Journal of Dermatological Treatment
ISSN: 0954-6634 (Print) 1471-1753 (Online) Journal homepage: http://www.tandfonline.com/loi/ijdt20
Squamous cell carcinoma originating from cutaneous cysts: The Henry Ford Experience and review of the literature Jesse J. Veenstra, Samreen Choudhry, Richard J. Krajenta & Melody J. Eide To cite this article: Jesse J. Veenstra, Samreen Choudhry, Richard J. Krajenta & Melody J. Eide (2015): Squamous cell carcinoma originating from cutaneous cysts: The Henry Ford Experience and review of the literature, Journal of Dermatological Treatment To link to this article: http://dx.doi.org/10.3109/09546634.2015.1054779
Published online: 24 Jun 2015.
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Date: 05 November 2015, At: 19:09
http://informahealthcare.com/jdt ISSN: 0954-6634 (print), 1471-1753 (electronic) J Dermatolog Treat, Early Online: 1–4 ! 2015 Informa UK Ltd. DOI: 10.3109/09546634.2015.1054779
ORIGINAL ARTICLE
Squamous cell carcinoma originating from cutaneous cysts: The Henry Ford Experience and review of the literature Jesse J. Veenstra1, Samreen Choudhry2, Richard J. Krajenta3, and Melody J. Eide2,3 Department of Oncology, Wayne State University School of Medicine, Detroit, MI, USA, 2Department of Dermatology, Henry Ford Hospital, Detroit, MI, USA, and 3Department of Public Health Sciences, Henry Ford Hospital, Detroit, MI, USA
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1
Abstract
Keywords
Purpose: Malignant transformation to squamous cell carcinoma (SCC) arising within cutaneous epidermal cysts is a very rare phenomenon. We provide a series of new cases and an overview of the literature. We sought to define the prevalence of and characterize SCC arising within epidermal and pilar cysts. Patient and methods: We searched Henry Ford Health System (HFHS) non-melanoma skin cancer (NMSC) registry from 2005 to 2009 to identify cases of SCC arising from epidermal cysts. Results: We identified 1904 cases of epidermal cysts at our institution between 2005 and 2014. Of these, three cases of SCC arose from an epidermal cyst and one case of SCC developed from a pilar cyst. All lesions occurred below the waist with the exception of the pilar cyst on the scalp. Conclusions: Given the extremely low incidence, propensity of malignant lesions to become symptomatic and efficacy of treatment, we do not recommend routine excision of all epidermal cysts. Instead, we recommend excision and pathology for all symptomatic epidermal cysts, or those that rapidly grow, or do not respond to medical therapy.
Case series, epidermal cyst, literature review, pilar cyst, squamous cell carcinoma History Received 17 April 2015 Accepted 17 May 2015 Published online 24 June 2015
Abbreviations: HFHS: Henry Ford Health System; NMSC: non-melanoma skin cancer; SCC: squamous cell carcinoma
Introduction Both epidermal cysts and squamous cell carcinoma (SCC) are common skin lesions. Although very rare, malignant transformation in cutaneous epidermal cysts has been reported, including SCC, basal cell carcinoma, Paget’s disease, Bowen’s disease, mycosis fungoides, Merkel cell carcinoma and melanoma (1). We present four cases of SCC arising within an epidermal cyst.
Methods From 2005 to 2009, 1902 cases of SCC were identified from the Henry Ford Health System non-melanoma skin cancer (NMSC) registry (2). These cases were cross-referenced for ‘‘cyst’’ in the pathology report, which identified eight cases that were further interrogated by manual chart review. Two of the eight cases were found to represent true SCC arising from an epidermal cyst based upon pathology findings (cases 1 and 2). Searching beyond the established timeframe of the Henry Ford NMSC registry, two other cases of SCC arising from a ‘‘cyst’’ were identified after 2009 (cases 3 and 4). A brief description of the four cases is
Correspondence: Melody J. Eide, MD MPH, Department of Dermatology, 3031 W. Grand Blvd, Suite 800, Detroit, MI 48202, USA. E-mail: [email protected]
outlined in Table 1. Past medical history was non-significant and negative for previous skin cancer unless otherwise noted.
Results The first case was a 46-year-old female with a history of multiple sebaceous cysts previously removed, who presented with an asymptomatic 2 cm cyst on her left medial thigh that had been present for more than 12 months. The cyst had previously been treated with incision and drainage (I&D) and antibiotics 1 year prior. Excision was performed and pathology revealed an invasive well-differentiated SCC. Re-excision with 2 cm margins was completed without evidence of residual carcinoma. She has been recurrence free for 10 years. The second case was an 89-year-old female who presented with a painful, draining 4 cm cyst in her suprapubic region, which had been present greater than 1 month. The cyst was treated with I&D and antibiotics in the emergency department. Three days later, the cyst was examined in the office with extension of the incision due to inadequate drainage. Biopsy was performed and revealed superficial squamous epithelium with suggestion of SCC. Wide excision was performed and histology confirmed a well-differentiated SCC arising from an epidermoid cyst. She was recurrence free for 4 years before death due to natural causes. The third case was a 61-year-old male with a history of sarcoidosis and heart transplant taking tacrolimus, who presented
2
J. J. Veenstra et al.
J Dermatolog Treat, Early Online: 1–4
Table 1. Henry Ford cases of SCC arising from cyst.
Age
Sex
Location
Max cyst diameter (cm)
Duration (months)
Cyst presentation and characteristics Asymptomatic, I&D and antibiotics 1 year prior Became painful with discharge 3 weeks prior, I&D and antibiotics 3 d prior Slowly growing asymptomatic cyst
Epidermal cyst 46 F L medial thigh
2
412
89
F
Suprapubic
4
41
61
M
L lateral thigh
1.2
46
Pilar cyst 66 M
R scalp
1
412
Slowly growing, occasional discharge, painful at times
Pathology
Treatment
Outcome
Well diff.
Excision, re-excision
10 yr, no recurrence
Well diff.
Excision
4 yr, no recurrence
Well diff.
Excision, re-excision
8 mo, no recurrence
Well diff.
Excision, re-excision
4 mo, no recurrence
Downloaded by [University of Pennsylvania] at 19:09 05 November 2015
F, female; M, male; R, right; L, left; diff, differentiated; mo, month; yr, year.
with an asymptomatic 1.2 cm cyst on his left lateral thigh that had been slowly growing for more than 6 months. Routine excision was performed and pathology revealed a well-differentiated SCC arising from an epidermoid cyst. Although the specimen was well differentiated, a metastatic lesion could not be ruled out. Reexcision with 5 mm margins was performed without evidence of residual carcinoma. No evidence of metastatic disease was found on physical exam, and the patient opted for close monitoring with serial skin exams rather than imaging studies. He has been recurrence free without evidence of metastasis for 8 months. The fourth case was a 66-year-old male who presented with a slowly growing, occasionally painful 1 cm cyst on his right scalp, which had been present for greater than a year. Excision was performed and pathology revealed atypical squamous epithelium extending from the wall of a pilar cyst and invading the surrounding dermis. Mohs surgery was performed with 1.5 mm free-appearing margins. He has been recurrence free for 4 months. Although this case involved a pilar cyst, and not an epidermal cyst, we thought it was pertinent to include in the case series.
Discussion Squamous cell carcinoma (SCC) arising from a cutaneous epidermal cyst is an unusual phenomenon. While the incidence historically reported has been high (2.2%) (3), this has been disputed in part due to questionable histological classification (4,5). To confirm the diagnosis of SCC arising from an epidermal cyst, the specimen must undergo histopathologic analysis to be differentiated from SCC with cystic changes, which necessitates establishing the absence of contact between the tumor and epidermis (4). In 1999, Lopez-Rios et al. reviewed 27 case reports in the literature over 55 years, and found that only eight cases had well-characterized microscopic evidence consistent with SCC arising within a cutaneous epidermal cyst. Furthermore, only a single SCC arising from an epidermal cyst among 2485 cases examined at their institution was found (5). Similarly, AntonBadiola et al. reported only one SCC of the 3700 cutaneous epidermal inclusion cysts examined at their hospital over a 22-year period (4). Of the literature to date, cases of welldocumented SCC arising from epidermal cysts have an incidence ranging from 0.011 to 0.045% (4–6,7). A current review of the literature is summarized in Table 2.
Chronic inflammation is a well-known mechanism of malignant transformation and may play a crucial role in the development of SCC from longstanding epidermal cysts (26). This may occur in periodically inflamed lesions or even in asymptomatic lesions subjected to low levels of recurrent irritation (e.g. friction and pressure). In support of this, rapid growth of previously stable cysts in relation to trauma was found in three cases in the literature (8,12,14,16). While it is unlikely that a single traumatic event was exclusively responsible for the malignant transformation, given the brief period between trauma and diagnosis, it is plausible that the subsequent inflammation provided an environment favorable for malignant growth. Immunosuppression, actinic damage, and HPV infection are other etiological factors that have been proposed to be involved in malignant transformation of cysts; however, confirmation has yet to be reported (7). Of the cases we described, only one patient was immunosuppressed due to heart transplant, which likely contributed to the malignant outgrowth of the lesion due to compromised immunosurveillance.
Conclusions Although the majority of cases reported were successfully diagnosed and treated without complication, it is important to note that three cases reported had advanced disease with metastases and death within 5–10 months after initial presentation (11,12,18). Even though most of the documented cases presented with symptomatic lesions (e.g. recent growth, pain, erythema, drainage) our cases and several previous reports (1,13,17) demonstrate that clinically asymptomatic lesions may also harbor SCC, which suggests even the most benign appearing cysts may contain malignancy. However, given the extremely low incidence, propensity of malignant lesions to become symptomatic and efficacy of treatment, we do not recommend routine excision of all epidermal cysts. Instead, we recommend excision and histologic examination of the entire lesion for all large epidermal cysts, or those that begin rapidly growing, ulcerate, do not respond to medical therapy, or rapidly recur. In addition, these findings reinforce the need for routine histopathologic analysis of all excised lesions to rule out incipient malignancy, with re-excision if necessary. Further studies are warranted to determine long-term prognosis of these patients.
32
34
38
48
48
50 52
55 57
58
59
63
65
65
66 67
68
68
69
74
76
(9)
(10)
(11)
(12)
(13) (14)
(15) (16)
(17)
(18)
(19)
(20)
(4)
(5) (6)
(21)
(3)
(1)
(22)
(23)
Age
(8)
Ref.
F
M
F
M
M
M M
M
M
M
F
M
F M
M M
M
F
F
M
M
Sex
R labia majora
L proximal thigh
Cheek
R preauricular region
L axilla
L retroauricular region R temple
R mastoid region
L neck
L nose
L thigh
R ear
L gluteal region L gluteal region
Head L forehead
L gluteal region
R lower abdomen
R gluteal region
L post. head
L index finger
Location
12.5
15
0.9
3
4
3 3
2
9
2
7
2.5
9 6
1.5 NS
5
9.2
20
8
0.5
Max cyst diameter(cm)
Table 2. Literature overview – cases of SCC arising from epidermal cyst.
Unknown
480
18
NS
24
2 3
2
420
36
NS
132
6 4240
36 2
336
4120
4240
240
144
Duration (months) Initially hit with hammer 12 yrs ago, reinjured at presentation, developed non-healer ulcer Considerable growth in past 24 mo Considerable growth in past 24 mo, otherwise asymptomatic Considerable growth in past 6 mo, painful Progressive growth 3 mo after trauma to cyst Asymptomatic Bumped forehead 2 mo prior, progressive swelling, drainage, failed antibiotics NS Progressive swelling and pain 1 mo after trauma to cyst Cyst did not collapse well after I&D Progressive swelling and pain in past 3 mo Painful in past 8 weeks, purulent discharge Considerable growth and bloody discharge in past 6 mo Recurred multiple times after initial excisions Fluctuating mass Enlarging, inflamed, failed antibiotics Considerable growth in past 1 mo, otherwise asymptomatic Appeared to be infected, did not respond to I&D and antibiotics Slowly growing, otherwise asymptomatic Considerable growth in past 36 mo, recent serosanguinous discharge
Cyst presentation and characteristics
Excision Excision
Well diff. Well diff.
Excision
Excision, re-excision
Well diff. Well diff.
Excision
Mod. diff.
Well diff. Mod. dif.
Excision, re-excision, RT, amputation Excision Excision, re-excision
Undiff. Mod. diff.
Excision, RT
Well diff.
Excision
Excision Excision
Excision, inguinal dissection, RT Excision Excision
Excision, 5FU/cisplatin
Excision
Excision, inguinal dissection Excision
Well diff.
NS
Well diff. Well diff.
NS NS
Well diff.
Poorly diff.
NS
Excision, RT
Well diff.
Well diff.
Treatment Amputation, RT
Pathology
Downloaded by [University of Pennsylvania] at 19:09 05 November 2015
SCC arising within cysts (continued )
5 mo, no recurrence
24 mo, no recurrence
4 mo, no recurrence
NS
NS
36 mo, no recurrence 6 mo, no recurrence
18 mo, no recurrence
NS
Inguinal mets, death at 6 mo NS
NS
NS 6 mo, no recurrence
Systemic mets, death at 10vmo Systemic mets, death at 5 mo 9 mo, no recurrence NS
12 mo, no recurrence
NS
NS
Outcome
DOI: 10.3109/09546634.2015.1054779
3
J. J. Veenstra et al. 10 mo, no recurrence 24–72 mo, no recurrence
Declaration of interest The authors report that they have no conflicts of interest.
Excision, re-excision NS
References
NS NS 3.5 NS R forehead Face/neck 2 Trunk 3 F2 F M3 92 21–80 (25) (7)
NS, not stated; RT, radiotherapy; F, female; M, male; R, right; L, left; diff, differentiated; Mod, moderately; mo, month; yr, year
Poorly diff. Well diff.
NS Excision Well diff. 276 4.1 Scrotum 83 (24)
M
Pathology Cyst presentation and characteristics Duration (months) Max cyst diameter(cm) Location Sex Age Ref.
Table 2. Continued
Downloaded by [University of Pennsylvania] at 19:09 05 November 2015
Treatment
Outcome
J Dermatolog Treat, Early Online: 1–4
Considerable growth over an unknown period Ruptured 2 d prior to presentation Gradually enlarging 5 cases, NS
4
1. Anastasios KV. Malignant transformation in a typical epidermal cutaneous cyst. J Med Cases. 2012;3:254–6. 2. Eide MJ, Tuthill JM, Krajenta RJ, et al. Validation of claims data algorithms to identify nonmelanoma skin cancer. J Invest Dermatol. 2012;132:2005–9. 3. Bauer BS, Lewis Jr VL. Carcinoma arising in sebaceous and epidermoid cysts. Ann Plastic Surg. 1980;5:222–6. 4. Anton-Badiola I, San Miguel-Fraile P, Peteiro-Cancelo A, Ortiz-Rey JA. Squamous cell carcinoma arising on an epidermal inclusion cyst: a case presentation and review of the literature. Actas Dermosifiliograficas. 2010;101:349–53. 5. Lopez-Rios F, Rodriguez-Peralto JL, Castano E, Benito A. Squamous cell carcinoma arising in a cutaneous epidermal cyst: case report and literature review. Am J Dermatopathol. 1999;21:174–7. 6. Cameron DS, Hilsinger Jr RL. Squamous cell carcinoma in an epidermal inclusion cyst: case report. Otolaryngol – Head Neck Surg: Off J Am Acad Otolaryngol – Head Neck Surg. 2003;129:141–3. 7. Morgan MB, Stevens GL, Somach S, Tannenbaum M. Carcinoma arising in epidermoid cyst: a case series and aetiological investigation of human papillomavirus. Br J Dermatol. 2001;145:505–6. 8. Davies MS, Nicholson AG, Southern S, Moss AH. Squamous cell carcinoma arising in a traumatically induced epidermal cyst. Injury. 1994;25:116–17. 9. Miller JM. Squamous cell carcinoma arising in an epidermal cyst. Arch Dermatol. 1981;117:683. 10. Debaize S, Gebhart M, Fourrez T, et al. Squamous cell carcinoma arising in a giant epidermal cyst: a case report. Acta Chirurgica Belgica. 2002;102:196–8. 11. Nemoto I, Shibaki A, Aoyagi S, et al. Aggressive squamous cell carcinoma developing in a giant epidermal cyst of the abdomen. Int J Dermatol. 2006;45:1444–6. 12. Jehle KS, Shakir AJ, Sayegh ME. Squamous cell carcinoma arising in an epidermoid cyst. Br J Hosp Med. 2007;68:446. 13. Ziadi S, Trimeche M, Hammedi F, et al. Squamous cell carcinoma arising from an epidermal inclusion cyst: a case report. N Am J Med Sci. 2010;2:46–7. 14. Davidson TM, Bone RC, Kiessling PJ. Epidermoid carcinoma arising from within an epidermoid inclusion cyst. Ann Otol Rhinol Laryngol. 1976;85:417–18. 15. Shah LK, Rane SS, Holla VV. A case of squamous cell carcinoma arising in an epidermal cyst. Indian J Pathol Microbiol. 1989;32: 138–40. 16. Wong TH, Khoo AK, Tan PH, Ong BH. Squamous cell carcinoma arising in a cutaneous epidermal cyst – a case report. Ann Acad Med, Singapore. 2000;29:757–9. 17. Yaffe HS. Squamous cell carcinoma arising in an epidermal cyst. Arch Dermatol. 1982;118:961. 18. Arianayagam S, Jayalakshmi P. Malignant epidermal cyst: a case report. Malaysian J Pathol. 1987;9:89–91. 19. Cappello ZJ, Kasdan ML, Augenstein AC, Shaheen SP. Squamous cell carcinoma in an epidermoid cyst. Eplasty. 2013;13:ic44. 20. Tokunaga M, Toya M, Endo Y, et al. A case of an undifferentiated squamous cell carcinoma arising from an epidermal cyst. Case Rep Dermatol Med. 2013;2013:469516. 21. Lin CY, Jwo SC. Squamous cell carcinoma arising in an epidermal inclusion cyst. Chang Gung Med J. 2002;25:279–82. 22. Chiu MY, Ho ST. Squamous cell carcinoma arising from an epidermal cyst. Hong Kong Med J ¼ Xianggang yi xue za zhi/Hong Kong. Acad Med. 2007;13:482–4. 23. Sumi Y, Yamamoto N, Kiyosawa T. Squamous cell carcinoma arising in a giant epidermal cyst of the perineum: a case report and literature review. J Plast Surg Hand Surg. 2012;46:209–11. 24. Yeh L-P, Kuo-Sheng L. Squamous cell carcinoma arising from an epidermal cyst of the scrotum. Tzu Chi Med J. 2013;25:117–18. 25. Malone JC, Sonnier GB, Hughes AP, Hood AF. Poorly differentiated squamous cell carcinoma arising within an epidermoid cyst. Int J Dermatol. 1999;38:556–8. 26. Coussens LM, Werb Z. Inflammation and cancer. Nature. 2002;420: 860–7.
ISSN: 0954-6634 (Print) 1471-1753 (Online) Journal homepage: http://www.tandfonline.com/loi/ijdt20
Squamous cell carcinoma originating from cutaneous cysts: The Henry Ford Experience and review of the literature Jesse J. Veenstra, Samreen Choudhry, Richard J. Krajenta & Melody J. Eide To cite this article: Jesse J. Veenstra, Samreen Choudhry, Richard J. Krajenta & Melody J. Eide (2015): Squamous cell carcinoma originating from cutaneous cysts: The Henry Ford Experience and review of the literature, Journal of Dermatological Treatment To link to this article: http://dx.doi.org/10.3109/09546634.2015.1054779
Published online: 24 Jun 2015.
Submit your article to this journal
Article views: 30
View related articles
View Crossmark data
Full Terms & Conditions of access and use can be found at http://www.tandfonline.com/action/journalInformation?journalCode=ijdt20 Download by: [University of Pennsylvania]
Date: 05 November 2015, At: 19:09
http://informahealthcare.com/jdt ISSN: 0954-6634 (print), 1471-1753 (electronic) J Dermatolog Treat, Early Online: 1–4 ! 2015 Informa UK Ltd. DOI: 10.3109/09546634.2015.1054779
ORIGINAL ARTICLE
Squamous cell carcinoma originating from cutaneous cysts: The Henry Ford Experience and review of the literature Jesse J. Veenstra1, Samreen Choudhry2, Richard J. Krajenta3, and Melody J. Eide2,3 Department of Oncology, Wayne State University School of Medicine, Detroit, MI, USA, 2Department of Dermatology, Henry Ford Hospital, Detroit, MI, USA, and 3Department of Public Health Sciences, Henry Ford Hospital, Detroit, MI, USA
Downloaded by [University of Pennsylvania] at 19:09 05 November 2015
1
Abstract
Keywords
Purpose: Malignant transformation to squamous cell carcinoma (SCC) arising within cutaneous epidermal cysts is a very rare phenomenon. We provide a series of new cases and an overview of the literature. We sought to define the prevalence of and characterize SCC arising within epidermal and pilar cysts. Patient and methods: We searched Henry Ford Health System (HFHS) non-melanoma skin cancer (NMSC) registry from 2005 to 2009 to identify cases of SCC arising from epidermal cysts. Results: We identified 1904 cases of epidermal cysts at our institution between 2005 and 2014. Of these, three cases of SCC arose from an epidermal cyst and one case of SCC developed from a pilar cyst. All lesions occurred below the waist with the exception of the pilar cyst on the scalp. Conclusions: Given the extremely low incidence, propensity of malignant lesions to become symptomatic and efficacy of treatment, we do not recommend routine excision of all epidermal cysts. Instead, we recommend excision and pathology for all symptomatic epidermal cysts, or those that rapidly grow, or do not respond to medical therapy.
Case series, epidermal cyst, literature review, pilar cyst, squamous cell carcinoma History Received 17 April 2015 Accepted 17 May 2015 Published online 24 June 2015
Abbreviations: HFHS: Henry Ford Health System; NMSC: non-melanoma skin cancer; SCC: squamous cell carcinoma
Introduction Both epidermal cysts and squamous cell carcinoma (SCC) are common skin lesions. Although very rare, malignant transformation in cutaneous epidermal cysts has been reported, including SCC, basal cell carcinoma, Paget’s disease, Bowen’s disease, mycosis fungoides, Merkel cell carcinoma and melanoma (1). We present four cases of SCC arising within an epidermal cyst.
Methods From 2005 to 2009, 1902 cases of SCC were identified from the Henry Ford Health System non-melanoma skin cancer (NMSC) registry (2). These cases were cross-referenced for ‘‘cyst’’ in the pathology report, which identified eight cases that were further interrogated by manual chart review. Two of the eight cases were found to represent true SCC arising from an epidermal cyst based upon pathology findings (cases 1 and 2). Searching beyond the established timeframe of the Henry Ford NMSC registry, two other cases of SCC arising from a ‘‘cyst’’ were identified after 2009 (cases 3 and 4). A brief description of the four cases is
Correspondence: Melody J. Eide, MD MPH, Department of Dermatology, 3031 W. Grand Blvd, Suite 800, Detroit, MI 48202, USA. E-mail: [email protected]
outlined in Table 1. Past medical history was non-significant and negative for previous skin cancer unless otherwise noted.
Results The first case was a 46-year-old female with a history of multiple sebaceous cysts previously removed, who presented with an asymptomatic 2 cm cyst on her left medial thigh that had been present for more than 12 months. The cyst had previously been treated with incision and drainage (I&D) and antibiotics 1 year prior. Excision was performed and pathology revealed an invasive well-differentiated SCC. Re-excision with 2 cm margins was completed without evidence of residual carcinoma. She has been recurrence free for 10 years. The second case was an 89-year-old female who presented with a painful, draining 4 cm cyst in her suprapubic region, which had been present greater than 1 month. The cyst was treated with I&D and antibiotics in the emergency department. Three days later, the cyst was examined in the office with extension of the incision due to inadequate drainage. Biopsy was performed and revealed superficial squamous epithelium with suggestion of SCC. Wide excision was performed and histology confirmed a well-differentiated SCC arising from an epidermoid cyst. She was recurrence free for 4 years before death due to natural causes. The third case was a 61-year-old male with a history of sarcoidosis and heart transplant taking tacrolimus, who presented
2
J. J. Veenstra et al.
J Dermatolog Treat, Early Online: 1–4
Table 1. Henry Ford cases of SCC arising from cyst.
Age
Sex
Location
Max cyst diameter (cm)
Duration (months)
Cyst presentation and characteristics Asymptomatic, I&D and antibiotics 1 year prior Became painful with discharge 3 weeks prior, I&D and antibiotics 3 d prior Slowly growing asymptomatic cyst
Epidermal cyst 46 F L medial thigh
2
412
89
F
Suprapubic
4
41
61
M
L lateral thigh
1.2
46
Pilar cyst 66 M
R scalp
1
412
Slowly growing, occasional discharge, painful at times
Pathology
Treatment
Outcome
Well diff.
Excision, re-excision
10 yr, no recurrence
Well diff.
Excision
4 yr, no recurrence
Well diff.
Excision, re-excision
8 mo, no recurrence
Well diff.
Excision, re-excision
4 mo, no recurrence
Downloaded by [University of Pennsylvania] at 19:09 05 November 2015
F, female; M, male; R, right; L, left; diff, differentiated; mo, month; yr, year.
with an asymptomatic 1.2 cm cyst on his left lateral thigh that had been slowly growing for more than 6 months. Routine excision was performed and pathology revealed a well-differentiated SCC arising from an epidermoid cyst. Although the specimen was well differentiated, a metastatic lesion could not be ruled out. Reexcision with 5 mm margins was performed without evidence of residual carcinoma. No evidence of metastatic disease was found on physical exam, and the patient opted for close monitoring with serial skin exams rather than imaging studies. He has been recurrence free without evidence of metastasis for 8 months. The fourth case was a 66-year-old male who presented with a slowly growing, occasionally painful 1 cm cyst on his right scalp, which had been present for greater than a year. Excision was performed and pathology revealed atypical squamous epithelium extending from the wall of a pilar cyst and invading the surrounding dermis. Mohs surgery was performed with 1.5 mm free-appearing margins. He has been recurrence free for 4 months. Although this case involved a pilar cyst, and not an epidermal cyst, we thought it was pertinent to include in the case series.
Discussion Squamous cell carcinoma (SCC) arising from a cutaneous epidermal cyst is an unusual phenomenon. While the incidence historically reported has been high (2.2%) (3), this has been disputed in part due to questionable histological classification (4,5). To confirm the diagnosis of SCC arising from an epidermal cyst, the specimen must undergo histopathologic analysis to be differentiated from SCC with cystic changes, which necessitates establishing the absence of contact between the tumor and epidermis (4). In 1999, Lopez-Rios et al. reviewed 27 case reports in the literature over 55 years, and found that only eight cases had well-characterized microscopic evidence consistent with SCC arising within a cutaneous epidermal cyst. Furthermore, only a single SCC arising from an epidermal cyst among 2485 cases examined at their institution was found (5). Similarly, AntonBadiola et al. reported only one SCC of the 3700 cutaneous epidermal inclusion cysts examined at their hospital over a 22-year period (4). Of the literature to date, cases of welldocumented SCC arising from epidermal cysts have an incidence ranging from 0.011 to 0.045% (4–6,7). A current review of the literature is summarized in Table 2.
Chronic inflammation is a well-known mechanism of malignant transformation and may play a crucial role in the development of SCC from longstanding epidermal cysts (26). This may occur in periodically inflamed lesions or even in asymptomatic lesions subjected to low levels of recurrent irritation (e.g. friction and pressure). In support of this, rapid growth of previously stable cysts in relation to trauma was found in three cases in the literature (8,12,14,16). While it is unlikely that a single traumatic event was exclusively responsible for the malignant transformation, given the brief period between trauma and diagnosis, it is plausible that the subsequent inflammation provided an environment favorable for malignant growth. Immunosuppression, actinic damage, and HPV infection are other etiological factors that have been proposed to be involved in malignant transformation of cysts; however, confirmation has yet to be reported (7). Of the cases we described, only one patient was immunosuppressed due to heart transplant, which likely contributed to the malignant outgrowth of the lesion due to compromised immunosurveillance.
Conclusions Although the majority of cases reported were successfully diagnosed and treated without complication, it is important to note that three cases reported had advanced disease with metastases and death within 5–10 months after initial presentation (11,12,18). Even though most of the documented cases presented with symptomatic lesions (e.g. recent growth, pain, erythema, drainage) our cases and several previous reports (1,13,17) demonstrate that clinically asymptomatic lesions may also harbor SCC, which suggests even the most benign appearing cysts may contain malignancy. However, given the extremely low incidence, propensity of malignant lesions to become symptomatic and efficacy of treatment, we do not recommend routine excision of all epidermal cysts. Instead, we recommend excision and histologic examination of the entire lesion for all large epidermal cysts, or those that begin rapidly growing, ulcerate, do not respond to medical therapy, or rapidly recur. In addition, these findings reinforce the need for routine histopathologic analysis of all excised lesions to rule out incipient malignancy, with re-excision if necessary. Further studies are warranted to determine long-term prognosis of these patients.
32
34
38
48
48
50 52
55 57
58
59
63
65
65
66 67
68
68
69
74
76
(9)
(10)
(11)
(12)
(13) (14)
(15) (16)
(17)
(18)
(19)
(20)
(4)
(5) (6)
(21)
(3)
(1)
(22)
(23)
Age
(8)
Ref.
F
M
F
M
M
M M
M
M
M
F
M
F M
M M
M
F
F
M
M
Sex
R labia majora
L proximal thigh
Cheek
R preauricular region
L axilla
L retroauricular region R temple
R mastoid region
L neck
L nose
L thigh
R ear
L gluteal region L gluteal region
Head L forehead
L gluteal region
R lower abdomen
R gluteal region
L post. head
L index finger
Location
12.5
15
0.9
3
4
3 3
2
9
2
7
2.5
9 6
1.5 NS
5
9.2
20
8
0.5
Max cyst diameter(cm)
Table 2. Literature overview – cases of SCC arising from epidermal cyst.
Unknown
480
18
NS
24
2 3
2
420
36
NS
132
6 4240
36 2
336
4120
4240
240
144
Duration (months) Initially hit with hammer 12 yrs ago, reinjured at presentation, developed non-healer ulcer Considerable growth in past 24 mo Considerable growth in past 24 mo, otherwise asymptomatic Considerable growth in past 6 mo, painful Progressive growth 3 mo after trauma to cyst Asymptomatic Bumped forehead 2 mo prior, progressive swelling, drainage, failed antibiotics NS Progressive swelling and pain 1 mo after trauma to cyst Cyst did not collapse well after I&D Progressive swelling and pain in past 3 mo Painful in past 8 weeks, purulent discharge Considerable growth and bloody discharge in past 6 mo Recurred multiple times after initial excisions Fluctuating mass Enlarging, inflamed, failed antibiotics Considerable growth in past 1 mo, otherwise asymptomatic Appeared to be infected, did not respond to I&D and antibiotics Slowly growing, otherwise asymptomatic Considerable growth in past 36 mo, recent serosanguinous discharge
Cyst presentation and characteristics
Excision Excision
Well diff. Well diff.
Excision
Excision, re-excision
Well diff. Well diff.
Excision
Mod. diff.
Well diff. Mod. dif.
Excision, re-excision, RT, amputation Excision Excision, re-excision
Undiff. Mod. diff.
Excision, RT
Well diff.
Excision
Excision Excision
Excision, inguinal dissection, RT Excision Excision
Excision, 5FU/cisplatin
Excision
Excision, inguinal dissection Excision
Well diff.
NS
Well diff. Well diff.
NS NS
Well diff.
Poorly diff.
NS
Excision, RT
Well diff.
Well diff.
Treatment Amputation, RT
Pathology
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SCC arising within cysts (continued )
5 mo, no recurrence
24 mo, no recurrence
4 mo, no recurrence
NS
NS
36 mo, no recurrence 6 mo, no recurrence
18 mo, no recurrence
NS
Inguinal mets, death at 6 mo NS
NS
NS 6 mo, no recurrence
Systemic mets, death at 10vmo Systemic mets, death at 5 mo 9 mo, no recurrence NS
12 mo, no recurrence
NS
NS
Outcome
DOI: 10.3109/09546634.2015.1054779
3
J. J. Veenstra et al. 10 mo, no recurrence 24–72 mo, no recurrence
Declaration of interest The authors report that they have no conflicts of interest.
Excision, re-excision NS
References
NS NS 3.5 NS R forehead Face/neck 2 Trunk 3 F2 F M3 92 21–80 (25) (7)
NS, not stated; RT, radiotherapy; F, female; M, male; R, right; L, left; diff, differentiated; Mod, moderately; mo, month; yr, year
Poorly diff. Well diff.
NS Excision Well diff. 276 4.1 Scrotum 83 (24)
M
Pathology Cyst presentation and characteristics Duration (months) Max cyst diameter(cm) Location Sex Age Ref.
Table 2. Continued
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Treatment
Outcome
J Dermatolog Treat, Early Online: 1–4
Considerable growth over an unknown period Ruptured 2 d prior to presentation Gradually enlarging 5 cases, NS
4
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