Gynecological Endocrinology, August 2009; 25(8): 543–545

CASE REPORT

Oral stomatitis induced by endogenous progesterone: Case report

ELIANA M. MINICUCCI1, ALINE B. CARRENHO1, SILKE A. T. WEBER2, FERNANDA M. BOMBINI1, RENATA A. M. A. RIBEIRO1, MARIANGELA E. A. MARQUES3, & DANIEL A. RIBEIRO4
Department of Dermatology and Radiotherapy, 2Department of Otorhinolaryngology and Ophthalmology, 3Department of Pathology, Botucatu Medical School, Sao Paulo State University, UNESP, Sao Paulo, Brazil, and 4Department of Biosciences, Federal University of Sao Paulo, UNIFESP, Santos, Sao Paulo, Brazil (Received 21 February 2008; revised 12 April 2009; accepted 27 April 2009)
1

Abstract Oral stomatitis induced by endogenous progesterone is a rare clinical condition which may be associated with cutaneous involvement. That is probably due to the peak of progesterone production during the luteal phase of the menstrual cycle. In the present case report, a 21-year-old patient displayed recurrent ulcerative lesions located on the buccal mucosa or the upper lip, on a monthly basis since the age of 15. Such lesions would always manifest themselves on the second day until the end of the menstrual cycle.

Keywords: Oral stomatitis, progesterone

Introduction Hypersensitivity induced by female sexual hormones is a rare clinical condition in which the patient develops a hypersensitivity reaction to endogenous progesterone. Such pathological condition occurs in patients ranging from 16 to 48 years of age with a predominance of young people [1]. Clinical manifestation is triggered every month during the luteal phase of the menstrual cycle, when the peak of progesterone production is reached. The clinical manifestations are variable [2] and include urticaria [3,4], erythema multiforme like-reaction [5], and eczema [6]. However, after the menstrual cycle, lesions disappear spontaneously. To date, a large number of studies have addressed clinical manifestations, especially on the skin, induced by endogenous progesterone. To the best of our knowledge, there are a few case reports addressing lesions specifically in the oral mucosa [2]. Therefore, such a circumstance justifies this case report as well as others; and, by taking into consideration, the current article describes a case report of oral stomatitis induced by endogenous progesterone.

Case report A 21-year-old Caucasian woman was referred to the Department of Dermatology, at the Ambulatory Care Center of Stomatology at Botucatu Medical School – Sao Paulo State University (UNESP), Brazil – complaining of ulcerative lesions in the perioral region, buccal mucosa, and upper lip (Figures 1 and 2). The patient reported that those lesions were painful. She also added that they had first appeared when she was 15 years old, on a monthly basis. However, the general conditions of health were good. Under clinical examination, no skin abnormalities were found. No drugs were used for minimizing the symptomatology. To exclude herpes as putative diagnosis, Tzanck’s test was performed [7]. The result was negative; and, as a consequence, incisional biopsy was performed. Microscopically, the lesion had sub- and intraepithelial vesicles associated with necrosis in the basal layer (Figure 3). Moderate inflammatory infiltrate consisting of lymphocytes, neutrophils and eosinophils was present, with some of the inflammatory cells located in the perivascular region (Figure 3). Skin testing

Correspondence: Daniel Araki Ribeiro, DDS, PhD, Departamento de Biocie ˆ ncias, Universidade Federal de Sa ˜ o Paulo – UNIFESP, Av. Ana Costa 95, 11060001 Santos, SP, Brazil. Tel: þ55-1332218058. Fax: þ55-1332232592. E-mail: daribeiro@unifesp.br ISSN 0951-3590 print/ISSN 1473-0766 online ª 2009 Informa UK Ltd. DOI: 10.1080/09513590903015585

but Tzanck’s test presented a negative result. The lesions occurred in the perioral region. Discussion Autoimmune reaction triggered by endogenous progesterone is a rare clinical condition. such as in the case of a viral infection [11. The occurrence of antibodies against endogenous progesterone has been demonstrated in patients presenting history of oral ulcers since their first menstruation [12]. was performed and no reaction in skin areas developed after 48-h evaluation. the patient Figure 1. In this case report. [8] have postulated that lesions induced by endogenous progesterone disappear 1 week after the menstrual cycle. Patients have reported cyclic lesions mainly in the skin. Another possibility is a crossreaction between endogenous progesterone and circulating antibodies produced by putative antigen present in the body. After that. Growing evidence suggests that synthetic progesterone may stimulate antibodies against endogenous progesterone in contraceptive users [13]. a derivative of progesterone. The patient reported that her clinical symptoms began to decline gradually over a few weeks. final diagnosis was perfomed taking into consideration the clinical history only. in which it was prescribed Tamoxifen (Nolvadex). Moghadam et al. The underlying mechanisms by which endogenous progesterone becomes antigenic remain unknown so far. and no recurrences were detected up to now (8 months after initial diagnosis). Clinical aspects of lesions in the buccal mucosa.544 E.E. The picture is characterized by recurrent cutaneous lesions during the luteal phase of the menstrual cycle. Clinical aspects of lesions in the upper lip. which are with estrogen (1 mg/ml) and Depo-Provera (1 mg/ ml). M. Therefore. these findings supported the final diagnosis of oral stomatitis induced by endogenous progesterone. was performed and no reaction in skin areas developed after 48-h evaluation. Figure 3. an antiestrogen agent. Skin testing with estrogen (1 mg/ml) and Depo-Provera (1 mg/ml).11]. Other authors have assumed hypersensitivity to be induced by endogenous progesterone. buccal mucosa. we have been able to report the instance of a woman with oral manifestations induced by endogenous progesterone. There is no relationship between oral stomatitis induced by progesterone and positive response in this test. The early clinical pattern seemed to be a herpes infection. followed a consultation with her gynecologist. Such lesions appear before menstruation and remain even after the menstrual cycle is over [2]. stain. and upper lip with symptomatology.12]. . Figure 2. It has been suggested that abnormalities in the composition of the hormone are present in women able to develop such autoimmune reaction [11]. when the levels of endogenous progesterone are increased. Photomicrography of the lesion (H. 640 magnification). Taken as a whole. such as anaphylaxis during the menstrual cycle [9]. a derivative of progesterone. such as Tamoxifen [14]. Therapy is the use of anti-estrogenic drugs. All symptoms disappear after some days [10. Minicucci et al. at a dosage of 20 mg/day for 2 months as described elsewhere [8]. estrogens (Premarin).

Hersini S.46:1177–1179. Ulcerative stomatitis caused by endogenous progesterone. 5. Gubinelli E. Campos A. 8. Brzoza Z. Cocuroccia B. Autoimmune progesterone dermatitis associated with infertility treatment. dentists and/ or endocrinologists should be aware of such concerns. Comparison of the Tzanck test and polymerase chain reaction in the diagnosis of cutaneous herpes simplex and varicella zoster virus infections. Int J Dermatol 2007. Int J Dermatol 2008. In this case. Villalmanzo JG. Guerrero M. Aktas E. Chatterton RT Jr.55:597–599. Iran J Allergy Asthma Immunol 2007. 12. Ozcan A. Robinson-Bostom L.48:257–261. at a dosage of 20 mg/day for 2 months as described elsewhere [8]. Herzberg AJ. References 1. Durmaz R. 14. 545 4. Sex hormones and urtica ´ ria. J Am Acad Dermatol 1995.Stomatitis and progesterone able to interrupt the ovulation process as well as the production of endogenous progesterone [2. Gaynor LV. Berger H. Declaration of interest: The authors report no conflicts of interest. 3. the patient received Tamoxifen (Nolvadex) after establishing the final diagnosis. 11. Autoimmune progesterone dermatitis responding to tamoxifen. J Am Acad Dermatol 2008. . Cirillo-Hyland VA. and no recurrences were detected up to now (8 months after initial diagnosis). Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1998. 10. Jenkins J. Gisondi P. Senol M. J Dermatol Sci 2008. Allergy 1987. Grave anaphylactic-like reaction in the course of menstruation: a case report. Baker BF. J Clin Endocrinol Metab 1985.121:135–137. Saglam H.22:54–56.85:537–541. Moghadam BKH. Farah FS.42:205–208. Ozerol IH. Nevertheless. Geng A. Radvany RM. Rogala B.32: 333–335. Wojnarowska FT. Autoimmune progesterone dermatitis. Autoimmune progesterone dermatitis. Strohmeyer CR. Scupham RK. The authors alone are responsible for the content and writing of the article. Stephens CJM. Identification of a 17 hydroxyporgesterone-binding immuno- 13. 15. Autoimmune progesterone dermatitis. Edwards CRW. Gynecol Endocrinol 2006. J Intern Med 1990.58:353–355. Farashahi MH. Autoimmune progesterone dermatitis. Teelucksingh S.113:426–430. oral stomatitis induced by endogenous progesterone is a rare disease. Walling HW. Basomba A. Girolomoni G. Gynecologists. Case report with histologic overlap of erythema multiforme and urticaria. J Allergy Clin Immunol 1971. Autoimmune progesterone dermatitis and stomatitis.3]. Histological confirmation of the clinical diagnosis is not essential in most cases. Cheesman KI. Willkinson JD. Hart R.42:477–479. Nabavi M.52:79–86.6:97–99. Shirkhoda Z. Ann Intern Med 1955. 9. globulin in the serum of a woman with periodic rashes.121:135–137. 6. several patients do not undergo any therapy [15]. Bemanian MH. Arch Dermatol 1977. Shbaklu Z. Gharagozlou M. 2. Autoimmune progesterone dermatitis. Seyhan M. The patient reported that her clinical symptoms began to decline gradually over a few weeks. Br J Dermatol 1989. Autoimmune progesterone anaphylaxis. Kasperska-Zajac A. 7. Autoimmune progesterone urtica ´ ria. As a conclusion.47:380–382.

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