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Clinical Brief
Bleomycin Induced Hyperpigmentation with Yolk Sac Tumor Nilgun Yaris1, Murat Cakir2, Mukaddes Kalyoncu2 and Aysenur Okten2 Departments of 1Pediatric Oncology, and 2Pediatrics Karadeniz Technical University, Faculty of Medicine, Trabzon, Turkey. [Received May 1, 2006; Accepted November 9, 2006]
ABSTRACT Hyperpigmentation is one of the cutaneous side effects of chemotherapautic agents, but it is usually accepted as a cosmetic problem. We report a child with yolk sac tumor who developed localized pigmentation after the first course of chemotherapy regimen that included cisplatin, etoposide and bleomycin. The hyperpigmentation was diffuse scattered, flagellate like and linear streaking which was thought to be mainly related to the skin toxicity of bleomycin. [Indian J Pediatr 2007; 74 (5) : 505-506] E-mail:
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Key words : Bleomycin-induced; Flagellate like; Hyperpigmentation
There are several side effects of chemotherapeutic agents that affect the function of different organs. Some of these side effects are life treating, while others merely limit the usefulness of the drug. 1 Although dermatological complications may be less serious, they can result in significant morbidity or even mortality such as extravasations. Most dermatological problems are cosmetic such as alopecia and hyperpigmentation.2
areas of linear streaking, dark brown hyperpigmentated streaking was noted over the neck, shoulders, and left thoracic and abdominal wall (Fig. 1). The skin lesions were not pruritic. Renal and hepatic functions were normal. The linear hyperpigmentation of the skin got lighter and never disappeared during 24 mth follow-up. A variety of chemotherapeutic agents may induce hyperpigmentation including alkylating agents, antibiotics and antimetabolites. 2 The skin, mucous
CASE REPORT A previously healthy 4-yr-old girl was admitted to authors hospital with a mass on her back. On physical examination, there was a mass 4 × 5 cm in diameter over her sacrococcygeal region and a mass was palpated in her pelvis. MRI showed a multilobular mass 8×12×13 cm in diameter, arising from sacrococcygeal region that had similar extrapelvic and intrapelvic extension. Her alfa fetoprotein level was 6413 ng/ml. After total excision of the mass, a yolk sac tumor was diagnosed by pathological examination. Chemotherapy was started with combined PEB protocol (Cisplatinum 100mg/m2, Etoposide 120 mg/m2, Bleomycin 15 mg/m2). Two wk after the first course, after she had already received only total of 9 mg bleomycin, 60 mg cisplatinum and 240 mg etoposide,
Correspondence and Reprint requests : Dr. Murat Çakir MD, Kazim Dirik Mah. Süvari Cad. No: 55, Mutlubaslar Apt Daire: 9, Bornova, , . Izmir/Turkiye; Phone and Fax : +90 462 377 54 73
Indian Journal of Pediatrics, Volume 74—May, 2007
Fig. 1. Note the Hiperpigmentation after the First Course of BEP Protocol. It was Diffuse Scattered, Flagellate Like, Linear Streaking Suggesting Associated with Bleomycin
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Nilgun Yaris et al membranes, hair, teeth and nails may be affected, and the reaction may be diffused or localized.2 In the present case, the skin pigmentation was diffuse scattered, flagellate like, linear streaking over the neck, shoulder, chest and abdominal wall. This type of hyperpigmentation is generally associated with bleomycin. 3-5 Cutaneous hyperpigmentation for bleomycin is dose related toxicity as in pulmonary fibrosis, which is the major side effect of bleomycin.1, 2 Hyperpigmentation generally occurs after a cumulative dose of 90-285 mg; but it has been reported rarely with doses as low as 5 mg given parenterally.1, 2, 5 The interval between the administration of the drug and the onset of the hyperpigmentation ranges from one wk to 6 mth and it is generally reversible. 1, 6 In the present patient, hyperpigmentation was noted about two wk after receiving a single dose of 15 mg/m 2 (totally 9 mg) bleomycin. Cisplatin-induced hyperpigmentation is generally occurred after intra-arterial treatment over the dorsal surface of the extremities and at sites of the trauma.2 It appears to be permanent. For the present case, cisplatin might have played an additional role for hyperpigmentation. The mechanisms responsible for chemotherapyinduced hyperpigmentation have not been clearly identified. Melanocyte accumulation, postinflamatory changes and direct toxicity of the chemotherapeutic agents are suggested causes.1, 2 It has also been purposed that the lack of hydrolyse, which inactivates the bleomycin in the lung and skin is responsible for
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mucocutaneous toxicity.7 The present case was interesting because, the dosages used were very low, time between the administration of the drug and the onset of the hyperpigmentation was very short, and the hyperpigmentation has persisted for more than 24 mth. Oncologists need to consider chemotherapeutic agents induced hyperpigmentation in order to enlighten parents, release their anxiety, and to avoid some unnecessary investigations.
REFERENCES 1. Haskell CM. Antineoplastic agents. In Haskell CM, eds. Cancer Treatment. 4th ed. Sounders WB Co/Med, 1995; 166. 2. Susser WS, Whitaker-Worrth DL, Grant-Kels JM. Mucocutaneous reactions to chemotherapy. J Am Acad Dermatol 1999; 40 : 367-398. 3. Mutafoglu-Uysal K, Sarialioglu F, Olgun N. Bleomycin induced hyperpigmentation and hypersensitivity reactions to etoposide and vinblastine in a child with endodermal sinus tumor. Turk J Pediatr 2001; 43 : 172-174. 4. Dubra P, Ilchyshyn A, Das R. Bleomycin-induced flagellate erythema. Clin Exp Dermatol 1991; 16 : 216-217. 5. Schuler G, Aubock J, Huber H. Bleomycin-induced linear hyperpigmentation. Hautarzt 1984; 35 : 383-386. 6. Vonhilsheimer GE, Norton SA. Delayed bleomycin-induced hyperpigmentation and pressure on the skin. J Am Acad Dermatol 2002; 46: 642-643. 7. Lindae ML, Hu CH, Nickoloff BJ. Pruritic erythematous linear plaques on the neck and back. “Flagellate“ erythema secondary to bleomycin therapy. Arch Dermatol 1987; 123: 395 398.
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