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Vulvar Langerhans cell histiocytosis: a case report.

Khoummane N, Guimeya C, Lipombi D, Gielen F - Pan Afr Med J (2014)

Bottom Line: The patient presented since 3 years with a vulvar lesion characterized by non-healing ulcers and a perineal granuloma on which she underwent surgery.Professionals should keep in mind not to treat straightforwardly lesions of the genital tract as simple sexually transmitted diseases.Chronic, atypical genital lesions seen in women need to be worked up and dealt with accordingly.

View Article: PubMed Central - PubMed

Affiliation: Department of Gynecology and Obstetrics, Oncology and High Risk Pregnancies- Maternité Souissi, Rabat, Morocco.

ABSTRACT
Langerhans cell histiocytoses (LCH) are a rare group of disorders that comprise a large spectrum of diseases initially known as histiocytosis X. In this case report, we relate a case of LCH affecting the vulva of a 47-year-old female. The patient presented since 3 years with a vulvar lesion characterized by non-healing ulcers and a perineal granuloma on which she underwent surgery. Professionals should keep in mind not to treat straightforwardly lesions of the genital tract as simple sexually transmitted diseases. Chronic, atypical genital lesions seen in women need to be worked up and dealt with accordingly.

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Macroscopic aspect of vulvar langerhans cell histiocytosis
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Figure 0001: Macroscopic aspect of vulvar langerhans cell histiocytosis

Mentions: The patient is a 47 year old, with no significant past medical history, presenting since 2009 with a perineal lesion for which a histopathological examrevealeda perineal granuloma. The patient sought consultation in June 2012 with the same complaint: a vulvar lesion for which she was operated a month later in July 2012. The lesion was excised, sent to the lab and the diagnosis of Langerhans cell histiocytosis surfaced (Figure 1, Figure 2, Figure 3, Figure 4, Figure 5). In this context, in order to determine if the histiocytosis is localized at a gynecological or systemic level, the patient underwent blood tests, a 24h urine collection, a chest and abdominal CT scan and a bone scintigraphy. Blood tests were within normal limits except for a slight eosinophilia (430/mm3). Renal function, hemogramme, liver function tests were all normal. We noticed minimal elevation in alkaline phosphatase reaching 260 U/l. Protein electrophoresis was in favor of an inflammation with elevated alpha-2 globulins: erythrocyte sedimentation rate was at 69mm/H with a normal C-reactive protein measuring 0.19mg/dl. Thyroid hormone levels as well as LH, FSH, Prolactin, cortisol, were all within normal limits. Urine osmolarity was normal and chest/abdominal CT scan was unremarkable. Bone scintigraphy did not show any specific lesion. There was no evidence of disease beyond the vulva. After working up the patient, we concluded to the diagnosis of Langerhans cell histiocytosis located in the vulvar area.


Vulvar Langerhans cell histiocytosis: a case report.

Khoummane N, Guimeya C, Lipombi D, Gielen F - Pan Afr Med J (2014)

Macroscopic aspect of vulvar langerhans cell histiocytosis
© Copyright Policy - open-access
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC4232193&req=5

Figure 0001: Macroscopic aspect of vulvar langerhans cell histiocytosis
Mentions: The patient is a 47 year old, with no significant past medical history, presenting since 2009 with a perineal lesion for which a histopathological examrevealeda perineal granuloma. The patient sought consultation in June 2012 with the same complaint: a vulvar lesion for which she was operated a month later in July 2012. The lesion was excised, sent to the lab and the diagnosis of Langerhans cell histiocytosis surfaced (Figure 1, Figure 2, Figure 3, Figure 4, Figure 5). In this context, in order to determine if the histiocytosis is localized at a gynecological or systemic level, the patient underwent blood tests, a 24h urine collection, a chest and abdominal CT scan and a bone scintigraphy. Blood tests were within normal limits except for a slight eosinophilia (430/mm3). Renal function, hemogramme, liver function tests were all normal. We noticed minimal elevation in alkaline phosphatase reaching 260 U/l. Protein electrophoresis was in favor of an inflammation with elevated alpha-2 globulins: erythrocyte sedimentation rate was at 69mm/H with a normal C-reactive protein measuring 0.19mg/dl. Thyroid hormone levels as well as LH, FSH, Prolactin, cortisol, were all within normal limits. Urine osmolarity was normal and chest/abdominal CT scan was unremarkable. Bone scintigraphy did not show any specific lesion. There was no evidence of disease beyond the vulva. After working up the patient, we concluded to the diagnosis of Langerhans cell histiocytosis located in the vulvar area.

Bottom Line: The patient presented since 3 years with a vulvar lesion characterized by non-healing ulcers and a perineal granuloma on which she underwent surgery.Professionals should keep in mind not to treat straightforwardly lesions of the genital tract as simple sexually transmitted diseases.Chronic, atypical genital lesions seen in women need to be worked up and dealt with accordingly.

View Article: PubMed Central - PubMed

Affiliation: Department of Gynecology and Obstetrics, Oncology and High Risk Pregnancies- Maternité Souissi, Rabat, Morocco.

ABSTRACT
Langerhans cell histiocytoses (LCH) are a rare group of disorders that comprise a large spectrum of diseases initially known as histiocytosis X. In this case report, we relate a case of LCH affecting the vulva of a 47-year-old female. The patient presented since 3 years with a vulvar lesion characterized by non-healing ulcers and a perineal granuloma on which she underwent surgery. Professionals should keep in mind not to treat straightforwardly lesions of the genital tract as simple sexually transmitted diseases. Chronic, atypical genital lesions seen in women need to be worked up and dealt with accordingly.

Show MeSH
Related in: MedlinePlus