Author Information
Mhaske N*,
(* Second Year Resident,
** Additional Professor, *** Assistant Professor, **** Senior Resident. Department
of Obstetrics/Gynecology, LTMMC and LTMGH, Mumbi , India
Abstract
Lymphangiectasias are acquired lymphatic
disorders. Vulval lymphangiectasias can mimic other disorders. Vulval
lymphangiectasias occurring with pregnancy is a rarity. A case of
post-tuberculous vulval lymphangiectasia accompanying pregnancy is described
here.
Introduction
Common vulval disorders
in pregnancy include viral warts and herpetic lesions. Rare lesions such as
lymphangiectasia can also exist, especially in the setting of risk factors like
previous tuberculosis or malignancy and radiotherapy.
Case Report
A 24 year old
primigravida presented in outpatient department in early second trimester with
vulval vesicular lesions. Patient had tuberculous lymphadenitis of neck three
years prior, with no involvement of other systems, for which treatment was
completed. Lesions in the vulval region, initially noticed few months prior to
pregnancy, were gradually progressive. There were no complaints of pain,
bleeding, discharge or pruritus.
Local genital
examination revealed multiple erythematous grouped vesicles over the vulval and
pubic region, resembling the classically described “frogspawn” appearance. There was no regional lymphadenopathy or
lower limb edema. Specialist
dermatological opinion was taken and clinical diagnosis of lymphangiectasia was
reached. Watchful expectancy was maintained. Investigations including
urinalysis, HIV, HBsAg, HCV,VDRL were all negative. Antenatal visits were
regular, uneventful.
Figure 1. Multiple
papulovesicular lesions over the vulva.
Onset of labor was
spontaneous at 38 weeks of gestation. Abdominal examination revealed a full
term uterus with a live fetus in cephalic presentation and per vaginal
examination revealed adequate pelvis. As shown in figure 1, vulval examination
revealed multiple vesicular lesions.
In the second stage of
labor, with descent of the presenting part, the labia splayed out and the
vesicular lesions got displaced laterally. Thus there was adequate room for a
mediolateral episiotomy, delivering a healthy female child of 2570 g.
Episiotomy was sutured without disturbing the lesions. Oral antibiotics were
given as per usual hospital protocol. Post-delivery period was uneventful.
Post-delivery biopsy
(figure 2) showed hyperkeratotic atrophic epidermis, multiple dilated lymphatic
channels, chronic inflammatory cells and dilated capillaries, suggestive of
lymphangiectasis secondary to chronic infective condition. Post-delivery follow
up showed a mild regression in the size of the lesions, and the patient
continues to be asymptomatic.
Figure 2.
Histopathological appearance of the lesion.
Discussion
Lymphangiectasia is a
disorder of superficial lymphatics which results from an obstruction of
previously normal deep lymphatics.[1] It has been long recognized
that vulval skin is susceptible to lymphangiectasia, the symptoms of which
include vesicular lesions, pain, bleeding and itching.[2]
Few cases of vulval
lymphangiectasia have been reported[3] and this entity has been
described as a diagnostic challenge.[4] While many causes exist, the
associations include malignancy, radiotherapy, tuberculosis and prior pelvic
surgical procedures.[5,,6,7,8] Female genital mutilation has also
been described as a cause.[9]
Lymphangiectasias
associated with pregnancy and spontaneous regression postpartum have been
described as a rarity that needs to be reported.[10] Management
options include watchful observation, carbon dioxide laser, immunotherapy and
surgery.[11]
In pregnancy, a
confident diagnosis is needed, because this entity needs to be distinguished
from conditions like viral warts and herpes, [3,12] which have different labor
management, including avoidance of
vaginal delivery. It is hoped that this report will add to existing knowledge
about this entity during pregnancy.
References
- Huilgol SC, Neill S, Barlow RJ. CO2 laser therapy of vulval lymphangiectasia and lymphangioma circumscriptum. Dermatol Surg 2002;28(7):575–7.
- Handfield-Jones SE, Prendiville WJ, Norman S. Vulval lymphangiectasia. Genitourin Med 1989;65(5):335–7.
- Haneef NS, Ramachandra S, Metta AK, Haritha K. Lymphangiectasias of vulva. Indian Dermatol Online J. 2011; 2(1): 40–42. Available from: http://www.idoj.in/text.asp?2011/2/1/40/79854
- Vlastos AT, Malpica A, Follen M. Lymphangioma circumscriptum of the vulva: a review of the literature. Obstet Gynecol 2003;101(5 Pt 1):946–54.
- Ghaemmaghami F, Karimi Zarchi M, Mousavi A. Major labiaectomy as surgical management of vulvar lymphangioma circumscriptum: three cases and a review of the literature. Arch Gynecol Obstet. 2008;278(1):57–60
- Spratt EAG, Batra P, Fischer MK, Pomeranz MK. Vesicular lesions on the vulva. Arch Dermatol. 2012;148(6):755–60.
- Stewart CJR, Chan T, Platten M. Acquired lymphangiectasia (’lymphangioma circumscriptum') of the vulva: a report of eight cases. Pathology 2009;41(5):448–53.
- Riyaz N, Nair VL. Cutaneous lymphangiectasia secondary to lymph node tuberculosis. Indian J Dermatol Venereol Leprol; 2000;66(6):314–5.
- Franco G, Toma L, Nosotti L, Muscardin LM, Morrone A. Vulvar lymphangiectases mimicking genital warts in female genital mutilation. Eur J Dermatol 2006;16(5):587–8.
- Verma S. Pregnancy-induced lymphangiectasias of the vulva. Int J STD AIDS. 2008; 19(3):211-2. doi:10.1258/ijsa.2007.007239
- Loche F, Schwarze HP, Bazex J. Treatment of acquired cutaneous lymphangiectasis of the thigh and vulva with a carbon dioxide laser. Acta Derm Venereol. 1999;79(4):335.
- Sharma R, Tomar S, Chandra M. Acquired vulval lymphangiectases mimicking genital warts. Indian J Dermatol Venereol Leprol; 2002;68(3):166–7.
Citation
Mhaske N, Fonseca
MN