A Case of Basal Cell Carcinoma Arising in Bilateral
Mastoidectomy Scars
CYLBURN E. SODEN, JR., MD, MA, DOUGLAS J. FIFE, MD,
AND
ALEXANDER MILLER, MD
The authors have indicated no significant interest with commercial supporters.
T
he first published report of a mastoidectomy
occurred nearly 350 years ago. This surgery
used to be a common treatment of chronic and
suppurative infections in the mastoid air cells.
Such infection usually resulted from chronic
otitis media that spread to the nearby mastoid
process. Mastoidectomy is now seldom needed,
because these infections are commonly treated
with antibiotics.
An extensive review of the literature revealed no
previously reported cases of bilateral primary basal
cell carcinoma (BCC) arising within mastoidectomy
scars.1 However, one previous report has been published documenting the occurrence of bilateral BCC
involving both external auditory canals, both middle
ear clefts, and both facial nerves. Here we report a
case of bilateral BCC arising in the scars of a previous bilateral mastoidectomy performed for the
treatment of chronic otitis media.
on the exposed skin of the scalp and upper
extremities. He had produced only two BCCs,
and both of these occurred deep within the
conical depression of each of the mastoidectomy scars, which were in a sun-shielded postauricular location.
The first BCC (nodular but clinically poorly
defined) occurred in the right postauricular location
in 1998 (Figure 1) and required excision with one
stage of Mohs surgery, leaving a 1.5- 1.1-cm defect
that healed nicely by secondary intention (Figure 2).
The second, and larger, infiltrating BCC was identified in the left mastoidectomy scar in late 2006
(Figure 3). This poorly defined tumor required four
stages of Mohs surgical excision, leaving a 4.2- 1.6-cm defect that successfully healed after a partial
flap reconstruction and secondary intention healing
Case Report
In 1932, at age 4, a fair-skinned 80-year-old man
had bilateral mastoidectomies performed using a
chisel and hammer technique in Vienna, Austria, for
pointing abscesses resulting from otitis media. Both
sites healed by secondary intention.
Throughout life, this patient had substantial sun
exposure, resulting in photodamaged skin with
actinic keratoses, and over the previous 10 years,
squamous cell carcinomas (SCCs) had generated
Figure 1. Nodular basal cell carcinoma in right mastoidectomy scar.
All authors are affiliated with the Department of Dermatology, University of California at Irvine, Orange, California
& 2008 by the American Society for Dermatologic Surgery, Inc. Published by Wiley Periodicals, Inc. ISSN: 1076-0512 Dermatol Surg 2009;35:135–138 DOI: 10.1111/j.1524-4725.2008.34394.x
135
BASAL CELL CARCINOMA ARISING IN MASTOIDECTOMY SCARS
Figure 2. Right posterior auricular Mohs defect.
(Figure 4). There has been no recurrence of either
BCC.
Figure 4. Left posterior auricular Mohs defect.
Discussion
Malignant tumors of the mastoid and middle ear
account for 5% to 26% of all ear neoplasms, making
them relatively uncommon.2,3 Of these neoplasms,
SCC tends to be the most prevalent,2,3 although its
bilateral occurrence is infrequent. Only two cases of
Figure 3. Infiltrating basal cell carcinoma in left mastoidectomy scar.
136
D E R M AT O L O G I C S U R G E RY
bilateral SCC and one case of bilateral BCC of the
mastoid have been reported.1,4
BCC arising in surgical scars is exceedingly rare,
with only 11 prior cases reported (Table 1).5–14 They
have occurred in patients aged 41 to 69, and the
latency period ranges from 9 months to 67 years
after the original surgery. BCCs are more commonly
reported in vaccination sites and burn scars. In burn
scars, they occur 7 to 10 times less frequently than
SCC.15,16 An extensive review of the literature revealed no reported cases of a primary BCC arising
within old mastoidectomy scars. There were also no
reports of a primary SCC arising within these scars,
an unexpected finding given that SCCs have a higher
frequency of occurrence within chronic wounds or
scars than BCCs.15,16 The etiology of the apparent
association between trauma and BCC has yet to be
elucidated but may be related to inflammation and
growth factors involved in wound healing.
The clinical relevance regarding the propensity for
BCC to occur within certain anatomic structures or
in a particular distribution is not fully understood,
although recent evidence supports a link between
SODEN ET AL
TABLE 1. Reported Cases of Basal Cell Carcinoma Arising in Surgical Scars
Procedure/Site
Age
Latency Period (Years)
Year
Reference
Colostomy site
Hair transplantation recipient site
Tracheostomy scar
Sternotomy (2 cases)
67
41
54
62
53
68
68
69
54
49
68
80
33
5
27
5
1
2–3
1
67
21
6
19
66–74
1975
1979
1983
1998
1998
1994
1999
2001
2004
2004
2004
2008
Didolkar et al.5
White6
Warren et al.7
Dolan et al.8
Hemithyroidectomy
Inguinal Herniorrhaphy
Cleft lip repair
Laparoscopic port site
Parotid gland excision
Midline sternotomy
Mastoidectomy (bilateral)
tumorigenesis and embryogenesis.17–19 Embryonic
fusion planes are the regions of mesenchymal
migration and fusion of the five primordial facial
processes during the fifth to tenth weeks of human
development.20 It has been postulated that these
structures offer ‘‘a path of least resistance’’21 for
tumor cells, facilitating their horizontal, deep, and
often subclinical spread because these planes extend
in a direction perpendicular to the surface of the
skin.22–25 A recent study showed that BCC was more
than four times as likely to occur on an embryonic
fusion plane as on other regions of the midface.26
In addition, CXCR4, a chemokine expressed during
embryogenesis, was recently identified in some BCCs
and may help to promote tumor growth and progression.27 These findings, when combined with the
recent theory that fusion planes are areas especially
receptive to angiogenesis, cell motility, and chemotaxic factors,28 may help to clarify why BCC tends to
occur along certain planes of fusion.
Although not directly related to the fusion of the five
primordial facial processes, planes of fusion involved
in merging also exist between the tympanic and
mastoid portions of the skull. In terms of mastoid
development, pneumatization of the mastoid begins
on the 33rd week embryologically and continues up
to the age of 8 to 9. In patients with chronic suppurative otitis media, the mastoid has a sclerotic
structure.29 Although it is unclear whether chronic
middle ear disease leads to inadequate development
Jorquero et al.9
Ozyazgan and Kontas10
Wright et al.11
Durrani et al.12
Robins and Schvartzman13
Lau et al.14
Soden et al.
of mastoid pneumatization or inadequate mastoid
pneumatization predisposes to chronic middle ear
disease, the recent developments with regard to BCC
embryogenesis and tumor development suggest that
congenital defects in mastoid pneumatization or
mastoid embryologic fusion may help to produce ‘‘a
path of least resistance’’21 along which BCCs may
develop and spread within the mastoid or within
mastoidectomy scars.
To the best of our knowledge, this is the first case of
bilateral BCC arising in mastoidectomy scars. An
extensive review of the literature revealed no reported cases of a primary BCC arising within old
mastoidectomy scars. Although it remains unclear
why the patient had BCC arising bilaterally and
within his mastoidectomy scars, recent evidence
suggests that factors associated with embryogenesis
and tumorigenesis may contribute to the predilection
for BCC to involve particular anatomic locations.
Although this is only one case of BCC arising within
mastoidectomy scars, it suggests the importance of
examining these scars completely, if present, when
performing skin examinations.
References
1. Mann SB, Yande R, Arora MM, et al. Bilateral basal cell carcinoma of the ears. (A case report). J Laryngol Otol 1982;96:951–4.
2. Martinez Subias J, Dominguez Ugidos LJ, Urpegui Garcia A, et al.
Middle ear carcinoma. Acta Otorrinolaringol Esp 1998;49:234–6.
3 5 : 1 : J A N U A RY 2 0 0 9
137
BASAL CELL CARCINOMA ARISING IN MASTOIDECTOMY SCARS
3. Newhart H. Primary carcinoma of the middle ear: report of a case.
Laryngoscope 1917;27:543–55.
4. Hakata H, Ohashi T, Suzuki T. Bilateral carcinoma of the ears.
Report of a case. Arch Otolaryngol 1976;102:112–4.
5. Didolkar MS, Douglas HO, Holyoke ED, Elias EG. Basal cell
carcinoma originating at the colostomy site: report of a case. Dis
Colon Rect 1975;18:399–402.
6. White JW. Basal cell carcinoma in a hair transplantation recipient
site. Cutis 1979;23:322–5.
7. Warren GH, Pearlmen NW, Cain TL. Carcinoma arising in a
tracheostomy scar. Arch Otolaryngol 1983;109:352–3.
8. Dolan OM, Lowe L, Orringer MB, et al. Basal cell carcinoma
arising in a sternotomy scar: a report of three cases. J Am Acad
Dermatol 1998;38:491–3.
9. Jorquero E, Moreno JC, Diaz Cano SJ, et al. Basal cell carcinoma
arising in a surgical scar: reconstructive surgical treatment.
J Dermatol Surg Oncol 1994;20:846–7.
10. Ozyazgan I, Kontas O. Previous injuries or scars as risk factors for
the development of basal cell carcinoma. Scand J Plast Reconstr
Surg Hand Surg 2004;38:11–5.
11. Wright MW, McCarthy RA, Kelly EB, et al. Basal cell carcinoma
arising in a cleft lip repair scar. Dermatol Surg 2001;27:195–7.
12. Durrani AJ, Miller RJ, Davies M. Basal cell carcinoma arising in a
laparoscopic port site scar at the umbilicus. Plast Reconstr Surg
2005;116:348–50.
13. Robins DN, Shvartzman LA. Basal cell carcinoma occurring in
scar tissue following excision of a parotid gland pleomorphic
adenoma. Dermatol Surg 2004;30:1412–4.
14. Lau YS, Banwell PE, Pay AD. Basal cell carcinoma arising in
scars: late presentation 16 years after a midline sternotomy. Plast
Reconstr Surg 2004;113:1297–9.
15. Kowal-Vern A, Criswell BK. Burn scar neoplasms: a literature
review and statistical analysis. Burns 2005;31:403–13. Epub 2005
Apr 1.
16. Jellouli-Elloumi A, Kochbati L, Dhraief S, et al. Cancers arising
from burn scars: 62 cases. Ann Dermatol Venereol 2003
Apr;130:413–6. French.
17. Thayer SP, di Magliano MP, Heiser PW, et al. Hedgehog is an
early and late mediator of pancreatic cancer tumorigenesis.
Nature 2003;425:851–6.
138
D E R M AT O L O G I C S U R G E RY
18. Berman DM, Karhadkar SS, Maitra A, et al. Widespread requirement for hedgehog ligand stimulation in growth of digestive
tract tumours. Nature 2003;425:846–51.
19. Watkins DN, Berman DM, Burkholder SG, et al. Hedgehog signaling within airway epithelial progenitors and in small-cell lung
cancer. Nature 2003;422:313–7.
20. Bannister LH, Williams PL. Gray’s anatomy. New York: Churchill
Livingstone; 1995. p. 271–88.
21. Salasche SJ. Curettage and electrodessication in the treatment of
midfacial basal cell epithelioma. J Am Acad Dermatol
1983;8:496–503.
22. Lang PG, Duncan IM, Hochman M. Occurrence of subclinical
tumor in excised facial subunits. Arch Facial Plast Surg
2004;6:158–61.
23. Granstrom G, Aldenborg F, Jeppsson PH. Influence of embryonal
fusion lines for recurrence of basal cell carcinomas in the head and
neck. Otolaryngol Head Neck Surg 1986;95:76–82.
24. Mora RG, Robins P. Basal-cell carcinomas in the center of the
face: special diagnostic, prognostic, and therapeutic considerations. J Dermatol Surg Oncol 1978;4:315–21.
25. Mohs FE, Lathrop TG. Modes of spread of cancer of skin. Arch
Dermatol 1952;66:427–439.
26. Newman JC, Leffell DJ. Correlation of embryonic fusion planes
with the anatomical distribution of basal cell carcinoma.
Dermatol Surg 2007;33:957–65.
27. Chen GS, Yu HS, Lan CC, et al. CXC chemokine receptor
CXCR4 expression enhances tumorigenesis and angiogenesis of
basal cell carcinoma. Br J Dermatol 2006;154:910–8.
28. Waner M, North PE, Scherer KA, et al. The nonrandom distribution of facial hemangiomas. Arch Dermatol 2003;139:869–5.
29. Bayramoğlu FN, Ardiç CO, Kara MZ, et al. Importance of mastoid pneumatization on secretory otitis media. Int J Pediatr
Otorhinolaryngol 1997;40:61–6.
Address correspondence and reprint requests to:
Cylburn Earl Soden, Jr., MD, MA, Department of
Dermatology, University of California Irvine, 101 The
City Drive South, Bldg 53, Room 302A, Orange, CA
92868, or e-mail: [email protected]