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Lacrimal Canalicular Duct Scarring in Patients With Lichen Planus FREE

Naomi K. Webber, MBBS, BSc, MRCPCH; Jane F. Setterfield, MBBS, BDS, FRCP; Fiona M. Lewis, MBBS, MD, FRCP; Sallie M. Neill, MBBS, FRCP
[+] Author Affiliations

Author Affiliations: St John's Institute of Dermatology, Guy's and St Thomas' National Health Service Foundation Trust, London, England.


Arch Dermatol. 2012;148(2):224-227. doi:10.1001/archdermatol.2011.580.
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Published online

ABSTRACT

Background Lichen planus (LP) is a mucocutaneous inflammatory disease that affects multiple sites, including the skin, oral cavity, vulva, and vagina and can result in scarring and stricture formation. It has also been shown to cause lacrimal canalicular blockage in a series of patients attending an ophthalmology clinic. We describe a cohort of women with vulvovaginal LP who also had signs of lacrimal canalicular scarring on examination.

Observations We report 9 cases of LP with scarring of the conjunctiva around the lacrimal ducts. Seven of 9 women had symptoms of epiphora, and in 2 women lacrimal canalicular scarring was an incidental finding. Seven of 9 cases were diagnosed by an ophthalmologist. All women had biopsy-proven LP at 1 mucocutaneous site each. Seven of 9 women had vulvovaginalgingival syndrome, which is a subgroup of severe erosive LP.

Conclusions Given the strong association between erosive mucocutaneous LP and multisite scarring sequelae, it is not unexpected that ocular inflammation may lead to lacrimal duct stenosis. We believe that this complication has been underreported among patients with LP and that an ophthalmological history and examination of the punctum of the lacrimal duct should be sought, especially in patients with the erosive subtype of LP.

Figures in this Article

Lichen planus (LP) is a mucocutaneous inflammatory dermatosis that can cause scarring alopecia and nail destruction. Forty percent of patients with cutaneous LP may also have mucosal lesions, most commonly involving the oral and genital mucosa, with stenosis, vaginal synechia, and loss of vulval architecture. In some cases, there may be involvement of the esophagus, resulting in strictures and dysphagia. Lacrimal duct obstruction secondary to LP was previously described among 8 patients (5 female and 3 male) by Durrani et al1 in 2008. Lacrimal canalicular obstruction had previously been described in a 67-year-old woman with LP.2 Among some women with vulvovaginalgingival (VVG) syndrome, a subtype of severe erosive LP, we have noted scarring around the punctum of the lacrimal duct, which we believe is related to their LP.

REPORT OF CASES

CASE 1

A 65-year-old woman of white race/ethnicity had biopsy-proven LP affecting the skin, scalp, and oral and vulval mucosa. Her disease was controlled using topical treatments alone. Her medical history included sclerosing cholangitis. She had a 3-year history of epiphora associated with redness and slight photophobia. An ophthalmological assessment revealed cicatricial conjunctivitis and scarring of the lower conjunctiva around the lower lacrimal duct openings bilaterally (Figure 1). A normal lacrimal duct opening is shown in Figure 2 for comparison. Her symptoms improved with daily eye drops of carmellose sodium, 0.5%, and prednisolone sodium phosphate, 0.5%, and she has not required intubation of the lacrimal duct outflow system.

Place holder to copy figure label and caption
Graphic Jump Location

Figure 1. Right lower eyelid showing white scarring (arrow) around the punctum of the lacrimal duct. A stellate stricture is contiguous to lacrimal duct scarring in the inferior fornix.

Place holder to copy figure label and caption
Graphic Jump Location

Figure 2. Right lower eyelid showing a normal lacrimal duct opening (arrow). No circumferential white scarring (as seen in Figure 1) is observed around the duct opening.

CASES 2 THROUGH 9

Eight additional women with LP had scarring of the conjunctiva around the lacrimal ducts. The details of these cases are summarized in the Table.

Table Graphic Jump LocationTable. Cases of Lichen Planus With Lacrimal Canalicular Duct Scarring

COMMENT

Lichen planus is an autoimmune disease that affects the skin and mucous membranes, including the ear canals, resulting in conductive deafness and esophageal involvement that leads to strictures and dysphagia.3,4 It has a slight female predominance (1.4:1) and affects 1% to 2% of adults.5 In most patients, it is self-limiting and follows a benign course. The mucosal variant of LP may involve the oral and genital mucosa and is characterized by sores and erosions. Scarring results in fibrous bands in the buccal mucosa, leading to a reduced vestibular sulcus and limitation of the oral aperture.6 In our series, 7 of 9 women had VVG syndrome, which is a severe subtype of LP characterized by erosions or desquamation of vulval, vaginal, and gingival mucosa, with predilection for scarring and stricture formation.6,7 The pathogenesis of LP is unclear, but it is believed to be caused by specific and nonspecific immunological mechanisms. Specific mechanisms probably include antigen presentation by basal keratinocytes and antigen-specific T cells that are cytotoxic to keratinocytes, whereas nonspecific mechanisms include activation of matrix metalloproteinases and mast cells.8 A potential association with the HLA-DQB1*0201 allele has been described in patients with VVG, suggesting a genetic predisposition; however, a strong association with autoimmune disorders in this variant of LP may in part explain the increased frequency of this allele.6 Five of 7 patients herein with VVG LP tested positive for the HLA- DQB1*0201 allele, whereas the 2 patients without VVG LP were negative for this allele. Given the strong association between erosive mucocutaneous LP, particularly of the VVG syndrome subtype, and multisite scarring sequelae, it is not unexpected that ocular inflammation may lead to subconjunctival fibrosis and lacrimal duct stenosis.

Lichen planus may manifest as ocular involvement, most commonly with lesions affecting the eyelid, which is the location of the lacrimal duct.9,10 The lacrimal gland produces tears that enter the lacrimal duct and drain into the nose. This system is composed of epithelium-lined lumina. Symptoms of lacrimal canalicular obstruction include epiphora secondary to the overproduction of tears or their inadequate drainage. Stagnant tears within the system can lead to infection, and spillage of tears can lead to eczematous changes on the lower eyelid.

Rarely, LP can affect the conjunctiva, resulting in conjunctival inflammation, cicatrization. and subepithelial scarring.11,12 One of our patients had LP that caused recurrent conjunctival injection and photophobia. Lichen planus has also been associated with keratitis and keratoconjunctivitis sicca.13

Lacrimal canalicular obstruction may be caused by trauma, eye surgery, Stevens-Johnson syndrome, ocular cicatricial pemphigoid, or use of systemic fluorouracil or certain topical medications.14 Unilateral obstruction may be seen following herpes simplex virus infection, usually in younger patients following primary periocular herpes simplex virus infection.15 Our patients had no history of eye trauma, eye surgery, herpes simplex virus infection, or use of systemic fluorouracil.

Lacrimal duct obstruction is treated by dacryocystorhinostomy with retrograde canaliculostomy. However, the severity of canalicular obstruction associated with LP often results in a poor outcome with this procedure, and primary or secondary placement of Jones tubes (lacrimal drainage bypass devices) is often needed.1 Four patients underwent lacrimal reconstructive surgery by an ophthalmologist: 1 patient had improvement in her symptoms, while the other 3 patients did not. Medical intervention has seldom been described. One patient with VVG syndrome and lacrimal canalicular stenosis was treated with systemic cyclosporin A (150 mg twice daily), which resulted in total resolution of her epiphora after 6 weeks of therapy.16 Four of 9 patients herein had been prescribed an immunosuppressant for their oral or vulval disease, none of whom reported improvement in their eye symptoms with immunosuppressant treatments.

In conclusion, lacrimal drainage obstruction secondary to LP has been previously reported in 8 patients by Durrani et al1 in 2008. Histologic findings in tissue from 2 blocked lacrimal ducts herein showed chronic predominantly lymphocytic subepithelial infiltrate, with a few macrophages and occasional mast cells. There was also scattered lymphocytic infiltration of the basal epithelium and thickening and fragmentation of the basement membrane; immunohistochemistry showed no immune complex deposition, and these changes were consistent with LP. Although the patients herein had no histologic diagnosis demonstrating LP in the lacrimal duct, we can assume that the lacrimal canalicular mucosa was affected by LP, causing punctal and canalicular stenosis and subsequent functional lacrimal duct obstruction. We believe that this complication has been underreported among patients with LP and that an ophthalmological history and examination of the punctal of the lacrimal duct should be sought in patients with the erosive subtype of LP.

ARTICLE INFORMATION

Correspondence: Naomi K. Webber, MBBS, BSc, John's Institute of Dermatology, Guy's and St Thomas' National Health Service Foundation Trust, Westminster Bridge Road, London SE1 7EH, England (naomi.webber@doctors.org.uk).

Accepted for Publication: August 21, 2011.

Author Contributions: All authors had full access to all the data in the study and take responsibility for the integrity of the data and the accuracy of the data analysis. Study concept and design: Webber and Neill. Acquisition of data: Webber, Setterfield, Lewis, and Neill. Analysis and interpretation of data: Webber, Setterfield, and Lewis. Drafting of the manuscript: Webber, Setterfield, Lewis, and Neill. Critical revision of the manuscript for important intellectual content: Webber, Setterfield, Lewis, and Neill. Study supervision: Neill.

Financial Disclosure: None reported.

REFERENCES

Durrani OM, Verity DH, Meligonis G, Rose GE. Bicanalicular obstruction in lichen planus: a characteristic pattern of disease.  Ophthalmology. 2008;115(2):386-389
PubMed   |  Link to Article
McNab AA. Lacrimal canalicular obstruction in lichen planus.  Orbit. 1998;17(3):201-202
PubMed   |  Link to Article
Martin L, Moriniere S, Machet MC, Robier A, Vaillant L. Bilateral conductive deafness related to erosive lichen planus.  J Laryngol Otol. 1998;112(4):365-366
PubMed   |  Link to Article
Wedgeworth EK, Vlavianos P, Groves CJ, Neill S, Westaby D. Management of symptomatic esophageal involvement with lichen planus.  J Clin Gastroenterol. 2009;43(10):915-919
PubMed   |  Link to Article
Vincent SD, Fotos PG, Baker KA, Williams TP. Oral lichen planus: the clinical, historical, and therapeutic features of 100 cases.  Oral Surg Oral Med Oral Pathol. 1990;70(2):165-171
PubMed   |  Link to Article
Setterfield JF, Neill S, Shirlaw PJ,  et al.  The vulvovaginal gingival syndrome: a severe subgroup of lichen planus with characteristic clinical features and a novel association with the class II HLA DQB1*0201 allele.  J Am Acad Dermatol. 2006;55(1):98-113
PubMed   |  Link to Article
Pelisse M, Leibowitch M, Sedel D, Hewitt J. A new vulvovaginogingival syndrome: plurimucous erosive lichen planus.  Ann Dermatol Venereol. 1982;109(9):797-798
PubMed
Farhi D, Dupin N. Pathophysiology, etiologic factors, and clinical management of oral lichen planus, part I: facts and controversies.  Clin Dermatol. 2010;28(1):100-108
PubMed   |  Link to Article
Itin PH, Buechner SA, Rufli T. Lichen planus of the eyelids.  Dermatology. 1995;191(4):350-351
PubMed   |  Link to Article
Sharma R, Singhal N. Lichen planus of the eyelids: a report of 5 cases.  Dermatol Online J. 2001;7(1):5.httsp://dermatology.cdlib.org/DOJvol7num1/case_reports/lichen/sharma.html. Accessed September 18, 2011
PubMed
Neumann R, Dutt CJ, Foster CS. Immunohistopathologic features and therapy of conjunctival lichen planus.  Am J Ophthalmol. 1993;115(4):494-500
PubMed
Thorne JE, Jabs DA, Nikolskaia OV, Mimouni D, Anhalt GJ, Nousari HC. Lichen planus and cicatrizing conjunctivitis: characterization of five cases.  Am J Ophthalmol. 2003;136(2):239-243
PubMed   |  Link to Article
Zijdenbos LM, Starink TM, Spronk CA. Ulcerative lichen planus with associated sicca syndrome and good therapeutic result of skin grafting.  J Am Acad Dermatol. 1985;13(4):667-668
PubMed   |  Link to Article
McNab AA. Lacrimal canalicular obstruction associated with topical ocular medication.  Aust N Z J Ophthalmol. 1998;26(3):219-223
PubMed   |  Link to Article
McLean CJ, Rose GE. Postherpetic lacrimal obstruction.  Ophthalmology. 2000;107(3):496-499
PubMed   |  Link to Article
Boyce AE, Marshman G, Mills RAD. Erosive mucosal lichen planus and secondary epiphora responding to systemic cyclosporin A treatment.  Australas J Dermatol. 2009;50(3):190-193
PubMed   |  Link to Article

Figures

Place holder to copy figure label and caption
Graphic Jump Location

Figure 1. Right lower eyelid showing white scarring (arrow) around the punctum of the lacrimal duct. A stellate stricture is contiguous to lacrimal duct scarring in the inferior fornix.

Place holder to copy figure label and caption
Graphic Jump Location

Figure 2. Right lower eyelid showing a normal lacrimal duct opening (arrow). No circumferential white scarring (as seen in Figure 1) is observed around the duct opening.

Tables

Table Graphic Jump LocationTable. Cases of Lichen Planus With Lacrimal Canalicular Duct Scarring

References

Durrani OM, Verity DH, Meligonis G, Rose GE. Bicanalicular obstruction in lichen planus: a characteristic pattern of disease.  Ophthalmology. 2008;115(2):386-389
PubMed   |  Link to Article
McNab AA. Lacrimal canalicular obstruction in lichen planus.  Orbit. 1998;17(3):201-202
PubMed   |  Link to Article
Martin L, Moriniere S, Machet MC, Robier A, Vaillant L. Bilateral conductive deafness related to erosive lichen planus.  J Laryngol Otol. 1998;112(4):365-366
PubMed   |  Link to Article
Wedgeworth EK, Vlavianos P, Groves CJ, Neill S, Westaby D. Management of symptomatic esophageal involvement with lichen planus.  J Clin Gastroenterol. 2009;43(10):915-919
PubMed   |  Link to Article
Vincent SD, Fotos PG, Baker KA, Williams TP. Oral lichen planus: the clinical, historical, and therapeutic features of 100 cases.  Oral Surg Oral Med Oral Pathol. 1990;70(2):165-171
PubMed   |  Link to Article
Setterfield JF, Neill S, Shirlaw PJ,  et al.  The vulvovaginal gingival syndrome: a severe subgroup of lichen planus with characteristic clinical features and a novel association with the class II HLA DQB1*0201 allele.  J Am Acad Dermatol. 2006;55(1):98-113
PubMed   |  Link to Article
Pelisse M, Leibowitch M, Sedel D, Hewitt J. A new vulvovaginogingival syndrome: plurimucous erosive lichen planus.  Ann Dermatol Venereol. 1982;109(9):797-798
PubMed
Farhi D, Dupin N. Pathophysiology, etiologic factors, and clinical management of oral lichen planus, part I: facts and controversies.  Clin Dermatol. 2010;28(1):100-108
PubMed   |  Link to Article
Itin PH, Buechner SA, Rufli T. Lichen planus of the eyelids.  Dermatology. 1995;191(4):350-351
PubMed   |  Link to Article
Sharma R, Singhal N. Lichen planus of the eyelids: a report of 5 cases.  Dermatol Online J. 2001;7(1):5.httsp://dermatology.cdlib.org/DOJvol7num1/case_reports/lichen/sharma.html. Accessed September 18, 2011
PubMed
Neumann R, Dutt CJ, Foster CS. Immunohistopathologic features and therapy of conjunctival lichen planus.  Am J Ophthalmol. 1993;115(4):494-500
PubMed
Thorne JE, Jabs DA, Nikolskaia OV, Mimouni D, Anhalt GJ, Nousari HC. Lichen planus and cicatrizing conjunctivitis: characterization of five cases.  Am J Ophthalmol. 2003;136(2):239-243
PubMed   |  Link to Article
Zijdenbos LM, Starink TM, Spronk CA. Ulcerative lichen planus with associated sicca syndrome and good therapeutic result of skin grafting.  J Am Acad Dermatol. 1985;13(4):667-668
PubMed   |  Link to Article
McNab AA. Lacrimal canalicular obstruction associated with topical ocular medication.  Aust N Z J Ophthalmol. 1998;26(3):219-223
PubMed   |  Link to Article
McLean CJ, Rose GE. Postherpetic lacrimal obstruction.  Ophthalmology. 2000;107(3):496-499
PubMed   |  Link to Article
Boyce AE, Marshman G, Mills RAD. Erosive mucosal lichen planus and secondary epiphora responding to systemic cyclosporin A treatment.  Australas J Dermatol. 2009;50(3):190-193
PubMed   |  Link to Article

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