lymphangioma of the penis: a rare anomaly

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CASE REPORT Amar Shah Lisa Meacock Bharat More Harish Chandran Lymphangioma of the penis: a rare anomaly Accepted: 5 November 2003 / Published online: 6 January 2005 Ó Springer-Verlag 2005 Abstract Lymphatic malformations are known to affect any part of the body. However, lymphangiomas involving the penile skin are rare. We report a case of a cutaneous lymphatic malformation of the penis. Keywords Penis Lymphangioma Child Case report An 11-year-old boy was referred with a swelling over the dorsal aspect of his penis. The swelling was noticed incidentally and had grown over a period of a few months. There was no history of trauma nor of any urinary complaints. Clinical examination showed a soft mass on the dorsal aspect of the penis. The full blood count and peripheral smear were normal. A magnetic resonance imaging scan showed a fluid-filled subcuta- neous lesion of the penis with some extension towards the root of the right hemiscrotum. There was no high flow or postcontrast enhancement. The patient underwent local resection of the lesion. Dissection extended to the level of Buck’s fascia of the penis (Fig. 1). The rest of the skin was trimmed and reapposed (Fig. 2). Microscopic examination revealed thickened epidermis with dilated lymphatics in the der- mis. Multinucleate giant cells were present in lymphoid aggregates, with loose fibroconnective stoma. The find- ings confirmed a lymphangiomatous malformation of the penile skin. The boy made an uneventful postoperative recovery. Eleven months after the procedure, minimal fullness is noted on the lateral aspect of the penis, suggesting recurrence. However, it is not causing any symptoms or cosmetic disfigurement. The boy is presently in regular follow-up with a plan for staged resection if the size of the lesion increases or causes disfigurement. Discussion Lymphangiomas are benign hamartomatous tumours of the lymphatic system characterised by multiple com- municating lymphatic channels and cystic spaces. Many lymphangiomas involve the skin and subcutaneous tis- sues, and association with vascular malformations is frequent. The common sites of involvement are the tongue, cheek, neck, chest, buttocks, extremities, and peritoneal cavity. We found only two previous reports in the English literature of lymphangioma affecting the penile skin [1, 2]. Lymphangiomas are now considered lymphatic malformations not communicating with the lymphatic system. Cutaneous lymphatic malformations may arise de novo or secondary to surgery or irradiation [3]. Minor trauma may also cause the lesions to become symptomatic. However, inflammatory, parasitic, and other causes of congenital and acquired lymphoedema need to be ruled out. Surgical excision is the treatment of choice [4]. Staged excision is required in at least one-third of cases [5]. Laser fulguration is described [6], but the treatment must be individualised according to the location of the lesion and preservation of vital functions. Other modalities of management, such as radiation treatment or injection of sclerosing agents, have not been of benefit in treating lymphangiomas. OK-432, a monoclonal antibody pro- duced by incubation and interaction of Streptococcus pyogenes and penicillin, can be used in extremely large cystic lesions in locations difficult to manage surgically. In our patient, all macroscopically abnormal tissue was excised with preservation of sufficient penile skin. Recurrence may necessitate staged excision, with or without skin grafting. A. Shah L. Meacock B. More H. Chandran (&) Paediatric Urology, Birmingham Children’s Hospital, Steelhouse Lane, Birmingham, B4 6NH, UK E-mail: [email protected] Tel.: +44-121-3338068 Pediatr Surg Int (2005) 21: 329–330 DOI 10.1007/s00383-004-1346-9

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Page 1: Lymphangioma of the penis: a rare anomaly

CASE REPORT

Amar Shah Æ Lisa Meacock Æ Bharat More

Harish Chandran

Lymphangioma of the penis: a rare anomaly

Accepted: 5 November 2003 / Published online: 6 January 2005� Springer-Verlag 2005

Abstract Lymphatic malformations are known to affectany part of the body. However, lymphangiomasinvolving the penile skin are rare. We report a case of acutaneous lymphatic malformation of the penis.

Keywords Penis Æ Lymphangioma Æ Child

Case report

An 11-year-old boy was referred with a swelling over thedorsal aspect of his penis. The swelling was noticedincidentally and had grown over a period of a fewmonths. There was no history of trauma nor of anyurinary complaints. Clinical examination showed a softmass on the dorsal aspect of the penis. The full bloodcount and peripheral smear were normal. A magneticresonance imaging scan showed a fluid-filled subcuta-neous lesion of the penis with some extension towardsthe root of the right hemiscrotum. There was no highflow or postcontrast enhancement.

The patient underwent local resection of the lesion.Dissection extended to the level of Buck’s fascia of thepenis (Fig. 1). The rest of the skin was trimmed andreapposed (Fig. 2). Microscopic examination revealedthickened epidermis with dilated lymphatics in the der-mis. Multinucleate giant cells were present in lymphoidaggregates, with loose fibroconnective stoma. The find-ings confirmed a lymphangiomatous malformation ofthe penile skin.

The boy made an uneventful postoperative recovery.Eleven months after the procedure, minimal fullness is

noted on the lateral aspect of the penis, suggestingrecurrence. However, it is not causing any symptoms orcosmetic disfigurement. The boy is presently in regularfollow-up with a plan for staged resection if the size ofthe lesion increases or causes disfigurement.

Discussion

Lymphangiomas are benign hamartomatous tumours ofthe lymphatic system characterised by multiple com-municating lymphatic channels and cystic spaces. Manylymphangiomas involve the skin and subcutaneous tis-sues, and association with vascular malformations isfrequent. The common sites of involvement are thetongue, cheek, neck, chest, buttocks, extremities, andperitoneal cavity. We found only two previous reports inthe English literature of lymphangioma affecting thepenile skin [1, 2]. Lymphangiomas are now consideredlymphatic malformations not communicating with thelymphatic system. Cutaneous lymphatic malformationsmay arise de novo or secondary to surgery or irradiation[3]. Minor trauma may also cause the lesions to becomesymptomatic. However, inflammatory, parasitic, andother causes of congenital and acquired lymphoedemaneed to be ruled out.

Surgical excision is the treatment of choice [4]. Stagedexcision is required in at least one-third of cases [5].Laser fulguration is described [6], but the treatment mustbe individualised according to the location of the lesionand preservation of vital functions. Other modalities ofmanagement, such as radiation treatment or injection ofsclerosing agents, have not been of benefit in treatinglymphangiomas. OK-432, a monoclonal antibody pro-duced by incubation and interaction of Streptococcuspyogenes and penicillin, can be used in extremely largecystic lesions in locations difficult to manage surgically.

In our patient, all macroscopically abnormal tissuewas excised with preservation of sufficient penile skin.Recurrence may necessitate staged excision, with orwithout skin grafting.

A. Shah Æ L. Meacock Æ B. More Æ H. Chandran (&)Paediatric Urology, Birmingham Children’s Hospital,Steelhouse Lane, Birmingham, B4 6NH, UKE-mail: [email protected].: +44-121-3338068

Pediatr Surg Int (2005) 21: 329–330DOI 10.1007/s00383-004-1346-9

Page 2: Lymphangioma of the penis: a rare anomaly

References

1. Luis L, Ortega P, Paez A, et al. (2002) Cavernous lymphangiomaof the prepuce. Br J Urol 28:138–139

2. Sadikoglu B, Kuran L, Ozcan H, et al. (1999) Cutaneous lym-phatic malformation of the penis and scrotum. J Urol 162:1445–1446

3. Enzinger FM, Weiss SW (1995) Tumors of lymph vessels. In:Enzinger FM, Weiss SW (eds) Soft tissue tumors, 3rd edn.Mosby, Philadelphia, p 683

4. Bauer BS, Kernahan DA, Hugo NE (1981) Lymphangiomacircumscriptum. A clinicopathological review. Ann Plast Surg7:318

5. Feins N (1998) Lymphatic disorders. In: O’Neill JA Jr, RoweMI, Grosfeld JL, Fonkalsrud EW, Coran AG (eds) Pediatricsurgery, 5th edn. Mosby, St. Louis, pp 1973–1981

6. Demir Y, Latifoglu O, Yenidunya S, Atabay K (2001) Extensivelymphatic malformation of penis and scrotum. Urology 58:105–106

Fig. 1 Photograph showing resection of the lesion with dissectiondown to the Buck’s fascia

Fig. 2 Postoperative picture

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