Chesover Alexander D, Harrington Jennifer, Mahmud Farid H
Division of Endocrinology, The Hospital for Sick Children, University of Toronto, Toronto, Ontario.
Paediatr Child Health. 2019 Dec 24;26(1):e52-e56. doi: 10.1093/pch/pxz141. eCollection 2021 Feb.
Subcutaneous fat necrosis (SCFN) can be complicated by severe hypercalcemia, which is frequently asymptomatic. Nephrocalcinosis is associated with hypercalcemia and, in other clinical settings, has been linked to furosemide and glucocorticoid use. First-line bisphosphonate therapy treating hypercalcemia in neonatal SCFN is not well described.
To describe the biochemical changes and risk of nephrocalcinosis in infants with hypercalcemia, secondary to neonatal SCFN, treated with initial pamidronate.
A retrospective chart review of five infants treated with initial pamidronate and without furosemide or glucocorticoids. Data were collected on the following: timing of presentation, therapeutic response, and presence of nephrocalcinosis.
Hypercalcemia resolved after 2.8±1.7 days; this is compared to 7.6±2.8 days from previously reported cases utilising alternative therapies (P=0.012). There were no episodes of rebound hypercalcemia or hypocalcemia. Nephrocalcinosis was present in four of five cases. When including published cases, age at diagnosis was associated with presenting serum calcium (P=0.003) and nephrocalcinosis was associated with higher serum calcium (P=0.014) and time from SCFN to hypercalcemia diagnosis (P=0.002).
This retrospective case series demonstrates that first-line pamidronate treatment was effective and safe in the resolution of hypercalcemia. Nephrocalcinosis was observed, despite the avoidance of furosemide and glucocorticoid therapy, and associated with greater disease severity and duration of hypercalcemia.
皮下脂肪坏死(SCFN)可并发严重高钙血症,且通常无症状。肾钙质沉着症与高钙血症相关,在其他临床情况下,与使用呋塞米和糖皮质激素有关。新生儿SCFN中治疗高钙血症的一线双膦酸盐疗法尚无详细描述。
描述接受初始帕米膦酸治疗的新生儿SCFN继发高钙血症婴儿的生化变化及肾钙质沉着症风险。
对5例接受初始帕米膦酸治疗且未使用呋塞米或糖皮质激素的婴儿进行回顾性病历审查。收集以下数据:发病时间、治疗反应及肾钙质沉着症情况。
高钙血症在2.8±1.7天后缓解;相比之下,先前报道的采用其他疗法的病例为7.6±2.8天(P = 0.012)。无高钙血症反弹或低钙血症发作。5例中有4例存在肾钙质沉着症。纳入已发表病例后,诊断时年龄与就诊时血清钙相关(P = 0.003),肾钙质沉着症与较高血清钙(P = 0.014)及从SCFN至高钙血症诊断的时间相关(P = 0.002)。
该回顾性病例系列表明,一线帕米膦酸治疗在解决高钙血症方面有效且安全。尽管避免了呋塞米和糖皮质激素治疗,但仍观察到肾钙质沉着症,且与疾病严重程度增加和高钙血症持续时间相关。