Luna Cecilia Inés, Fernández Cordero Marisa, Escruela Romina, Sierra Valeria, Córdoba Antonela, Goñi Ignacio María, Berridi Ricardo
Servicio de Clínica Pediátrica, Hospital Zonal Especializado Dr. Noel H. Sbarra.
Unidad de Residencia de Clínica Pediátrica, Hospital Zonal Especializado Dr. Noel H. Sbarra, La Plata, Argentina.
Arch Argent Pediatr. 2014 Oct;112(5):e196-9. doi: 10.5546/aap.2014.e196.
The objective of this study is to describe the unexpected association between the congenital generalized lipodystrophy (CGL) and Dandy Walker anomaly. We report the case of a 1-year-old infant who was hospitalized at her fourth month of life with Dandy Walker anomaly diagnosis and an increased social risk. During her hospitalization, she developed progressively: acromegaloid aspect, triangular fascia, hirsutism, lipoatrophy, muscle hypertrophy, clitoromegaly, abdominal distention, progressive hepatomegaly, and hypertriglyceridemia. This led to the clinical diagnosis of congenital generalized lipodystrophy. Importance should be given to the examination of clinical aspects as well as the interdisciplinary follow-up for proper detection of insulin resistance and diabetes, early puberty, cardiomyopathy, among others. In case of Dandy Walker anomaly, it should be checked the evolution to search intracranial hypertension signs. Due to its autosomal recessive nature, it is important to provide genetic counseling to the parents.
本研究的目的是描述先天性全身性脂肪营养不良(CGL)与丹迪·沃克畸形之间意外的关联。我们报告了一名1岁婴儿的病例,该婴儿在出生后第四个月因丹迪·沃克畸形诊断和社会风险增加而住院。在住院期间,她逐渐出现:肢端肥大样面容、三角筋膜、多毛症、脂肪萎缩、肌肉肥大、阴蒂肥大、腹胀、进行性肝肿大和高甘油三酯血症。这导致了先天性全身性脂肪营养不良的临床诊断。应重视临床方面的检查以及跨学科随访,以正确检测胰岛素抵抗和糖尿病、性早熟、心肌病等。对于丹迪·沃克畸形,应检查其进展情况以寻找颅内高压体征。由于其常染色体隐性遗传性质,为父母提供遗传咨询很重要。