Indian J Sex Transm Dis Indian J Sex Transm Dis
Official Publication of the Indian Association for the Study of Sexually Transmitted Diseases
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CASE REPORT
Year : 2008  |  Volume : 29  |  Issue : 1  |  Page : 37-39
 

Vulval lymphangiomata secondary to tubercular inguinal lymphadenitis with HIV


Department of Dermatology, Venereology and Leprosy, J. J. M. Medical College, Davangere, Karnataka, India

Correspondence Address:
P Madavamurthy
Department of Dermatology, Venereology and Leprosy, J. J. M. Medical College, Davangere-577 004, Karnataka
India
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Source of Support: None, Conflict of Interest: None


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   Abstract 

Acquisition of vulval lymphangiomata is a rare occurrence; clinically characterized by the presence of circumscribed eruptions of thin walled, translucent vesicles. Here, we report a case of 18-year-old HIV-positive female with multiple smooth to warty, flesh colored papules and nodules covering labia and mons pubis with bilateral inguinal linear puckered scars with enlarged right cervical group of lymph nodes. Our case is unique because of its association with HIV.


Keywords: HIV positive, tubercular inguinal lymphadenopathy, vulval lymphangiomata


How to cite this article:
Madavamurthy P, Rajashekhar N, Chatura, Sugareddy, Desai PK. Vulval lymphangiomata secondary to tubercular inguinal lymphadenitis with HIV. Indian J Sex Transm Dis 2008;29:37-9

How to cite this URL:
Madavamurthy P, Rajashekhar N, Chatura, Sugareddy, Desai PK. Vulval lymphangiomata secondary to tubercular inguinal lymphadenitis with HIV. Indian J Sex Transm Dis [serial online] 2008 [cited 2020 Jul 2];29:37-9. Available from: http://www.ijstd.org/text.asp?2008/29/1/37/42715


Lymphangioma circumscriptum is a rare congenital malformation of the lymphatic system, characterized by persistent multiple clusters of translucent vesicles containing clear lymph fluid. Acquisition of vulval lymphangiomata is a rare occurrence; clinically characterized by the presence of circumscribed eruptions of thin walled, translucent vesicles. [1] However, epithelial changes and tissue organization may occasionally give rise to firmer nodules or even warty lesions, seen in association with scarring process due to recurrent infections, [2] radiotherapy, or surgeries. [3] We report such a case of vulval lymphangiomata due to tubercular inguinal lymphadenopathy in a HIV-positive patient.


   Case Report Top


An 18-year-old female patient presented to our OPD with complaints of genital lesions since one year. The patient had a history of fever and cough along with multiple swellings and ulcerations over both the inguinal regions. On examination, smooth to warty, flesh colored papules, vesicles, and nodules covering labia majora and mons pubis were present [Figure 1]. Bilateral inguinal puckered scars were present. Right cervical group of lymph nodes were enlarged, tender, and matted. Laboratory investigations showed routine blood and urine examinations within normal limits. HIV was reactive (by capillus, tridot, and Coombs methods), and Venereal Disease and Research Laboratory test as well as HbsAg were nonreactive. Chest X-ray showed resolving TB, FNAC of right cervical lymph nodes showed TB lymphadenitis, Mantoux showed strong positivity [Figure 2]. CD4 cell count was 515 cells/cumm. Histopathology showed acanthosis, papillomatosis, and hyperkeratosis of epidermis, dilated thin walled lymphatic channels in the superficial papillary dermis [Figure 3], dense infiltrate of lymphocytes and neutrophils in the dermis, and scattered neutrophils, plasma cells, and pigmented macrophages. The patient was initially treated with isoniazid 300 mg, rifampicin 450 mg, pyrazinamide 1500 mg, and ethambutol 800 mg for two months and subsequently with two drugs, isoniazid 300 mg and rifampicin 450 mg. The lesions regressed initially with one month treatment and thereafter remained static [Figure 4]. The patient was followed up for four months.


   Discussion Top


Acquired or secondary lymphangioma arise following damage to previously normal deep lymphatic vessels. [4] In lymphangioma circumscriptum, dilated superficial lymphatics occur due to a congenital anomaly of the deep dermal and subcutaneous lymphatics. The pathogenesis of lymphangioma circumscriptum was studied by Whimster, who concluded that large lymphatic vessels running mainly vertically through the dermis transmit pressure from underlying abnormal cisterns that do not communicate with the deeper dermal lymphatics, and therefore form a closed system. [5] Lymphangiomas can be congenital or can be acquired from local factors, such as surgery, neoplasms (including lymphoma), radiotherapy, [3] erysipelas, lymphogranuloma venereum, tuberculosis, or filariasis. [6] They are most often observed in the areas of neck, upper trunk, tongue, and proximal extremities. [7] Lymphangioma of the vulva are also described following cancer treatment, tuberculous inguinal lymphadenopathy, [8] and genital involvement with Crohn's disease. [9] Our case is following tubercular inguinal lymphadenopathy in association with HIV. Histopathologically also, it satisfies required findings consistent with acquired lymphangioma. Other treatment modalities include radical surgery, intralesional administration of sclerosant, for example, doxycycline [10] or picibanil [11] (OK-432), simple electricautery, and vaporization with CO 2 laser. [12] Recognition and appropriate treatment of vulval lymphangioma is important, primarily because the lesions may act as portals of entry for infections and secondarily to prevent complications. In addition, persistent leakage of lymphatic fluid, the cosmetic appearance, and the social repercussions of misdiagnosis as a STD may all pose significant problems.

 
   References Top

1.Harwood CA, Mortimer PS. Acquired vulvul lymphangiomata mimicking genital warts. Br J Dermatol 1993;129:334-6.  Back to cited text no. 1  [PUBMED]  
2.Buckley DA, Barnes L. Vulvar lymphangiectasia due to recurrent cellulitis. Clin Exp Dermatol 1996;21:215-6.  Back to cited text no. 2  [PUBMED]  
3.Jappee V, Zimmermann T, Kahle B, Petzoldt D. Lymphangioma circumscriptum of the vulva following surgical and radiological therapy of cervical cancer. Sex Trans Dis 2002;29:533-5.  Back to cited text no. 3    
4.Mortimer PS. Lymphatics. In: Champion R, Pye RJ, editors. Advances in dermatology. London: Churchill Livingstone; vol 8. 1989. p. 175-92.  Back to cited text no. 4    
5.Whimster IW. The pathology of lymphangioma circumscriptum. Br J Dermatol 1976;94:473-86.  Back to cited text no. 5  [PUBMED]  
6.Gupta S, Radotra BD, Javaheri SM, Kumar B. Lymphangioma circumscriptum of penis mimicking venereal lesions. J Eur Acad Dermatol Venereol 2003;17:598-600.  Back to cited text no. 6  [PUBMED]  [FULLTEXT]
7.Tsur H, Urson S, Schewach-Millet M. Lymphangioma circumscriptum of the glans penis. Cutis 1981;28:642-3.   Back to cited text no. 7  [PUBMED]  
8.Heuvel NV, Stolz E, Notowicz A. Lymphangiectases of the vulva in a patient with lymphnode tuberculosis. Int J Dermatol 1979;18:65-6.  Back to cited text no. 8  [PUBMED]  
9.Dap RF, van der Meijaden WI. Vulvar lymphangiectasia in Crohn's disease. Ned Tijdschr Geneeskd 2000;144:1692-5.   Back to cited text no. 9    
10.Molitch HI, Unger EC, Witte CL, vanSonnenberg E. Percutaneous sclerothrapy of lymphangiomas. Radiology 1995;194:343-7.  Back to cited text no. 10  [PUBMED]  [FULLTEXT]
11.Mikhail M, Kennedy R, Cramer B, Smith T. Sclerosing of recurrent lymphangioma using OK-432. J Pediater Surg 1995;30:1159-60.  Back to cited text no. 11    
12.Fliezzri YD, Sklar SA. Lymphangioma circumscriptum review and evaluation of CO2 laser vapourization. J Dermatol Surg Oncol 1988;141:357-36.  Back to cited text no. 12    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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