Acquired Cutaneous Lymphangiectasia Secondary to Cervical Cancer Treatment

Abstract

To the Editor: A 78-year-old female patient presented with a 10-year history of pedunculated nodules on her vulvar area. She remembered that the nodules had increased both in number and size over the past 10 years. The patient did not complain of any subjective symptoms, such as itching or pain. Her history included total hysterectomy and radiation therapy for cervical cancer 35 years previously, and transurethral resection of bladder tumor for bladder cancer 2 years ago with no other risk factors for lymphedema reported. Physical examination revealed multiple pinkish pedunculated translucent nodules on the labia majora (Fig. 1). Localized edema on the vulvar area was seen. A biopsy specimen was obtained through harvesting a whole nodule with a punch. The biopsy demonstrated ectatic channels in the superficial and deep dermis (Figs. 2A, B). The spaces between collagen bundles were widened, reflecting an edematous condition. No occlusive lesions or related vascular components were identified. The channel was diffusely labeled with D2–40 (Fig. 2C). A diagnosis of acquired cutaneous lymphangiectasia (ACL) was made. The patient refused treatment after realizing the benign nature of the skin lesion.FIGURE 1.: Edematous appearance of vulvar area with overlying multiple vesiculonodular lesions.FIGURE 2.: Low power (A, hematoxylin–eosin, ×40) and high power (B, hematoxylin–eosin, ×100) histologic features show dilated lymphatic channels in the papillary dermis with hyperkeratotic epidermis. No atypical cells are observed. Endothelial cell linings of ectatic channels show positive reaction to D2–40 stain (C, D2–40, ×100).Lymphedema manifests as a result of ineffective lymphatic drainage due to architectural disruption with resultant increased hydrostatic pressure.1 Common causes of acquired lymphatic compromise are surgery and radiation therapy for the treatment of malignancy. Other predisposing factors are recurrent cellulitis, Crohn disease, and urogenital tuberculosis.2 A key histologic finding for lymphedema is edematous widening of intercollagenous spaces in the papillary dermis.3 In a similar way, ACL represents acquired dilatation of lymphatic channels secondary to external causes, but it results from a more chronic process.4–6 Clinically, ACL consists of clustered translucent vesicles and nodules ranging from colorless to purple due to varying red blood cell content.7 At the time of the very first development, the new lesion appears rather vesicular while it gradually becomes nodular as a result of tissue reorganization. Histologically, ACL shows ectatic lymphatic vessels lined by normal endothelial cells.5 The overlying epidermis may show hyperkeratosis, acanthosis, and the effacement of rete ridges. The pathologic findings in this case were consistent with these features of both lymphedema and lymphangiectasia. Differential diagnosis of this unusual manifestation must include more common conditions such as genital warts, condyloma accuminatum, and molluscum contagiosum. Reported treatment options for ACL in the literature are surgical excision, laser vaporization, cryotherapy, sclerotherapy, and electrodessication. Because ACL is infamous for its high recurrence rate when deeper lymphatic abnormalities are not sufficiently destroyed, radical excision is often favored. Hamida et al,2 suggested CO2 laser ablation as a conservative alternative to surgical intervention because of high patient tolerance and good cosmetic results.