Lichen planus of uterine cervix - the first report of a novel site of occurrence: a case report
© Gupta et al; licensee BioMed Central Ltd. 2009
Received: 30 November 2009
Accepted: 11 December 2009
Published: 11 December 2009
Lichen planus is an immune mediated inflammatory lesion involving skin and mucosal sites including oral mucosa, vulva and rarely vagina. Lichen planus occurring at mucosal sites has been shown to be associated with squamous cell carcinoma in a proportion of cases. To the best of our knowledge, no case of lichen planus of uterine cervix has been reported in the available literature.
A 45-year-old female underwent vaginal hysterectomy for uterine prolapse. The resected specimen showed a bluish-colored area in the non-dependent part of the ectocervix. Microscopic sections from this area showed dense lymphocytic infiltrate at the junction of mucosa and submucosa causing disruption of the basal cell layer. On immunohistochemical examination there was predominance of CD8+ T lymphocytes at the junction with scattered CD4+ T lymphocytes, characteristic of lichen planus. Based on the history and negative serum antibody titers, other differential diagnoses including lupus erythematosus and drug reaction were excluded. The patient did not have any cutaneous or oral lesions of lichen planus.
Lichen planus of uterine cervix is a hitherto unreported entity, and is worth studying considering the premalignant potential of lichen planus at other mucosal sites.
Lichen planus is chronic inflammatory mucocutaneous disease with an immunologic etiopathogenesis [1, 2]. It most commonly involves skin with rare involvement of oral cavity, nails, vulva and vaginal mucosa . Occurrence of squamous cell carcinoma (SCC) has been reported in oral lichen planus and vulvar lichen planus [4, 5]. In oral lichen planus, SCC has been reported in upto 3% of cases . Though uterine cervix is also a mucosal site, no case of lichen planus has been reported in the English literature.
We report the first case of cervical lichen planus with histopathological and immunohistochemical confirmation. Since cervical cancer is one of the commoner cancers in females, report of lichen planus, a pre-neoplastic condition, becomes important. More such cases need to be reported to delineate the biologic significance of the lesion at this site.
A 45-year-old Indian female presented to the gynecologist with a six-month history of a mass descending down the vagina. She was post menopausal for the last three years and there was no relevant medical or surgical history. Local examination revealed second-degree uterine prolapse with cystocoele and rectocoele. Cervix showed a bluish-colored area measuring 2.5 × 3 cm in size. This area was not related to the most dependent part of cervix. Vulva and vagina were unremarkable. Routine cervical smear was reported as atrophic smear with inflammation. Pre-operative biochemical investigations were unremarkable and she underwent vaginal hysterectomy.
Lichen planus is a chronic immune-mediated mucocutaneous disease with characteristic violaceous polygonal flat-topped papules and plaques. It occurs mainly on flexor surfaces of extremities and trunk, though can also involve scalp, nails, oral and genital mucosa . Genital lichen planus is seen typically on the vulva, and rarely on the vagina, as part of the vulvo-vaginal-gingival syndrome . In contrast to lesions of cutaneous LP, mucosal lesions, including oral cavity, do not show compact orthokeratosis or hypergranulosis. Instead, parakeratosis with frequently atrophic epithelium is seen, sometimes with ulceration of the epithelium . Till date no case of lichen planus involving uterine cervix has been reported in the available literature. The lesion in our case was seen as an isolated finding, in the absence of cutaneous lesions. The same is true in a proportion of oral lesions which occur without cutaneous involvement.
The pathogenesis of lichen planus is not certain, though many studies have supported an immunologic mechanism. The role of T lymphocytes has been emphasized with cytotoxic activity of the CD8+ve subset of T lymphocytes responsible for keratinocyte damage [1, 2]. In our case, immunohistochemistry for T cell subsets showed specific localization of CD8-positive cells at the junction of mucosa and submucosa associated with basement membrane disruption, supporting the role of CD8-positive T cells in the pathogenesis of this entity. The etiology of LP in oral cavity has been investigated extensively. Various infectious agents, including bacterial, fungal (Candida) and viral organisms have been implicated as etiologic agents of oral LP. Among viruses, human papilloma virus (HPV) has also been found in oral LP . Since HPV infection is common in cervix, its role in causation of LP of cervix needs to be evaluated after more cases of cervical LP are added in the literature. In our patient, staining for HPV could not be performed. However, we propose that uterine prolapse in our patient may have exposed the cervix to the environmental agents leading to the development of lichen planus.
Malignant transformation of cutaneous lesions of lichen planus has been reported in less than 1% of cases . Similar phenomenon has been documented in 0.3-3% of cases of oral lesions [4, 10]. Rare cases of squamous cell carcinoma have been reported in vulvar lichen planus . Cervix is another mucosal site, where we report the first case of lichen planus, its pre-malignant potential remains to be seen.
The gross appearance of cervical lesion needs to be differentiated from congestion due to prolapse of uterus. Congestion occurs as bluish-brown discoloration on the dependent portion of the cervix whereas lichen planus has no such preference. Histologic examination differentiates between congestion and lichen planus, since congestion does not exhibit the band-like infiltrate observed in lichen planus with destruction of the overlying epithelium. Characteristic localization of T-cell subsets using Immunohistochemistry for CD4 and CD8 further resolves the matter. Other lesions, like involvement in systemic lupus erythematosus and drug reactions were excluded with the history and negative serologic findings.
Cervical lichen planus is a hitherto unreported entity. We report the first case of lichen planus of uterine cervix confirmed by histopathology and immunohistochemistry. Since squamous cell carcinoma has been reported in long-standing lesions of mucosal lichen planus, existence of lichen planus in uterine cervix needs to be identified and more cases must be added to the literature in order to assist in the further study on this subject.
Written consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review from the journal's Editor-in-Chief.
- Ishii T: Immunohistochemical demonstration of T cell subsets and accessory cells in oral lichen planus. J Oral Pathol. 1987, 16: 356-361. 10.1111/j.1600-0714.1987.tb00708.x.View ArticlePubMedGoogle Scholar
- Matthews JB, Scully CM, Potts AJ: Oral lichen planus: an immunoperoxidase study using monoclonal antibodies to lymphocyte subsets. Br J Dermatol. 1984, 3: 587-595. 10.1111/j.1365-2133.1984.tb06629.x.View ArticleGoogle Scholar
- Boyd AS, Neldner KH: Lichen planus. J Am Acad Dermatol. 1991, 25: 593-619. 10.1016/0190-9622(91)70241-S.View ArticlePubMedGoogle Scholar
- Kaplan B, Barnes L: Oral lichen planus and squamous cell carcinoma: case report and update of the literature. Arch Otolaryngol. 1985, 111: 543-547.View ArticlePubMedGoogle Scholar
- Lewis FM, Harrington CI: Squamous cell carcinoma arising in vulval lichen planus. Br J Dermatol. 1994, 131: 703-705. 10.1111/j.1365-2133.1994.tb04987.x.View ArticlePubMedGoogle Scholar
- Pelisse M: The vulvo-vaginal-gingival syndrome: a new form of erosive lichen planus. Int J Dermatol. 1989, 28: 381-384. 10.1111/j.1365-4362.1989.tb02484.x.View ArticlePubMedGoogle Scholar
- Shklar G: Erosive and bullous oral lesions of lichen planus. Arch Dermatol. 1968, 97: 411-416. 10.1001/archderm.97.4.411.View ArticlePubMedGoogle Scholar
- Kashima HK, Kutcher M, Kessis T, Levi LS, de Villiers EM, Shah K: Human papilloma virus in squamous cell carcinoma, leukoplakia, lichen planus and clinically normal epithelium of oral cavity. Ann Otol Rhinol Laryngol. 1990, 99: 55-61.PubMedGoogle Scholar
- Sigurgeirsson B, Lindelof B: Lichen planus and malignancy: an epidemiologic study of 2071 patients and a review of the literature. Arch Dermatol. 1991, 127: 1684-1688. 10.1001/archderm.127.11.1684.View ArticlePubMedGoogle Scholar
- Castano E, Lopez-Rios F, Alvarez-Fernandez JG, Rodriguez-Peralto JL, Iglesias L: Verrucous carcinoma in association with hypertrophic lichen planus. Clin Exp Dermatol. 1997, 22: 23-25. 10.1046/j.1365-2230.1997.d01-241.x.View ArticlePubMedGoogle Scholar
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