Effect of injection bleomycin on lymphangioma circumscriptum with keloid formation and secondary infection

Ann Willie Thomas; Emy Abi Thomas; Abhilasha Williams

doi:10.4103/cjhr.cjhr_84_22

CASE REPORT

Year : 2022 | Volume : 9 | Issue : 4 | Page : 288-289

Effect of injection bleomycin on lymphangioma circumscriptum with keloid formation and secondary infection

Ann Willie Thomas, Emy Abi Thomas, Abhilasha Williams
Department of Dermatology, Christian Medical College, Ludhiana, Punjab, India

Date of Submission	05-Aug-2022
Date of Decision	30-Nov-2022
Date of Acceptance	10-Dec-2022
Date of Web Publication	17-Mar-2023

Correspondence Address:
Ann Willie Thomas
Department of Dermatology, Christian Medical College, Brown Road, Ludhiana - 141 008, Punjab
India

Source of Support: None, Conflict of Interest: None

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DOI: 10.4103/cjhr.cjhr_84_22

Abstract

Lymphangioma circumscriptum is a microcystic lymphatic malformation localized to an area of the skin, subcutaneous tissue, and muscle which presents with vesiculo-papules or warty lesions, resembling a “frog spawn.” Keloids develop due to the proliferation of dermal tissue following skin injury. We present a case of lymphangioma circumscriptum with secondary infection and keloid formation treated with intralesional bleomycin.

Keywords: Bleomycin, keloids, lymphangioma circumscriptum

How to cite this article:
Thomas AW, Thomas EA, Williams A. Effect of injection bleomycin on lymphangioma circumscriptum with keloid formation and secondary infection. CHRISMED J Health Res 2022;9:288-9

How to cite this URL:
Thomas AW, Thomas EA, Williams A. Effect of injection bleomycin on lymphangioma circumscriptum with keloid formation and secondary infection. CHRISMED J Health Res [serial online] 2022 [cited 2023 Apr 1];9:288-9. Available from: https://www.cjhr.org/text.asp?2022/9/4/288/371946

Introduction

Lymphangioma circumscriptum is a microcystic lymphatic malformation which presents with vesiculo-papules or warty lesions, resembling a “frog spawn” at birth or during childhood. The usual sites involve the proximal parts of the limbs, flanks, tongue, or perineum.^[1] Keloids develop due to the proliferation of dermal tissue following skin injury.^[2] Although frequently asymptomatic, patients usually seek therapy due to complications such as lymphorrhea, infection, or for cosmetic reasons. We herein describe a case of lymphangioma circumscriptum with secondary infection and keloid formation treated with intravenous antibiotics and intralesional bleomycin.

Case Report

A 24-year-old male presented to our dermatology outpatient department with complaints of fluid-filled lesions on the left upper back and arms from 14 years of age which gradually progressed in size and number. He also noticed pus-filled lesions over it for 2 days and a fever for 1 day. Cutaneous examination revealed multiple translucent, red-to-blue–black vesicles (lymph blisters), and a few warty papules bulging on the skin surface noted at the left upper back, along with multiple keloids over it. Multiple scattered vesicles at the posterior aspect of the left arm and shoulder were seen giving a frog spawn appearance [Figure 1]a. Nails, scalp, oral cavity, and genitalia appeared normal. Skin biopsy showed cystically dilated superficial lymphatics, filled with pale eosinophilic secretions accompanied by lymphocytes, histiocytes, neutrophils, melanophages, and few red blood cells. The adjoining dermis showed dense inflammatory B-cell infiltrate composed of lymphocytes, histiocytes, plasma cells, and rare eosinophils [Figure 2]. We decided to treat the patient with a 3-day course of antibiotics for secondary infection and injection bleomycin for lymphangioma circumscriptum and keloid formation.

Figure 1: (a) Multiple translucent, red-to-blue–black vesicles (lymph blisters), few warty papules bulging on the skin surface, with multiple keloids over it noted at the left upper back giving a frog spawn appearance, (b) 2 weeks later, and (c) 4 weeks later

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Figure 2: Cystically dilated superficial lymphatics, filled with pale eosinophilic secretions accompanied by lymphocytes, histiocytes, neutrophils, melanophages, and few RBCs. RBC: Red blood cell

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Injection augmentin 1.2 g Q8 hourly after sensitivity testing was administered for 3 days. After the secondary infection improved, injection bleomycin 1 mg/ml (2 mg) was injected intralesionally. The injections were largely aimed at the lymphatic channels in the papillary dermis. The patient was regularly followed up biweekly [Figure 1]b and injection bleomycin 1 mg/ml (2 mg) was repeated after 4 weeks [Figure 1]c. Then, it was repeated on a monthly basis. Following this, good improvement was observed in both lymphangioma and keloid.

Discussion

Vascular malformations (VMs) are progressively enlarging aberrant and ectatic vessels, which may be capillary, venous, arterial, lymphatic, and combined malformations.^[3] Histologically, a lymphatic malformation is characterized by the size of malformed channels: either microcystic, macrocystic, or combined.^[4] Lymphangioma circumscriptum is a microcystic lymphatic malformation localized to an area of the skin, subcutaneous tissue, and muscle.^[3] Keloids develop due to the proliferation of dermal tissue following skin injury.^[2] Different treatment modalities such as surgical excision, ablative lasers, sclerotherapy, cryosurgery, and electrocoagulation have been used with varying results. Various sclerosing agents such as hypertonic saline, picibanil, doxycycline, and polidocanol were used previously.^[1] Bleomycin in the strength of 1 mg/ml with lignocaine has sclerosing effects on endothelial cells by inciting a mild inflammatory effect which can be used off-label in recalcitrant warts, keloids, hypertrophic scars, VMs, condyloma acuminate, cutaneous malignancies, etc., It has antibacterial, antineoplastic, antiviral, and antiangiogenic properties. Local anesthetics disrupt cell membrane structure, thereby increasing bleomycin uptake and enhancing its cytotoxicity. Cutaneous toxicities at the site of injection include local pain, erythema, swelling, ulceration, hyperpigmentation, fibrosis, and gangrene.^[3] Our patient did not develop any of the side effects except pain during the injection of bleomycin.

Conclusion

Lymphangioma circumscriptum with keloid formation is a rarely encountered entity. Bleomycin injection (intralesional) along with lignocaine is a safe and effective sclerosant for both lymphangioma circumscriptum and keloid.

Declaration of patient consent

The patient understands that his name and initials will not be published and due efforts will be made to conceal his/her identity.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

References

1.	Khurana A, Gupta A, Ahuja A, Sardana K, Malhotra P. Lymphangioma circumscriptum treated with combination of Bleomycin sclerotherapy and Radiofrequency ablation. J Cosmet Laser Ther 2018;20:326-9.
2.	Farahnaz FN, Jamshid N, Koroush A. Comparison of therapeutic response of keloids and hypertrophic scars to cryotherapy plus intralesional steroid and bleomycin tattoo. Indian J Dermatol 2005;50:129-32. [Full text]
3.	Sharma RK, Gupta M. Intralesional bleomycin in lymphangioma circumscriptum of tongue. Indian J Drugs Dermatol 2018;4:82-4. [Full text]
4.	Greene AK, Perlyn CA, Alomari AI. Management of lymphatic malformations. Clin Plast Surg 2011;38:75-82.