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Unusual Multiple Recurrence of Lipschütz Ulcer of the Vulva in a Young Woman: Case Report and Review of the Literature

Received: 30 September 2015    Accepted: 16 October 2015    Published: 28 October 2015
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Abstract

Case: A case of 23-year-old woman, virgin, presenting a painful ulcerated vulvar lesion of the vestibular mucosa, with characteristics of Lipschütz ulcer, is reported. Lab tests showed no significant abnormalities, positivity for IgG of HSV1, rubeola, and VZV (with very high title: 2700 mUI/ml), positivity for EBV IgG of VCA and EBNA components and IgM negative (indicating a past infection serologic profile). A moderate insufficiency of Vitamin D was found. A treatment with topical antibiotics and oral antinflammatory drugs was planned and healing was complete without scars in less than 2 weeks. During a period of 6 months the lesion relapsed twice, with the same benign course of the first episode. DISCUSSION: such homogeneous clinical pattern is related to a heterogeneous etiologic spectrum, in which Epstein-Barr virus appears somehow prevalent, but not exclusive, as demonstrated by review of the literature. It is important to have well in mind this differential diagnosis, as Lipschütz ulcer may be overestimated and treated as for other more alarming causes of genital ulceration, with more complicated and unnecessary diagnostic and therapeutic procedures. These four points must be stressed: (1) Acute mononucleosis should always be in a clinician's differential diagnosis of Acute Genital Ulcer (AGU); (2) Failure to consider causes other than genital aphthosis (Behçet) or herpes may expose patients and their family to unnecessary investigations, treatments, and stress; (3) The symptoms of AGU are essentially nonspecific; (4) The physiopathology and aetiology of nonherpetic AGU still constitute broadly unexplored research fields in which further prospective clinical and microbiological studies are needed. Moreover, according to the findings of the reported case, a recurrent Lipschutz ulcer of the vulva in a young women could be related to Herpes Simplex or Varicella Zoster virus.

Published in Journal of Gynecology and Obstetrics (Volume 3, Issue 6)
DOI 10.11648/j.jgo.20150306.11
Page(s) 107-110
Creative Commons

This is an Open Access article, distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution and reproduction in any medium or format, provided the original work is properly cited.

Copyright

Copyright © The Author(s), 2024. Published by Science Publishing Group

Keywords

Vulva, Ulcus Vulvae Acutum, Lipschutz Ulcer, Recurrence, Varicella Zoster Virus

References
[1] Lipschütz B. Uber eine eigenartige Geschwürsform des weiblichen Genitales (Ulcus vulvae acutum). Arch Dermatol Syph (Berlin). 1913; 114: 363-96.
[2] Zeitsch. F. Geburtsh. U. Gynak. Womans Clin 1947; 128: 307-26.
[3] Berlin C. The pathogenesis of the so-called ulcus vulvae acutum. Acta Derm Venereol 1965; 45: 221-2.
[4] Bhat RM, Furtado S. Lipschutz ulcer. Indian J Sex Transm Dis 2007; 28: 106-7.
[5] Brinca A, Canelas MM, Carvalho MJ, Vieira R, Figueiredo A. Lipschütz ulcer (ulcus vulvae acutum) - a rare cause of genital lesion. An Bras Dermatol. 2012; 87(4): 622-4.
[6] Chanal J, Carlotti A, Laude H, Wallet-Faber N, Avril MF, Dupin N. Lipschütz genital ulceration associated with mumps. Dermatology. 2010; 221(4): 292-5.
[7] Wetter DA, Bruce AJ, MacLaughlin KL, Rogers RS 3rd. Ulcus vulvae acutum in a 13-year-old girl after influenza A infection. Skinmed. 2008; 7: 95-8.
[8] Pelletier F, Aubin F, Puzenat E, Deprez P, Blanc D, Estavoyer JM, et al. Lipschütz genital ulceration: rare manifestation of paratyphoid fever. Eur J Dermatol. 2003;13:297-8.
[9] Korting GW, Hinterberger G. Ulcus vulvae acutum with cold-agglutinin-positive, Mycoplasma-caused atypical pneumonia. Hautarzt. 1979; 30: 550-2.
[10] Rosman IS, Berk DR, Bayliss SJ, White AJ, Merritt DF. Acute genital ulcers in nonsexually active young girls: case series, review of the literature, and evaluation and management recommendations. Pediatr Dermatol. 2012; 29(2): 147-53.
[11] Farhi D, Wendling J, Molinari E, Raynal J, Carcelain G, Morand P, et al. Non-sexually related acute genital ulcers in 13 pubertal girls: a clinical and microbiological study. Arch Dermatol. 2009; 145: 38-45.
[12] Lynch PJ, Moyal-Barracco M, Bogliatto F, Micheletti L, Scurry J. 2006 ISSVD classification of vulvar dermatoses: pathologic subsets and their clinical correlates. Journal of Reproductive Medicine. 2007; 52: 3-9.
[13] International Study Group for Behcet’s Disease. Criteria for diagnosis of Behcet’s disease. Lancet, 1990; 335: 1078–1080.
[14] Housmans S, Moerman P, Amant F, Koninckx Ph, Donders G, and Verguts J. Vulvar ulcers: a differential diagnosis between Behçet’s disease and Lipschütz ulcus. Eur Clinics Obstet Gynaecol 2007, 3: 59–62.
[15] Martín JM, Godoy R, Calduch L, Villalon G, Jordá E. Lipschütz acute vulval ulcers associated with primary cytomegalovirus infection. Pediatr Dermatol. 2008; 25(1): 113-5.
[16] Huppert JS. Lipschutz ulcers: evaluation and management of acute genital ulcers in women. Dermatol Ther. 2010; 23(5): 533-40.
[17] Brown ZA, Stenchever MA. Genital ulceration and infectious mononucleosis: report of a case. Am J Obstet Gynecol 1977; 127: 673–674.
[18] Portnoy J, Ahronheim GA, Ghibu F, Clecner B, Joncas JH. Recovery of Epstein-Barr virus from genital ulcers. New Engl J Med. 1984; 311: 966-8.
[19] Wilson RW. Genital ulcer and Mononucleosis. Pediatr Infect Dis 1993; 12: 418.
[20] Lampert A, Assier-Bonnet H, Chevallier B, Clerici T, Saiag P. Lipschütz’s genital ulceration: a manifestation of Epstein Barr virus primary infection. Br J Dermatol 1996; 135: 663-5.
[21] Hudson LB, Periman SE. Necrotizing genital ulcerations in premenarcheal female with mononucleosis. Obstet Gynecol. 1998;92:185-7.
[22] Taylor S, Drake SM, Dedicoat M, Wood MJ. Genital ulcers associated with acute Epstein-Barr virus infection. Sex Transm Infect. 1998; 74(4): 296-7.
[23] Sisson BA, Glick L. Genital ulceration as a presenting manifestation of infectious mononucleosis. J Pediatr Adolesc Gynecol 1998; 11: 185-7.
[24] Cheng SX, Chapman MS, Margesson LJ, Birenbaum D. Genital ulcers caused by Epstein-Barr virus. J Am Acad Dermatol. 2004; 51: 824-6.
[25] Halvorsen JA, Brevig T, Aas T, Skar AG, Slevolden EM, Moi H. Genital ulcers as initial manifestation of Epstein-Barr virus infection: two new cases and a review of the literature. Acta Derm Venereol. 2006; 86(5): 439-42.
[26] Trĉko K, Beliĉ M, and Miljković J. Ulcus vulvae acutum. Acta Dermatoven APA 2007; 16: 174-6.
[27] Vieira-Baptista P, Lima-Silva J, Beires J, Martinez-de-Oliveira J. Lipschütz ulcers: should we rethink this? An analysis of 33 cases. Eur J Obstet Gynecol Reprod Biol. 2015 Aug 6. pii: S0301-2115(15)00253-5. [Epub ahead of print]
[28] Horie C, Kano Y, Takayo Mitomo T, Shiohara T. Possible Involvement of Mycoplasma fermentans in the Development of Nonsexually Acquired Genital Ulceration (Lipschütz Ulcers) in 3 Young Female Patients. JAMA Dermatol. Published online September 02, 2015.
[29] Haidari G, MacMahon E, Tong CY, White JA. Genital ulcers: it is not always simplex … Int J STD AIDS. 2015 Jan; 26(1): 72-3.
[30] Kinyó Á, Nagy N, Oláh J, Kemény L, Bata-Csörgő Z. Ulcus vulvae acutum Lipschütz in two young female patients. Eur J Dermatol. 2014 May-Jun; 24(3): 361-4.
Cite This Article
  • APA Style

    Carriero Carmine, Schonauer Luca Maria, Loverro Giuseppe. (2015). Unusual Multiple Recurrence of Lipschütz Ulcer of the Vulva in a Young Woman: Case Report and Review of the Literature. Journal of Gynecology and Obstetrics, 3(6), 107-110. https://doi.org/10.11648/j.jgo.20150306.11

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    ACS Style

    Carriero Carmine; Schonauer Luca Maria; Loverro Giuseppe. Unusual Multiple Recurrence of Lipschütz Ulcer of the Vulva in a Young Woman: Case Report and Review of the Literature. J. Gynecol. Obstet. 2015, 3(6), 107-110. doi: 10.11648/j.jgo.20150306.11

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    AMA Style

    Carriero Carmine, Schonauer Luca Maria, Loverro Giuseppe. Unusual Multiple Recurrence of Lipschütz Ulcer of the Vulva in a Young Woman: Case Report and Review of the Literature. J Gynecol Obstet. 2015;3(6):107-110. doi: 10.11648/j.jgo.20150306.11

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  • @article{10.11648/j.jgo.20150306.11,
      author = {Carriero Carmine and Schonauer Luca Maria and Loverro Giuseppe},
      title = {Unusual Multiple Recurrence of Lipschütz Ulcer of the Vulva in a Young Woman: Case Report and Review of the Literature},
      journal = {Journal of Gynecology and Obstetrics},
      volume = {3},
      number = {6},
      pages = {107-110},
      doi = {10.11648/j.jgo.20150306.11},
      url = {https://doi.org/10.11648/j.jgo.20150306.11},
      eprint = {https://article.sciencepublishinggroup.com/pdf/10.11648.j.jgo.20150306.11},
      abstract = {Case: A case of 23-year-old woman, virgin, presenting a painful ulcerated vulvar lesion of the vestibular mucosa, with characteristics of Lipschütz ulcer, is reported. Lab tests showed no significant abnormalities, positivity for IgG of HSV1, rubeola, and VZV (with very high title: 2700 mUI/ml), positivity for EBV IgG of VCA and EBNA components and IgM negative (indicating a past infection serologic profile). A moderate insufficiency of Vitamin D was found. A treatment with topical antibiotics and oral antinflammatory drugs was planned and healing was complete without scars in less than 2 weeks. During a period of 6 months the lesion relapsed twice, with the same benign course of the first episode. DISCUSSION: such homogeneous clinical pattern is related to a heterogeneous etiologic spectrum, in which Epstein-Barr virus appears somehow prevalent, but not exclusive, as demonstrated by review of the literature. It is important to have well in mind this differential diagnosis, as Lipschütz ulcer may be overestimated and treated as for other more alarming causes of genital ulceration, with more complicated and unnecessary diagnostic and therapeutic procedures. These four points must be stressed: (1) Acute mononucleosis should always be in a clinician's differential diagnosis of Acute Genital Ulcer (AGU); (2) Failure to consider causes other than genital aphthosis (Behçet) or herpes may expose patients and their family to unnecessary investigations, treatments, and stress; (3) The symptoms of AGU are essentially nonspecific; (4) The physiopathology and aetiology of nonherpetic AGU still constitute broadly unexplored research fields in which further prospective clinical and microbiological studies are needed. Moreover, according to the findings of the reported case, a recurrent Lipschutz ulcer of the vulva in a young women could be related to Herpes Simplex or Varicella Zoster virus.},
     year = {2015}
    }
    

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    T1  - Unusual Multiple Recurrence of Lipschütz Ulcer of the Vulva in a Young Woman: Case Report and Review of the Literature
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    AB  - Case: A case of 23-year-old woman, virgin, presenting a painful ulcerated vulvar lesion of the vestibular mucosa, with characteristics of Lipschütz ulcer, is reported. Lab tests showed no significant abnormalities, positivity for IgG of HSV1, rubeola, and VZV (with very high title: 2700 mUI/ml), positivity for EBV IgG of VCA and EBNA components and IgM negative (indicating a past infection serologic profile). A moderate insufficiency of Vitamin D was found. A treatment with topical antibiotics and oral antinflammatory drugs was planned and healing was complete without scars in less than 2 weeks. During a period of 6 months the lesion relapsed twice, with the same benign course of the first episode. DISCUSSION: such homogeneous clinical pattern is related to a heterogeneous etiologic spectrum, in which Epstein-Barr virus appears somehow prevalent, but not exclusive, as demonstrated by review of the literature. It is important to have well in mind this differential diagnosis, as Lipschütz ulcer may be overestimated and treated as for other more alarming causes of genital ulceration, with more complicated and unnecessary diagnostic and therapeutic procedures. These four points must be stressed: (1) Acute mononucleosis should always be in a clinician's differential diagnosis of Acute Genital Ulcer (AGU); (2) Failure to consider causes other than genital aphthosis (Behçet) or herpes may expose patients and their family to unnecessary investigations, treatments, and stress; (3) The symptoms of AGU are essentially nonspecific; (4) The physiopathology and aetiology of nonherpetic AGU still constitute broadly unexplored research fields in which further prospective clinical and microbiological studies are needed. Moreover, according to the findings of the reported case, a recurrent Lipschutz ulcer of the vulva in a young women could be related to Herpes Simplex or Varicella Zoster virus.
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Author Information
  • Dept. of Biomedical Sciences and Human Oncology, Section Gynaecology, University of Bari, Bari (Italy).

  • Dept. of Biomedical Sciences and Human Oncology, Section Gynaecology, University of Bari, Bari (Italy).

  • Dept. of Biomedical Sciences and Human Oncology, Section Gynaecology, University of Bari, Bari (Italy).

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