新生儿皮下脂肪坏死:病例报告和病理生理学模型的系统文献综述。

IF 2.4 Q1 PEDIATRICS Molecular and cellular pediatrics Pub Date : 2022-11-24 DOI:10.1186/s40348-022-00151-1
Leonie Frank, Stephanie Brandt, Martin Wabitsch
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引用次数: 3

摘要

背景:新生儿皮下脂肪坏死(SCFN)是一种发生在生命最初几天的罕见疾病。婴儿的特征是皮下组织有硬结节,颜色为紫色或红斑,出现在上背部、脸颊、臀部和四肢。在大多数病例中,SCFN是一种自限性疾病,结节会在6个月内消失。与SCFN相关的严重并发症是高钙血症。引起SCFN或相关高钙血症的病理生理机制尚不完全清楚。方法:系统的文献研究,包括六个最大的医学研究数据库,识别所有已发表的SCFN病例报告。已确定N = 206份出版物,其中包含N = 320例病例报告。所有病例被分为四个亚组(取决于报告的血钙水平):高钙血症、正常钙血症、低钙血症或无资料。已报道的母体因素、出生特征、SCFN的细节、诊断、治疗和长期观察均摘自出版物。结果:这是第一个系统的文献研究,总结了1948 - 2018年所有已发表的SCFN病例。64.3%的病例提供了血钙水平信息。其中,大多数表现为高钙血症(70.5%)(正常钙血症25.1%,低钙血症4.3%)。89.3%的新生儿高钙血症表现出甲状旁激素水平的抑制。妊娠期孕妇糖尿病、妊娠期孕妇高血压疾病、巨大儿(> 4000g)、窒息和治疗性低温是SCFN的危险因素。组织学结果显示98%的病例为肉芽肿性炎症。结论:我们发现母亲、出生特征和治疗措施可能是SCFN的危险因素。在新生儿护理中应考虑到这些风险因素。
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Subcutaneous fat necrosis in newborns: a systematic literature review of case reports and model of pathophysiology.

Background: Subcutaneous fat necrosis of the newborn (SCFN) is a rare disease occurring in the first days of life. Characteristically, the infants show hard nodules in subcutaneous tissue, purple or erythematous in color and appear on the upper back, cheeks, buttocks and limbs. In most cases, SCFN is a self-limiting disease, as the nodules disappear in up to 6 months. A severe complication associated with SCFN is hypercalcaemia. Pathophysiological mechanisms causing SCFN or associated hypercalcaemia are not fully understood yet.

Methods: A systematic literature research including the six biggest databases for medical research has been used to identify all published case reports of SCFN. N = 206 publications has been identified containing n = 320 case reports. All cases have been classified into four subgroups (depending on reported serum-calcium-level): hypercalcaemia, normocalcaemia, hypocalcaemia or no information given. Reported maternal factors, birth characteristics, details about SCFN, diagnostics, therapy and long-term observations have been extracted from publications.

Results: This is the first systematic literature research that summed up all published cases of SCFN from 1948 up to 2018. Information about serum calcium level was given in 64.3% of the cases. From those, the majority showed hypercalcaemia (70.5%) (normocalcaemia 25.1%, hypocalcemia 4.3%). 89.3% of newborns with hypercalcaemia showed suppressed levels of the parathormone. Maternal gestational diabetes, maternal hypertensive diseases during pregnancy, macrosomia (> 4000g), asphyxia and therapeutic hypothermia are risk factors for SCFN. Histological findings showed a granulomatous inflammation in 98% of cases.

Conclusion: We identified that maternal, birth characteristics and therapeutic measures are probably risk factors for SCFN. These risk factors should be taken into account within the care of neonates.

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