Ann Dermatol.  2019 Aug;31(4):450-453. 10.5021/ad.2019.31.4.450.

Cutaneous Lymphangioma Circumscriptum Occurred Seven Years after Surgery with Soft Tissue Cystic Lymphangioma

• Jung MJ
• Choi YW
• Son JH
• Kim HO
• Chung BY
• Park CW

• KMID: 2451485
• DOI: http://doi.org/10.5021/ad.2019.31.4.450

Abstract

Lymphangiomas are rare benign congenital lymphatic malformations. They can be divided into three groups: cutaneous lymphangioma circumscriptum (CLC), cavernous, and cystic. CLC is the most common type, and rarely occurs with cavernous or cystic lymphangioma under the lesion. Here, we describe the case of a 9-year-old girl who presented with an asymptomatic vesicular lesion on her back. She was finally diagnosed with CLC by clinical manifestations, dermoscopic findings, and histologic findings. Seven years ago, there was a history of surgical operation in the department of general surgery, and the surgery was soft tissue cystic lymphangioma removal surgery. There was no skin lesion for seven years after surgery, but one occurred a month ago. Herein, we present a case of CLC that occurred after a long interval after surgery for soft tissue cystic lymphangioma.

Keyword

Dermoscopy; Lymphangioma; Lymphangioma; cystic; Neoplasm recurrence; local

MeSH Terms

Child
Dermoscopy
Female
Humans
Lymphangioma*
Lymphangioma, Cystic*
Skin

Figure

• 

  Fig. 1 (A, B) Localized plaque composed of yellowish or red-colored vesicles and slightly warty papules, is observed on the back. (C) On chest computed tomography, a mass about 5.2×2.7 cm was observed on the right posterior chest wall seven years ago. (D) The boundary was clear and there was no fat attenuation, so a benign soft tissue tumor was suspected.

• 

  Fig. 2 (A) No soft tissue mass was observed on ultrasound. (B) By dermoscopy, light brownish lacunae with pale septa and dark-reddish areas in some lacunae were observed. Hypopyon-like features suggesting lymphangioma were observed and marked with arrows. (C, D) Histopathologic examination showed a large number of irregularly enlarged lymphatic vessels bordered by very thin lymphatic endothelial cells in the upper dermis. A pale pink lymphatic fluid was was observed (H&E: C, ×40; D, ×100).

Reference

1. Patel GA, Schwartz RA. Cutaneous lymphangioma circumscriptum: frog spawn on the skin. Int J Dermatol. 2009; 48:1290–1295.

2. Samdani AJ, Bana SH, Azam T. Lymphangioma circumscriptum: report of three cases from Saudi Arabia. Int J Dermatol. 2006; 45:840–842.

3. Bauer BS, Kernahan DA, Hugo NE. Lymphangioma circumscriptum--a clinicopathological review. Ann Plast Surg. 1981; 7:318–326.

4. Morris M. Lymphangioma circumscriptum. In : Unna PG, Morris M, Duhring LA, Leloir H, editors. International atlas of rare skin diseases. London: Lewis;1889. p. 1–4.

5. Massa AF, Menezes N, Baptista A, Moreira AI, Ferreira EO. Cutaneous Lymphangioma circumscriptum-dermoscopic features. An Bras Dermatol. 2015; 90:262–264.

6. Arpaia N, Cassano N, Vena GA. Dermoscopic features of cutaneous lymphangioma circumscriptum. Dermatol Surg. 2006; 32:852–854.

7. Amini S, Kim NH, Zell DS, Oliviero MC, Rabinovitz HS. Dermoscopic-histopathologic correlation of cutaneous lymphangioma circumscriptum. Arch Dermatol. 2008; 144:1671–1672.

8. Gencoglan G, Inanir I, Ermertcan AT. Hypopyon-like features: new dermoscopic criteria in the differential diagnosis of cutaneous lymphangioma circumscriptum and haemangiomas? J Eur Acad Dermatol Venereol. 2012; 26:1023–1025.

9. Fioramonti P, Maruccia M, Ruggieri M, Onesti MG. A rare case of lymphangioma in the gluteal region: surgical treatment combined with sclerotherapy and laser therapy. Aesthetic Plast Surg. 2013; 37:960–964.

10. Browse NL, Whimster I, Stewart G, Helm CW, Wood JJ. Surgical management of 'lymphangioma circumscriptum'. Br J Surg. 1986; 73:585–588.

11. Alqahtani A, Nguyen LT, Flageole H, Shaw K, Laberge JM. 25 years' experience with lymphangiomas in children. J Pediatr Surg. 1999; 34:1164–1168.

12. Seber S, Köse N, Turgut A, Ozçelik A, Göktürk E. Giant cystic lymphangioma cavernousum of lower limb with overlying lymphangioma circumscriptum. J Pediatr Orthop B. 2004; 13:284–286.

13. Irvine AD, Sweeney L, Corbett JR. Lymphangioma circumscriptum associated with paravesical cystic retroperitoneal lymphangioma. Br J Dermatol. 1996; 134:1135–1137.

14. Rao MV, Thappa DM, Ratnakar C. Lymphangioma circumscriptum of the skin and tongue associated with cystic hygroma. Indian J Otolaryngol Head Neck Surg. 1998; 50:266–268.

15. Flanagan BP, Helwig EB. Cutaneous lymphangioma. Arch Dermatol. 1977; 113:24–30.

16. Preeti A, Kumar R. Cystic hygroma: cytological and radiological co-relation. J Clin Diagn Res. 2011; 5:1008–1010.