Rare case of hyaline fibromatosis syndrome

被引:0
作者
Gupta, Ashok Kumar [1 ]
Moriangthem, Amita [2 ]
Naranje, Kirti [3 ]
Singh, Anita [3 ]
机构
[1] Dr Ram Manohar Lohia Inst Med Sci, Pediat, Lucknow, Uttar Pradesh, India
[2] Sanjay Gandhi Post Grad Inst Med Sci, Med Genet, Lucknow, Uttar Pradesh, India
[3] Sanjay Gandhi Post Grad Inst Med Sci, Neonatol, Lucknow, India
关键词
Genetics; Neonatal intensive care; Congenital disorders; INFANTILE SYSTEMIC HYALINOSIS; ULTRASTRUCTURAL FINDINGS; MUTATIONS; GENE;
D O I
10.1136/bcr-2024-260969
中图分类号
R5 [内科学];
学科分类号
1002 ; 100201 ;
摘要
Hyaline fibromatosis syndrome is a rare, progressive and fatal autosomal recessive disorder characterised by multiple subcutaneous skin nodules, osteopenia, joint contractures, failure to thrive, diarrhoea and frequent infections. There is diffuse deposition of hyaline material in the skin, gastrointestinal tract, muscle and endocrine glands. The disease is often underdiagnosed since infants affected with the disease pass away early prior to establishing a final diagnosis. We describe an infant presenting with failure to thrive, progressive severe joint contractures and skin changes. Clinical exome sequencing revealed homozygous novel missense variation in exon 3 of the anthrax toxin receptor 2 gene confirming the diagnosis.
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页数:4
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