Tc-99m Sulfur Colloid Lymphoscintigraphy with Single-photon Emission Computed Tomography/Computed Tomography in a Case of Acquired Vulval Lymphangiomas.

We describe the lymphoscintigraphy findings of a 25-year-old female patient who was undergoing presurgical workup for lymphangiomas of the vulva. She had a history of treatment for disseminated tuberculosis 6 years back and presented with herpetiform oozing vesicles in the external genitalia. Single-photon emission computed tomography/computed tomography (SPECT/CT) confirmed cutaneous tracer accumulation in the vulval lesions and demonstrated the presence of densely calcified inguinal nodes secondary to healed tuberculosis as the etiology of secondary lymphangioma.

A 25-year-old female patient was referred to our department for Tc-99m sulfur colloid lymphoscintigraphy (SCLS). She was undergoing presurgical workup for lymphangioma of the vulva. She had a history of treatment for disseminated tuberculosis. At present, she had a gradually increasing swelling of the left lower limb and oozing vesicles in the vulva. Serial anterior static SCLS images [Figure 1a - 30 min, b - 1 h, and c - 3 h] revealed dermal backflow (black arrow) in the left lower limb and in the right thigh, with visualization of a few inguinal and iliac lymph nodes. Tracer accumulation was also noted in the vulvar region (white block arrow). Single-photon emission computed tomography/computed tomography (SPECT/CT) images [Figure 1d] showed skin thickening and tracer uptake in the mons pubis and labia majora region corresponding to multiple vesicles over labia and suprapubic area on inspection. Densely calcified right inguinal nodes (white arrow) and pelvic nodes were noted on CT.

Figure 1

Tc-99m sulfur colloid planar anterior and posterior images showing the dermal backflow and tracer accumulation in vulvar region (a) at 30 min, (b) at 60 min and (c) at 3 hours and single-photon emission computed tomography/computed tomography, (d) at the level of vulva showing tracer accumulation on the skin surface. Also noted was a calcified right inguinal node (arrow)

Lymphangiomas are malformations of lymphatic channels consisting of dilated lymphatic channels due to congenital or acquired causes. Acquired or secondary lymphangiomas are rare and occur following radiotherapy in carcinoma cervix, tuberculous inguinal adenitis,[12] pelvic and intra-abdominal surgeries,[3] scrofuloderma,[4] Crohn's disease,[5] scarring after keloids, recurrent cellulitis, and filariasis.[6] The lymphatic vessels of the superficial dermal plexus drain a fixed area of skin to the deep plexus. Damage to the deep lymphatic vessels leads to back pressure and dermal backflow, with subsequent dilatation of the superficial lymphatics as seen in this case. Densely calcified healed inguinal lymphadenitis was the underlying cause lymphatic obstruction leading to lower limb lymphedema and vulvar lymphangiomas which was well elucidated on SPECT/CT, thus aiding in understanding of the physiopathology of edema. Thus, SPECT/CT is a promising additional to lymphoscintigraphy in investigation of lymphangioma.

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