Shumer Daniel E, Thaker Vidhu, Taylor George A, Wassner Ari J
Division of Endocrinology, Boston Children's Hospital, Boston, Massachusetts, USA.
Department of Radiology, Boston Children's Hospital, Boston, Massachusetts, USA.
Arch Dis Child Fetal Neonatal Ed. 2014 Sep;99(5):F419-21. doi: 10.1136/archdischild-2014-306069. Epub 2014 Jun 6.
Subcutaneous fat necrosis (SCFN) is a rare form of panniculitis in infants that generally occurs following birth trauma, meconium aspiration, or therapeutic cooling. Severe hypercalcaemia occurs in a subset of patients, but data on its presentation, management and outcomes are limited. This report details the clinical course and complications of infants treated for severe hypercalcaemia (peak serum calcium ≥3.0 mmol/L) due to SCFN.
Chart review of all infants with SCFN seen at a single paediatric centre over a 13-year period.
Seven infants with SCFN developed severe hypercalcaemia, with median peak serum calcium 4.1 mmol/L (range 3.3-5.1).
Severe hypercalcaemia occurred before 6 weeks of age, and was asymptomatic in 3/7 patients (43%). Most patients were treated with intravenous hydration, furosemide, glucocorticoids and low-calcium formula, which restored normocalcaemia in a median of 9 days (range 2-42). Fever developed during treatment in 4/7 infants (57%): two patients had bacterial infections and two had no infectious source identified. Nephrocalcinosis was present in 5/6 patients (83%) who were evaluated by renal ultrasound. Nephrocalcinosis failed to resolve in all cases over a median follow-up of 20 months (range 8-48), but no renal dysfunction was observed. Eosinophilia, which has not been reported previously in SCFN, was present in 6/7 patients (86%).
In this largest series to date of infants with severe hypercalcaemia due to SCFN, novel findings include the common occurrence of fever and a high incidence of persistent nephrocalcinosis without evidence of adverse renal outcomes.
皮下脂肪坏死（SCFN）是婴儿脂膜炎的一种罕见形式，通常发生在出生创伤、胎粪吸入或治疗性降温之后。一部分患者会出现严重高钙血症，但其临床表现、治疗及预后的数据有限。本报告详细介绍了因SCFN接受治疗的严重高钙血症（血清钙峰值≥3.0 mmol/L）婴儿的临床病程及并发症。
对一家儿科中心13年间诊治的所有SCFN婴儿进行病历回顾。
7例SCFN婴儿出现严重高钙血症，血清钙峰值中位数为4.1 mmol/L（范围3.3 - 5.1）。
严重高钙血症出现在6周龄之前，3/7例患者（43%）无症状。多数患者接受了静脉补液、呋塞米、糖皮质激素及低钙配方奶治疗，血钙恢复正常的中位数为9天（范围2 - 42天）。4/7例婴儿（57%）在治疗期间出现发热：2例患者发生细菌感染，2例未发现感染源。6例接受肾脏超声检查的患者中有5例（83%）存在肾钙质沉着症。在中位随访20个月（范围8 - 48个月）期间，所有病例的肾钙质沉着症均未消退，但未观察到肾功能障碍。嗜酸性粒细胞增多症在6/7例患者（86%）中出现，此前SCFN中未见相关报道。
在迄今为止关于因SCFN导致严重高钙血症婴儿的最大系列研究中，新发现包括发热常见以及持续性肾钙质沉着症发生率高，但无不良肾脏预后证据。
