Hung Shuo-Hsun, Tsai Wen-Yu, Tsao Po-Nien, Chou Hung-Chieh, Hsieh Wu-Shiun
Department of Pediatrics, National Taiwan University Hospital, College of Medicine, National Taiwan University, Taipei, Taiwan.
J Formos Med Assoc. 2003 Nov;102(11):801-4.
Hypercalcemia is occasionally found in newborns with subcutaneous fat necrosis and carries potential life-threatening risk. Bisphosphonates have been recently introduced in the treatment of subcutaneous fat necrosis in newborns. We report a case of extensive subcutaneous fat necrosis in a female infant complicated with intractable hypercalcemia. Standard treatment for hypercalcemia was given, including saline hydration, a low calcium diet, furosemide, and glucocorticoid, but without response. Serum 1,25-dihydroxyvitamin D level was elevated at 126 pg/mL, 25-hydroxyvitamin D level was normal, and intact parathyroid hormone was suppressed at < 1 pg/mL. Oral clodronate disodium, a second-generation bisphosphonate, was administered, and resulted in the normalization of serum calcium, urine N-telopeptide, urine calcium/creatinine ratio, and serum intact parathyroid hormone level. This case suggests that oral clodronate may be an effective treatment for subcutaneous fat necrosis with hypercalcemia in newborns.
高钙血症偶尔会在患有皮下脂肪坏死的新生儿中发现，并具有潜在的危及生命的风险。双膦酸盐类药物最近已被用于治疗新生儿皮下脂肪坏死。我们报告一例女婴广泛皮下脂肪坏死并发难治性高钙血症的病例。给予了高钙血症的标准治疗，包括生理盐水水化、低钙饮食、呋塞米和糖皮质激素，但无反应。血清1,25-二羟维生素D水平升高至126 pg/mL，25-羟维生素D水平正常，完整甲状旁腺激素被抑制至<1 pg/mL。给予第二代双膦酸盐口服氯膦酸二钠，结果血清钙、尿N-端肽、尿钙/肌酐比值和血清完整甲状旁腺激素水平恢复正常。该病例表明，口服氯膦酸可能是治疗新生儿皮下脂肪坏死伴高钙血症的有效方法。
