Mîndru Dana Elena, Țarcă Elena, Braha Elena Emanuela, Curpăn Alexandrina-Ștefania, Roșu Solange Tamara, Anton-Păduraru Dana-Teodora, Adumitrăchioaiei Heidrun, Bernic Valentin, Pădureț Ioana-Alexandra, Luca Alina Costina
Department of Mother and Child Medicine, University of Medicine and Pharmacy "Gr. T. Popa", 700115 Iasi, Romania.
Department of Surgery II-Pediatric Surgery, University of Medicine and Pharmacy "Gr. T. Popa", 700115 Iasi, Romania.
Diagnostics (Basel). 2024 Feb 24;14(5):491. doi: 10.3390/diagnostics14050491.
Gangliosidosis (ORPHA: 79255) is an autosomal recessive lysosomal storage disease (LSD) with a variable phenotype and an incidence of 1:200000 live births. The underlying genotype is comprised GLB1 mutations that lead to β-galactosidase deficiency and subsequently to the accumulation of monosialotetrahexosylganglioside (GM1) in the brain and other organs. In total, two diseases have been linked to this gene mutation: Morquio type B and Gangliosidosis. The most frequent clinical manifestations include dysmorphic facial features, nervous and skeletal systems abnormalities, hepatosplenomegaly, and cardiomyopathies. The correct diagnosis of GM1 is a challenge due to the overlapping clinical manifestation between this disease and others, especially in infants. Therefore, in the current study we present the case of a 3-month-old male infant, admitted with signs and symptoms of respiratory distress alongside rapid progressive heart failure, with minimal neurologic and skeletal abnormalities, but with cardiovascular structural malformations. The atypical clinical presentation raised great difficulties for our diagnostic team. Unfortunately, the diagnostic of GM1 was made postmortem based on the DBS test and we were able to correlate the genotype with the unusual phenotypic findings.
神经节苷脂沉积症(孤儿病编号:79255)是一种常染色体隐性溶酶体贮积病(LSD),其表型多样,发病率为1:200000活产儿。潜在基因型由GLB1突变组成,该突变导致β-半乳糖苷酶缺乏,进而导致单唾液酸四己糖神经节苷脂(GM1)在大脑和其他器官中蓄积。总共有两种疾病与这种基因突变有关:B型黏多糖贮积症和神经节苷脂沉积症。最常见的临床表现包括面部畸形、神经和骨骼系统异常、肝脾肿大以及心肌病。由于这种疾病与其他疾病的临床表现重叠,尤其是在婴儿中,GM1的正确诊断具有挑战性。因此,在本研究中,我们报告了一例3个月大的男婴病例,该患儿因呼吸窘迫体征和症状入院,同时伴有快速进展的心力衰竭,神经和骨骼异常轻微,但存在心血管结构畸形。这种非典型的临床表现给我们的诊断团队带来了很大困难。不幸的是,GM1的诊断是在患儿死后通过干血斑检测做出的,我们能够将基因型与不寻常的表型发现相关联。