Ophthal Plast Reconstr Surg, Vol. 28, No.

5, 2012 Case Reports

7. Bharti KH, Lodha ND, Wald MS. Mucinous eccrine carcinoma of

lower eyelid: a case report. Indian J Pathol Microbiol 2007;50:764–5.
8. Breiting L, Christensen L, Dahlstrøm K, et al. Primary mucinous
carcinoma of the skin: a population-based study. Int J Dermatol
2008;47:242–5.
9. Abe S, Matsumoto Y, Fujita T. Primary mucinous carcinoma of the
skin. Plast Reconstr Surg 1997;99:1160–4.
10. Mendoza S, Helwig EB. Mucinous (adenocystic) carcinoma of the
skin. Arch Derm 1971;103:68–78.
11. Kelly BC, Koay J, Driscoll MS, et al. Report of a case: primary
mucinous carcinoma of the skin. Dermatol Online J 2008;14:4.
12. Yeung KY, Stinson JC. Mucinous (adenocystic) carcinoma of sweat
glands with widespread metastasis. Case report with ultrastructural
study. Cancer 1977;39:2556–62.
13. Bindra M, Keegan DJ, Guenther T, Lee V. Primary cutaneous muci-
nous carcinoma of the eyelid in a young male. Orbit 2005;24:211–4.
14. Ortiz KJ, Gaughan MD, Bang RH, et al. A case of primary muci-
nous carcinoma of the scalp treated with mohs surgery. Dermatol
Surg 2002;28:751–4. FIG. 1.  1-cm well-demarcated nodular mass in the tarsal
15. Cecchi R, Rapicano V. Primary cutaneous mucinous carcinoma: conjunctiva of the right upper eyelid.
report of two cases treated with Mohs’ micrographic surgery.
Australas J Dermatol 2006;47:192–4.
16. Bertagnoli R, Cook DL, Goldman GD. Bilateral primary mucinous
carcinoma of the eyelid treated with Mohs surgery. Dermatol Surg
1999;25:566–8.
17. Wright JD, Font RL. Mucinous sweat gland adenocarcinoma of
eyelid: a clinicopathologic study of 21 cases with histochemi-
cal and electron microscopic observations. Cancer 1979;44:
1757–68.
18. Chauhan A, Ganguly M, Takkar P, Dutta V. Primary mucinous
carcinoma of eyelid: a rare clinical entity. Indian J Ophthalmol
2009;57:150–2.

Metastatic Adenoid Cystic

Carcinoma of the Eyelid
Do Young Park, M.D.*, Jung Hye Lee, M.D.*, Yeon-Lim Suh,
M.D., Ph.D.†, Kyung In Woo, M.D., Ph.D.*, and Yoon-Duck
Kim, M.D., Ph.D.*

Abstract: A 48-year-old man presented with an enlarging

mass of the right upper eyelid over a 1-month period.
Nine years prior, the patient was diagnosed with adenoid
cystic carcinoma (ACC) of the right maxillary sinus
and underwent a total maxillectomy followed by local
radiotherapy. Over the previous year, the patient had been
treated with chemotherapy and radiotherapy for multiple
organ metastases, including the spine, chest, and oral cavity.
Ophthalmic examination revealed a white, round mass on
the tarsal conjunctiva of the right upper eyelid, measuring
approximately 1 × 1 cm, and 2 palpable subcutaneous
masses near the inferior orbital rim. Excisional biopsies
of the masses were performed, and the histopathological
findings were consistent with ACC with solid and cribriform
patterns. Herein, the authors describe a case of metastatic
ACC of the eyelid, which has not been described in the
literature to their knowledge.

*Department of Ophthalmology and †Department of Diagnostic

Pathology, Samsung Medical Center, Sungkyunkwan University School of
Medicine, Seoul, Korea
Accepted for publication October 26, 2011.
The authors have no financial or conflict of interest to disclose.
Address correspondence and reprint requests to Dr. Yoon-Duck
Kim, M.D., Ph.D., Department of Ophthalmology of Samsung Medical FIG. 2.  A, Postcontrast axial CT scan shows a well-enhanced,
Center, Sungkyunkwan University School of Medicine, 50 Irwon-dong, 1-cm nodular lesion (arrow) in the right upper eyelid. B,C, Post-
Gangnam-gu, Seoul 135-710, Korea. E-mail: ydkimoph@skku.edu contrast coronal CT scans show 2 nodular enhancing lesions
DOI: 10.1097/IOP.0b013e31823f63f0 (arrows) at the inferior orbital rim.

© 2012 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc. e111
Case Reports Ophthal Plast Reconstr Surg, Vol. 28, No. 5, 2012
A denoid cystic carcinoma (ACC) is a rare malignant tumor
arising from the secretory epithelial cells of the salivary
glands. It accounts for less than 2% of all head and neck malig-
nancies and approximately 10% of all salivary neoplasms.1,2
ACC is characterized by a high rate of perineural spread, local
recurrences, and distant metastases.1 Although metastases to
distant sites, including lung, bone, kidney, brain, and liver, have
all been previously described, metastatic ACC to the eyelid has
yet to be reported.3
CASE REPORT
A 48-year-old man was referred for a 1-month history
of a slowly enlarging, painless mass of the right upper eyelid.
The patient’s past medical history was notable for a diagnosis
of ACC of the right maxillary sinus with bony invasion in 2001.
He underwent a right total maxillectomy followed by adjuvant
radiotherapy. In 2006, the patient visited the authors’ clinic for
cicatricial lower eyelid retraction, which developed after the
maxillectomy. He underwent adhesiolysis of the scar, subperi-
osteal suborbicularis oculi fat lift, and right lower eyelid eleva-
tion with a human dermal allograft (SureDerm, Hans Biomed
Corp. Korea). The lower eyelid position was well maintained
postoperatively. In 2008, the patient underwent a radical lymph
node dissection and radiotherapy of the neck due to cervical
lymph node metastases. In 2009, metastases to the spine, chest,
neck, and oral cavity were discovered, and the patient underwent
further chemotherapy and radiotherapy.
On examination, his vision was 20/20 bilaterally. A non-
tender, well-demarcated nodular mass, approximately 1 × 1 cm,
was palpated on the right upper eyelid. The white, well-vascular-
ized mass was grossly visible with eyelid eversion on the tarsal
conjunctiva (Fig. 1). Additionally, there were 2 other palpable
masses around the inferior orbital rim without inflammatory
signs, such as swelling, redness, or pain. They were mobile and
slightly smaller than the upper eyelid mass. The rest of the oph-
thalmic examination was unremarkable. On contrast-enhanced
FIG. 3.  Microscopic findings of right upper eyelid mass. A, the solid type of adenoid cystic carcinoma consists of irregular shaped
islands of basaloid cells. (hematoxylin-eosin, ×12). B, At high magnification, the tumor consists of sheets of small uniform basaloid cells
(hematoxylin-eosin, ×200).
e112 © 2012 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.
CT scans, all the masses enhanced homogenously, and there was
no orbital involvement of the lesions (Fig. 2).
The patient underwent excisional biopsies of the masses.
The upper eyelid mass was highly vascular, and tumor infil-
tration was noted on the conjunctiva and the skin. The upper
eyelid mass was excised completely with a pentagonal wedge
resection, including 5-mm resection margins, and tumor-free
margins were confirmed via frozen section analysis. Eyelid
reconstruction involved direct closure after a lateral canthot-
omy and superior cantholysis. Attention was then paid to the 2
masses of the inferior orbital rim, which were excised via skin
incision. Microscopic examination of all of the excised masses
demonstrated solid histopathological subtypes of ACC with a
nodular growth pattern (Fig. 3).
One month postoperatively, the patient developed
severe back pain and difficulty with urination. Systemic evalu-
ation revealed progression of multiple bone metastasis and
hematogenous and lymphangitic metastasis of the lung. The
patient refused further chemotherapy or radiotherapy and died
3 months later.
DISCUSSION
Eyelid metastases are rare, accounting for less than 1%
of all malignant lesions of the eyelid.4 Riley5 reported that the
most common primary origins of eyelid metastasis were from
the breast, skin, stomach, and lung. Metastases to the eyelids
usually tended to develop late in the course of the disease and
can manifest diverse clinical features.5,6 Ophthalmologists
should always consider the possibility of eyelid metastasis for
patients with known systemic cancer. These patients generally
harbor multiple metastatic sites, both ocular and nonocular, and
their systemic prognosis is poor.6
ACC is a rare epithelial tumor that accounts for less than
2% of all head and neck malignancies and less than 10% of all
salivary neoplasms.2 It has distinct characteristics of an indo-
lent but persistent growth and a high propensity for perineural
Ophthal Plast Reconstr Surg, Vol. 28, No. 5, 2012 Case Reports
spread, local recurrence, and distant metastasis.2 ACC originates
predominantly from the minor salivary glands.1 Distant metasta-
sis of ACC develops in approximately half of the patients and is
associated with an adverse final outcome.1 The onset of distant
metastasis of ACC generally occurs late, with the median time
between diagnosis of the primary and detection of the metasta-
sis being 60 months.7 The predominant site of the metastases is
the lung, although other sites include the bone, liver, kidney, and
brain.3 To the best of the authors’ knowledge, metastatic ACC to
the eyelid has not been reported previously.
Histologically, ACC can be composed of 3 subtypes:
cribriform, tubular, and solid. Among these, the solid subtype of
ACC is associated with more serious prognosis than the tubular
and cribriform type, and it is caused by the advanced stage and the
development of distant metastasis.2 In this case, upon microscopic
examination, specimens from the right upper eyelid and inferior
orbital rim revealed a typical solid type of ACC histopathologi-
cal specimen, composed of sheets of small uniform basaloid cells.
In conclusion, the authors report the first case of meta-
static ACC to the eyelid. Although it is rare, metastatic ACC to
the eyelids should be considered in the differential diagnosis of
eyelid masses, particularly when the patient has a prior history
of ACC of the head and neck region.
REFERENCES
1. Bradley PJ. Adenoid cystic carcinoma of the head and neck: a re-
view. Curr Opin Otolaryngol Head Neck Surg 2004;12:127–32.
2. Matsuba HM, Spector GJ, Thawley SE, et al. Adenoid cystic sali-
vary gland carcinoma. A histopathologic review of treatment failure
patterns. Cancer 1986;57:519–24.
3. Sung MW, Kim KH, Kim JW, et al. Clinicopathologic predic-
tors and impact of distant metastasis from adenoid cystic carci-
noma of the head and neck. Arch Otolaryngol Head Neck Surg
2003;129:1193–7.
4. Weiner JM, Henderson PN, Roche J. Metastatic eyelid carcinoma.
Am J Ophthalmol 1986;101:252–4.
5. Riley FC. Metastatic tumors of the eyelids. Am J Ophthalmol
1970;69:259–64.
6. Bianciotto C, Demirci H, Shields CL, et al. Metastatic tumors to
the eyelid: report of 20 cases and review of the literature. Arch
Ophthalmol 2009;127:999–1005.
7. Rapidis AD, Givalos N, Gakiopoulou H, et al. Adenoid cystic car-
cinoma of the head and neck. Clinicopathological analysis of 23
patients and review of the literature. Oral Oncol 2005;41:328–35.
Orbital Inflammation After
Dental Procedures
Christina H. Choe, M.D.*,
Lauren A. Eckstein, M.D., Ph.D.*,
and M. Reza Vagefi, M.D.†
Abstract: This study reports 3 cases of acute orbital
inflammation that occurred within 3 weeks of various dental
procedures and offers a possible mechanism as to their cause.
The charts of 3 patients were retrospectively examined.
*Department of Ophthalmology, Scheie Eye Institute, University of Penn­
sylvania, Philadelphia, Pennsylvania; and †Department of Ophthalmology,
University of California San Francisco, San Francisco, California, U.S.A.
Accepted for publication November 9, 2011.
Presented at the American Society of Ophthalmic Plastic and Reconstruc­
tive Surgery Annual Fall Meeting, October 2011, Orlando, FL, U.S.A.
The authors have no financial or conflict of interest to disclose.
Address correspondence and reprint requests to Dr. M. Reza Vagefi, M.D.,
University of California San Francisco, 10 Koret Way, K-201, San Francisco,
California 94143-0730, U.S.A. E-mail: vagefir@vision.ucsf.edu
DOI: 10.1097/IOP.0b013e318242ab34
© 2012 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc. e113
Clinical notes, laboratory testing, and imaging studies were
reviewed. The cases involved a 36-year old woman, a 61-year-
old woman, and a 44-year-old woman who developed acute
dacryoadenitis after tooth extraction in the former case and
after routine dental cleaning in the latter 2. All cases were
initially treated with an oral steroid taper over 6 to 8 weeks.
The first 2 cases resolved promptly and have remained
quiescent. The last individual had recurrent symptoms
prompting lacrimal gland biopsy that demonstrated chronic,
nongranulomatous inflammation without monoclonality.
The patient subsequently responded to periorbital steroid
injection only to have a recurrent bout of inflammation after
repeat dental cleaning. Another periorbital steroid injection
resulted in resolution of inflammation. The authors propose
that a subset of acute orbital inflammation may represent
an autoimmune response triggered by dental manipulation.
These cases are suggestive of an atypical variant of
noninfectious, microbe-induced inflammation.
N
oninfectious orbital inflammation may present acutely
with varying degrees of pain, periorbital edema, visual
disturbances, extraocular motility restriction, and proptosis.1
Inflammation may be localized or diffusely involve orbital
structures. In some cases, a systemic autoimmune condition,
such as granulomatosis with polyangiitis, giant cell arteritis,
systemic lupus erythematosus, or rheumatoid arthritis, is found
to be the underlying etiology. More often, it is idiopathic, and
the inciting cause of the inflammation is never identified.
The authors report 3 cases of noninfectious orbital
inflammation that occurred in close temporal association with
dental procedures. To the authors’ knowledge, no other cases
of such a correlation have been reported in the literature. The
authors also explore the potential etiologies of orbital inflamma-
tion after dental procedures.
CASE REPORT
Case 1. A 36-year-old woman presented with a 3-day history
of left periorbital pain and photophobia. She denied history of
fever or chills. At presentation, she reported a tooth extraction
due to infection 3 weeks prior. Accordingly, the patient was
placed on amoxicillin for 1 month. The visual acuity was 20/20
OU. Pupils were round and reactive to light without relative
afferent pupillary defect, and extraocular motility was intact.
External exam demonstrated significant periorbital edema and
tenderness over the lacrimal gland on the left side. Orbital exam
demonstrated 3 mm of proptosis on the left side with increased
resistance to retropulsion. Slit lamp exam was only significant
for chemosis on the left side but was otherwise within normal
limits, as was the dilated fundus exam.
Laboratory testing was remarkable only for a mild eleva-
tion in white blood cell count (13,000 cells/ml) without neu-
trophilic shift. Complete laboratory evaluation otherwise ruled
out other orbital inflammatory etiologies. In addition, syphilis,
tuberculosis, and HIV testing was negative. CT imaging dem-
onstrated an enlarged lacrimal gland on the left side (Fig. 1A).
Once infectious etiologies were ruled out, the patient com-
pleted an oral prednisone taper over 6 weeks with complete
resolution of inflammation. She has remained symptom free
for over 1 year.
Case 2. A 61-year-old woman presented complaining of sudden
onset periorbital swelling, erythema, and tenderness. The symp-
toms initially began on the left side 1 week after routine dental
cleaning. The patient denied history of fever or chills. She was
previously treated by a general ophthalmologist with a 5-day