoderma gangrenosum: a report of 27 patients. Int J Low Extrem Wounds 2016;15(2):148–54. [5] Wollina U. Pyoderma gangrenosum – a review. Orph J Rare Dis 2007;2:19–27.
The authors declare that they have no competing interests. Financial disclosure This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.
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Contents lists available at ScienceDirect
Otolaryngology Case Reports journal homepage: www.elsevier.com/locate/xocr
Pyoderma gangrenosum after stapedotomy - First report in otolaryngology literature
T
Luiz C.A. Sousaa,b, Aldo J. Bellodia,b, Daiane O. Bragaa,b, Henrique F. Paunac,∗ a
Clínica Paparella, Ribeirão Preto, São Paulo, Brazil Department of Otorhinolaryngology, University of Ribeirão Preto, Ribeirão Preto, São Paulo, Brazil c Department of Otorhinolaryngology, Head and Neck Surgery, University of Campinas, Campinas, São Paulo, Brazil b
A R T I C L E I N F O
A B S T R A C T
Keywords: Pyoderma gangrenosum Stapedotomy Inflammatory diseases Titanium prostheses
Introduction: Pyoderma gangrenosum is an inflammatory disease of unknown etiology characterized by a rapidly progressive necrotizing fasciitis. Our objective is to report a case of pyoderma gangrenosum after stapedotomy. Case report: A 46 years-old female, presented an ulcerated skin lesion above the left pinna after stapedotomy, diagnosed as pyoderma gangrenosum. Clinical treatments failed, and surgical removal of the prosthesis lead to completed healing of the lesion. Discussion: Pyoderma gangrenosum has been associated as a complication of many surgical procedures. However, to the best of our knowledge, this is the first study to describe this disease as complication of a stapedotomy. Conclusion: The healing of pyoderma gangrenosum after the prosthesis removal suggests its participation on the genesis of the disease. This fact had not yet been reported in the literature.
Introduction Pyoderma gangrenosum (PG) is an inflammatory disease of unknown etiology, rarely seen in surgical disciplines, characterized by a rapidly progressive necrotizing fasciitis of the wound edges with typical morphologic features [1,2]. Here, we present a case of PG after stapedotomy. Case presentation A 46 years-old, female Caucasian, was diagnosed with bilateral conductive hearing loss due to otosclerosis. She underwent to stapedotomy on her left in October 2006, and on the right in June 2007. It was used a titanium prosthesis for both ears, with good hearing outcomes after the surgeries. One year after the second procedure, she started to develop an ulcerative skin lesion over the left pinna and left temporal area, with no other systemic symptoms (Fig. 1). Biopsy of the affected area showed irregular epithelium hyperplasia, serous-neutrophilic crusts and dermal fibrosis with foci of perivascular lympho-histiocytic infiltration, and neutrophil exudate (Figs. 2 and 3). Additionally, no foci of malignancy were found within the samples.
Culture was not performed from the sample. A dermatologic evaluation resulted in the diagnosis of pyoderma gangrenosum, and the experienced team of dermatologists recommended the removal of the prosthesis from the left ear. Otoscopy, audiogram, and CT scan of the temporal bone were normal, during this period. However, few months later, the patient started to complain about tinnitus in her left ear. A new audiogram was performed, and a severe sensorineural hearing loss was observed on her left ear. Another CT scan was obtained, and it was again, normal. After discussion with the Otology team, the patient agreed to remove the prosthesis. During the surgery, middle ear looked clear, the prosthesis was normally vibrating, and no drainage was observed from the vestibule. After three months of the procedure, the patient was in good shape, with excellent healing aspect. There was no recurrence of the dermatologic lesion in seven years of follow-up. Discussion Pyoderma gangrenosum is considered a highly destructive neutrophilic dermatosis, non-infectious, mainly located in lower body (77%) whereas 7.8% occurs on the head [3]. It is usually presented as superficial ulcers with mucopurulent exudate, eventually hemorrhagic
∗ Corresponding author. Department of Otorhinolaryngology, Head and Neck Surgery, 126, Tessália Vieira de Camargo St, University of Campinas – UNICAMP, Campinas, 13083-887, SP, Brazil. E-mail address: [email protected] (H.F. Pauna).
https://doi.org/10.1016/j.xocr.2017.12.001 Received 24 September 2017; Received in revised form 28 November 2017; Accepted 4 December 2017 Available online 06 December 2017 2468-5488/ © 2017 Published by Elsevier Inc. This is an open access article under the CC BY license (http://creativecommons.org/licenses/BY/4.0/).
Otolaryngology Case Reports 6 (2018) 22–24
L.C.A. Sousa et al.
Fig. 1. Image of the patient after one year of the second procedure. Note the ulcerative lesion at the left temporal region of the head. No bleeding or active inflammatory reaction was seen (A). A closer picture of the ulcerative lesion above the left pinna (B).
Fig. 3. Histopathology of the biopsy: skin with irregular epithelial hyperplasia, serousneutrophilic (arrows) and fibrotic foci with perivascular lymphocytic infiltrate and neutrophilic exudate (asterisk) (H&E, 20×, A; H& E, 60×, B).
It is important to note that the incidence peak of PG occurs in older patients (> 50 years, 44.4%), and predominantly in women [5]. Additionally, many patients present early lesions spreading despite preventive antibiotics and no bacterial growth from wound swabs [2,5]. Our patient presented the signals and symptoms with no association to other systemic diseases and was not responsive to clinical treatment. Interestingly, no single medical treatment is effective in PG patients [5]. Systemic steroids with or without immunosuppressive therapy are the gold standard, but other agents have been successfully reported, such as azathioprine, sulfasalazine, dapsone, thalidomide, colchicine, tacrolimus, methotrexate, intravenous immunoglobulin, systemic antibiotics (like tetracyclines and rifampicin), anti-TNF-α (tumor necrosis factor-alfa) – in cases of resistant PG – negative pressure therapy, and hyperbaric oxygen [1,4,5]. No previous reports of PG after stapedotomy have been described. In this case, the disease developed at the side of the second surgery. The lesion was initially managed as infections with antibiotics and removal of necrotic tissues, which made the ulcerative skin disease worse. Finally, after the prosthesis removal, patient evolved with good healing of the lesion.
Fig. 2. Histopathology of the biopsy: irregular epithelial hyperplasia, neutrophilic (arrows) and fibrotic foci with perivascular invasion (asterisk) (H&E, 2×).
[3]. Pathology is typically an ulcerative lesion with polynuclear neutrophil infiltrate and associated capillary network disorder (mainly, venous thrombosis), but it helps a little [2]. It has been associated with leukemia, inflammatory bowel disease, and induction by a trauma may be considered one of the pathogenic processes, as well as the surgical trauma of our patient [4]. PG has been associated as a complication of many surgical procedures. However, to the best of our knowledge, this is the first study to describe PG as complication of a stapedotomy. Interestingly, this fact happened after two years of the first surgery.
Conclusion Pyoderma gangrenosum is known to be initiated at the site of surgical trauma. Early diagnosis of PG is vital to prevent secondary 23
Otolaryngology Case Reports 6 (2018) 22–24
L.C.A. Sousa et al.
infection of the wound, and to avoid more aggressive treatment.
Appendix A. Supplementary data
Consent
Supplementary data related to this article can be found at http://dx. doi.org/10.1016/j.xocr.2017.12.001.
An informed consent was obtained previously to all procedures.
References
Conflict of interest
[1] Rothenburger M, Tjan TD, Schmid C, Schmidt C, Schwarz T, Scheld HH. Pyoderma gangrenosum after aortic valve replacement. Ann Thorac Surg 2001;71(1):349–51. [2] Kasper K, Manger B, Junger A, Reichert B, Sievers R, Herdtle S. Pyoderma gangrenosum following AICD implantation: differential diagnosis to necrotizing fasciitis. Anaesthesist 2012;61(1):47–51. [3] Kurniadi I, Imanishi H, Furukawa H, Sowa-Osako J, Tsuruta D. Case of facial pyoderma gangrenosum. J Dermatol 2016;43(11):1373–4. [4] Adšen E, Erduran F, Gürer MA. Pyoderma gangrenosum: a report of 27 patients. Int J Low Extrem Wounds 2016;15(2):148–54. [5] Wollina U. Pyoderma gangrenosum – a review. Orph J Rare Dis 2007;2:19–27.
The authors declare that they have no competing interests. Financial disclosure This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.
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