日本新生児成育医学会雑誌　第32巻　第2号　100 ～ 103頁（2020年）

日本新生児成育医学会雑誌　32(2)：338-341；2020　

受付日：2019．09．24

受理日：2019．12．16

皮下型乳児血管腫が疑われた病変に対してプロプラノロールが有効であった新生児慢性肺疾患児の1 例

Propranolol-Effective Infantile Hemangioma in an Infant with Chronic Lung Disease

近畿大学　医学部　小児科学教室

Kindai University Pediatrics department

田中　藍・小西悠平・南方俊祐・草野信義・和田紀久・杉本圭相

Ai TANAKA，Yuhei KONISHI，Shunsuke MINAKATA，Nobuyoshi KUSANO， Norihisa WADA，Keisuke SUGIMOTO

Key Words：infant hemangioma，propranolol，chronic lung disease in the newborn， hypoxic pulmonary vasoconstriction，very low birth weight

　近年，本邦で乳児血管腫に対してプロプラノロール塩酸塩シロップ製剤の使用が承認された。しかし，新生児慢性肺疾患（Chronic Lung Disease，CLD）ではプロプラノロールの肺血管収縮作用が懸念され，投与報告はほとんどない。今回，プロプラノロール投与により治療し得た乳児血管腫を伴うCLD 児例を経験した。症例は生後3 か月の女児。経膣分娩にて在胎26 週4 日，734g で出生し，CLDⅢ型と診断した。右頬部の腫瘤に対し，画像検査から動静脈奇形または皮下型乳児血管腫と考え，プロプラノロールを1mg/kg/day で投与開始した。低酸素性肺血管収縮の増強を疑わせる所見を認めず，腫瘤は退縮傾向となり，皮下型乳児血管腫と考えた。現在，プロプラノロールの内服を継続しており，開始後7 か月で外表上明らかでなくなった。皮下型乳児血管腫の治療には，プロプラノロールが有効であり，CLD の早産児に対しても，呼吸循環状態に影響することなく安全に投与できると思われた。

Laser therapy has been routinely employed for the treatment of infantile hemangioma. The use of propranolol hydrochloride for the treatment of infantile hemangioma was recently approved in Japan；however, its efficacy in children with chronic lung disease is not well characterized. Here we report the case of an infant with chronic lung disease who presented with subcutaneous infantile hemangioma and was successfully treated with oral propranolol therapy.
The patient was a 3-month-old infant. She was born in transvaginal labor at the gestational age of 26 weeks and 4 days （birth weight：734g）. She experienced persistent breathing difficulties, and a diagnosis of chronic lung disease type Ⅲ was established. Fifty days after birth, a soft elastic tumor with a smooth surface was found on the right cheek；subsequently, the tumor showed enlargement. Ultrasonography and contrast-enhanced computed tomography enabled identification of the tumor as an arteriovenous malformation or subcutaneous infantile angioma. Owing to progressive increase in tumor size, she was prescribed propranolol syrup 1mg/kg. In the absence of any respiratory adverse effects or signs of enhanced hypoxic pulmonary vasoconstriction, the dose was increased to 3mg/kg. Subsequently, a definitive diagnosis of subcutaneous infantile hemangioma was established as the mass exhibited a clear tendency to retract. The infant was discharged 143 days after birth with ambulatory oxygen supply. The tumor showed further regression after 7 months, suggesting that propranolol therapy is effective for the treatment of subcutaneous infantile hemangioma. Propranolol may be safe for the treatment of premature infants with severe chronic lung disease.