Table of Contents  
CASE REPORT
Year : 2016  |  Volume : 12  |  Issue : 2  |  Page : 66-68

Florid pseudoepitheliomatous transformation in a chronic pressure ulcer


Department of Plastic and Reconstructive Surgery, National Orthopaedic Hospital Dala, Kano, Nigeria

Date of Web Publication17-Mar-2017

Correspondence Address:
Dafiewhare O Rex
Department of Plastic and Reconstructive Surgery, National Orthopaedic Hospital, Kano
Nigeria
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0794-9316.202441

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  Abstract 

The case is reported of a florid manifestation of pseudoepitheliomatous hyperplasia in a chronic recurrent right ischial pressure ulcer of 26 years duration in a 60-year-old man with post-traumatic parapereisis. The peculiar characteristics seen were an incidental finding at surgery. Associated left ischial and sacral ulcers did not have these changes. Histological diagnosis of a benign lesion was at variance with a high clinical suspicion of malignant transformation based on the morphological appearance and chronicity. He had ulcer excision and primary flap cover. He was carefully followed up for 4 years and has been without recurrence or disease progression after excision and primary flap cover.

Keywords: Malignant transformation, Marjolin’s ulcer, pre-malignant, pressure ulcer, pseudoepithelomatosis, squamous cell carcinoma


How to cite this article:
Rex DO. Florid pseudoepitheliomatous transformation in a chronic pressure ulcer. Nigerian J Plast Surg 2016;12:66-8

How to cite this URL:
Rex DO. Florid pseudoepitheliomatous transformation in a chronic pressure ulcer. Nigerian J Plast Surg [serial online] 2016 [cited 2024 Mar 28];12:66-8. Available from: https://www.njps.org/text.asp?2016/12/2/66/202441


  Introduction Top


Pseudoepitheliomatous hyperplasia (PEH), also called pseudo carcinomatous hyperplasia, is described as a reactive epithelial hyperplasia that mimics the epithelial hyperplasia of squamous cell carcinoma (SCC). It is a benign lesion suggesting chronic irritation. It has been described as being more of a reaction than a lesion per se.[1] It is commonly seen in lesions affecting epidermal cell linings such as the upper digestive tract and cutaneous lesions. The diagnosis is histological requiring full depth tissue biopsy.[2],[3] It is not classified as a pre-malignant condition but may be present in lesions undergoing transformation to SCC.[4],[5] The case is presented of a florid manifestation in a long standing ischial pressure ulcer. An online search did not yield similar reported case in literature.


  Case history Top


A 60-year-old male trader with post-traumatic parapareisis of 26 years duration was referred from an orthopedic unit in our facility to the plastic surgery unit. The referring team had observed pressure ulcers in the sacral and right ischial regions. The ulcers were said to be recurrent over the past 26 years with many episodes of healing and breakdown. They ulcers had been managed at home with various pharmaceutical agents procured at a chemist shop, but the exact nature of which could not be ascertained. The ulcers were not a matter of much concern to him as they were painless, and he was able to continue his job as a textile trader. He was only on admission for amputation of the right leg following ascending gangrene complicating flame burns sustained 6 months prior to admission. He developed post-traumatic parapereisis 26 years ago secondary to a road traffic accident and has been unable to walk. He was also diagnosed diabetic during work up for the amputation. Blood sugar levels were controlled at the time of referral. There was no history of smoking or use of alcohol.

Examination revealed an elderly man with a small body frame. He was not pale and did not look chronically ill. There was an oblique right sacral ulcer extending towards the anal verge and measuring 7 cm × 3 cm. There was also a hypertrophic, dyschromic scar overlying an ulcer cavity in the left ischial area with a 5 mm central opening. No active discharge was observed from this opening [Figure 1]. Sensory level was at L3. Lower limb power was 2/5. He was worked up for ulcer excision and flap cover. At surgery, excision of the left ischial ulcer revealed an ulcer cavity 6 cm × 5 cm × 4 cm lined by pale colored filiform wart-like excrescences [Figure 2]. The ulcer was excised with a 5 mm margin and closed with a posterior thigh cutaneous flap over an active drain. The sacral ulcer was also excised and closed directly. The specimen was sent for histology. Histology revealed sections of ulcerated skin tissue showing acanthotic squamous epithelium exhibiting hyperkeratosis and parakeratosis with papillomatous and in bending of the rete pegs. The underlying fibrovascular stroma showed focal infiltrates of chronic inflammatory cells. There was no evidence of malignancy in the received specimen. A diagnosis of PEH was made.
Figure 1: Pre-operative appearance

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Figure 2: Intra-operative appearance

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The wounds broke down post-operatively further heightening the concerns of possible malignant transformation. The patient was again worked up and had further wound excision and flap closure. Specimens taken for histology a second time showed no evidence of malignancy.

All wounds healed subsequently without event [Figure 3]. The patient has remained well with no recurrence 4 years on follow-up.
Figure 3: Post-operative appearance

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  Discussion Top


PEH is a known mimicker of SCC.[6] Care must be taken not to misdiagnose it as well-differentiated SCC. The long-standing nature and gross vegetative changes seen at surgery and subsequent wound breakdown raised the suspicion of a malignant transformation[6] into Marjolin’s ulcer in this patient. SCC is commonly diagnosed at an advanced stage due to delays in making the diagnosis.[4] This was also a concern for this patient who had lived with this recurrent ulcer for 26 years. There were no clinical stigmata associated with malignancy. A diagnosis of PEH can be difficult even for the experienced histopathologist. PEH shares many similarities with well-differentiated SCC. Poorly cut or shaved specimens may be the cause of erroneous judgment.[7],[8],[9] Full thickness epithelial excision or multiple site biopsies make for more accurate diagnosis.[5] Repeat biopsies with second opinions may give a clearer picture.[3] This patient had the benefit of having the ischial ulcer totally excised for histological examination.

PEH can occur when there is prolonged irritation of epithelium like in chronic infections and infestations.[10],[11] Prolonged use of unknown agents for dressing the ulcers at home and neighborhood “chemists” (medicine stores) which may be irritating in nature could have contributed to the reactive changes seen in the ischial ulcer.[1]

PEH typically demonstrates exuberant down growing of rete pegs into but not beyond the basal lamina (pseudo invasion), the cells are mature, there is non- or mild dysplasia and dyskeratosis[1],[9],[12] SCC typically demonstrates invasion of basal lamina into the dermis, high grade nuclear atypia, mitotic figures, benign looking to anaplastic cells, and keratin pearls. This description may not be clear-cut. Local invasion may be missed and passed on for PEH while PEH may exhibit mitotic-like activity and some degree of atypia and passed off as malignancy.[13] Genetic studies are helping to provide answers; reduced expression of E-cadherin and increased expression of MM-9 (both associated with invasion) has been observed in SCC.[7],[14] P53 elaboration held some promise in detecting malignancy[2] but it is probably more important as a marker of immature cells than a marker of malignancy.[7],[15] Su et al.[13] demonstrated distinct differences in the expression of the genes C15orf48 and KRT9 with high accuracy in a limited study. (The roles of these genes in tumor genesis were unclear.) Genetic studies are, however, both expensive and many times not clinically practicable. Careful histological differentiation prevented unnecessary subjection of this patient to the psychological upheavals of being diagnosed with a malignancy and subjection to anticancer regimens.[2],[3]


  Conclusion Top


PEH continues to be a pathological entity of curious interest. It is probably under reported in plastic surgery as a specialty. Subjecting a patient to radical surgeries and potentially injurious anticancer regimens without histological confirmation for a non-malignant condition would raise ethical questions. It may be noteworthy to add that conversely, care is needed not to overlook a well-differentiated SCC in a patient. This goes to underscore the importance of a well-equipped histopathology unit and trained personnel.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Sarangarajan R, Vedam VK, Sivadas G, Krishnaraj R, Sarangarajan A, Shanmugam KT. Pseudoepitheliomatous hyperplasia: Relevance in oral pathology. J Int Oral Health 2015;7:132-6.  Back to cited text no. 1
    
2.
Choi KH, Lim JH, Lee JH, Kwon KS, Lee H, Park HS et al. Expression of claudin-1, p53 and E-cadherin in pseudoepitheliomatous hyperplasia and squamous cell carcinoma of the head and neck. Korean J Pathol 2008;42:287-93.  Back to cited text no. 2
    
3.
Suvadip C, Preeti RC, Deepak A, Shreyas S. Pseudoepitheliomatous hyperplasia: A clinical entity mistaken for squamous cell carcinoma. J Cutan Aesthet Surg 2014;7:232-4.  Back to cited text no. 3
    
4.
Vanessa OZ, Aline MA, Renata FD, Nurimar CF, Tullia CM, Juan PM. Marjolin’s ulcer: A twelve-case report. An Bras Dermatol 2006;81:355-8.  Back to cited text no. 4
    
5.
Kingsley O, Otene IC. Marjolin’s ulcers: A review. Niger Health J 2011;11:107-11.  Back to cited text no. 5
    
6.
Han JS, Lee SW, Suh KH, Kim SY, Hyun JJ, Jung SW et al. Pseudoepitheliomatous hyperplasia mimicking SCC in a patient with esophageal stricture. Korean J Gastroenterol 2014;63:366-8.  Back to cited text no. 6
    
7.
Teresa P, Maria GZ, Doriana M, Luigi S. Pseudoepitheliomatous hyperplasia arising from hypertrophic lichen planus mimicking squamous cell carcinoma: Limited value of immunohistochemistry. Acta Dermatovenerol Croat 2012;20:112-5.  Back to cited text no. 7
    
8.
Sami PM, Rose C, Owen W, Marie-Jo O, Dorothée BD, Annie L. Squamous cell carcinoma of the lip: Case report and review of the literature. J Clin Oncol 2011;29:e549-52.  Back to cited text no. 8
    
9.
Zarovnaya E, Black C. Distinguishing pseudoepitheliomatous hyperplasia from squamous cell carcinoma in mucosal biopsy specimens from the head and neck. Arch Pathol Lab Med 2005;129:1032-6.  Back to cited text no. 9
    
10.
Michael S, Gail LW. Clinical and laboratory update on blastomycosis. Clin Microbiol Rev 2010;23:367-81.  Back to cited text no. 10
    
11.
Heukelbach J, Sahebali S, Van Marck E, Sabóia Moura RC, Feldmeier H. An unusual case of ectopic tungiasis with pseudoepitheliomatous hyperplasia. Braz J Infect Dis 2004;8:465-8.  Back to cited text no. 11
    
12.
Tan KB, Tan SH, Aw DC, Jaffar H, Lim TC, Lee SJ et al. Simulators of squamous cell carcinoma of the skin: Diagnostic challenges on small biopsies and clinicopathological correlation. J Skin Cancer 2013;2013:752864. doi: 10.1155/2013/752864  Back to cited text no. 12
    
13.
Su A, Ra S, Li X, Zhou J, Binder S. Differentiating cutaneous squamous cell carcinoma and pseudoepitheliomatous hyperplasia by multiplex qRT-PCR. Mod Pathol 2013;26:1433-7.  Back to cited text no. 13
    
14.
Tack KY, Kyoung MK, Sang JN, Jun SB, Kyu YJ, Myoung JC et al. Expression of E-cadherin, cortactin and MMP-9 in pseudoepitheliomatous hyperplasia and squamous cell carcinoma of the head and neck: Their relationships with clinicopathologic factors and prognostic implication. Korean J Pathol 2012;46:331-40.  Back to cited text no. 14
    
15.
Lee YS, Teh M. p53 expression in pseudoepitheliomatous hyperplasia, keratoacanthoma, and squamous cell carcinoma of skin. Cancer 1994;73:2317-23.  Back to cited text no. 15
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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