Pamidronate as treatment of severe hypercalcemia in SCFN of the newborn: A case report

Raiwathy Krishnasamy, A. Wan, Nalini M Selveindran
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Abstract

Subcutaneous fat necrosis of newborn (SCFN) is an uncommon entity that occurs in neonates who experienced perinatal stress. We report use of pamidronate, to control persistent hypercalcemia in a 5- week-old infant with SCFN not responding to initial treatment. A term male neonate was born by emergency LSCS due to non-reassuring fetal status. Antenatally mother had gestational diabetes mellitus, group B streptococcus carrier and an antenatal scan at 29 weeks detected a fetus with dilated small bowel. Baby was born vigorous but complicated with bowel perforation requiring fluid resuscitation and a bedside glove drain. He underwent laparotomy for small bowel perforation secondary to ileal atresia and required TPN postoperatively. At 1 month of life, he had palpable purplish lumps at his trunk and limbs associated with severe hypercalcemia supporting the diagnosis of subcutaneous fat necrosis. Despite receiving initial treatment of hyperhydration and frusemide for two weeks, the patient's hypercalcemia peaked to 4.11mmol/L. His renal ultrasound showed nephrocalcinosis with renal and bladder calculi. He was given IV Pamidronate. Post single dose of IV Pamidronate, calcium levels reduced ranging 2.2-3 mmol/L and frusemide was discontinued. On discharge he was maintained on low calcium milk. During his first follow up the calcium level remained stable and repeated ultrasound showed resolution of the renal pelvis and bladder calculi with persistence of the medullary nephrocalcinosis. SCFN has a potentially life-threatening complication due to development of severe hypercalcemia. For patients with severe hypercalcemia not responding to hydration, and low calcium intake, pamidronate is an effective and safe treatment.  Good supportive management is very important and should be done prior to pamidronate.  
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帕米膦酸钠治疗新生儿SCFN严重高钙血症1例
新生儿皮下脂肪坏死(SCFN)是一种罕见的实体,发生在新生儿谁经历围产期压力。我们报告使用帕米膦酸盐,以控制持续高钙血症的5周大婴儿SCFN不响应初始治疗。1例足月男婴因胎儿状态不稳定而通过紧急LSCS出生。产前母亲患有妊娠糖尿病,B组链球菌携带者,29周时产前扫描发现胎儿小肠扩张。婴儿出生时精力充沛,但并发肠穿孔,需要液体复苏和床边手套引流。因回肠闭锁致小肠穿孔行开腹手术,术后行TPN。1个月大时,他的躯干和四肢出现可触及的紫色肿块,伴有严重的高钙血症,支持皮下脂肪坏死的诊断。尽管患者最初接受了高水合和氟塞米两周的治疗,但患者的高钙血症最高达到4.11mmol/L。肾脏超声显示肾钙质沉着症伴肾及膀胱结石。给予静脉注射帕米膦酸钠。单剂量静脉注射帕米膦酸钠后,钙水平降低2.2-3 mmol/L,停用氟塞米。出院时,他继续喝低钙牛奶。在他的第一次随访中,钙水平保持稳定,反复超声显示肾盂和膀胱结石消退,但肾髓质钙沉着症持续存在。由于严重高钙血症的发展,SCFN具有潜在的危及生命的并发症。对于严重的高钙血症,对水合无反应,钙摄入量低的患者,帕米膦酸盐是一种有效且安全的治疗方法。良好的支持性管理非常重要,应在帕米膦酸钠之前进行。
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