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一名叙利亚难民父母的孩子在美国被诊断出患有LHX3缺乏症,伴有严重发育迟缓。

LHX3 deficiency presenting in the United States with severe developmental delay in a child of Syrian refugee parents.

作者信息

Ahern Susan, Daniels Mark, Bhangoo Amrit

机构信息

Division of Endocrinology, UCLA School of Medicine, Ventura, California, USA.

Division of Pediatric Endocrinology, Children's Hospital of Orange County, Orange, California, USA.

出版信息

Endocrinol Diabetes Metab Case Rep. 2018;2018. doi: 10.1530/EDM-18-0079. Epub 2018 Nov 22.

Abstract

In this case report, we present a novel mutation in Lim-homeodomain (LIM-HD) transcription factor, LHX3, manifesting as combined pituitary hormone deficiency (CPHD). This female patient was originally diagnosed in Egypt during infancy with Diamond Blackfan Anemia (DBA) requiring several blood transfusions. Around 10 months of age, she was diagnosed and treated for central hypothyroidism. It was not until she came to the United States around two-and-a-half years of age that she was diagnosed and treated for growth hormone deficiency. Her response to growth hormone replacement on linear growth and muscle tone were impressive. She still suffers from severe global development delay likely due to delay in treatment of congenital central hypothyroidism followed by poor access to reliable thyroid medications. Her diagnosis of DBA was not confirmed after genetic testing in the United States and her hemoglobin normalized with hormone replacement therapies. We will review the patient's clinical course as well as a review of LHX3 mutations and the associated phenotype. Learning points: Describe an unusual presentation of undertreated pituitary hormone deficiencies in early life Combined pituitary hormone deficiency due to a novel mutation in pituitary transcription factor, LHX3 Describe the clinical phenotype of combined pituitary hormone deficiency due to LHX3 mutations.

摘要

在本病例报告中,我们展示了一种新型的Lhx3(一种含Lim同源结构域的转录因子)突变,其表现为联合垂体激素缺乏症(CPHD)。该女性患者婴儿期在埃及最初被诊断为先天性纯红细胞再生障碍性贫血(DBA),需要多次输血。约10个月大时,她被诊断出中枢性甲状腺功能减退并接受治疗。直到她两岁半左右来到美国,才被诊断出生长激素缺乏并接受治疗。她对生长激素替代治疗在身高增长和肌肉张力方面的反应令人印象深刻。她仍患有严重的全面发育迟缓,可能是由于先天性中枢性甲状腺功能减退治疗延迟,随后又难以获得可靠的甲状腺药物。在美国进行基因检测后,她的DBA诊断未得到证实,其血红蛋白通过激素替代疗法恢复正常。我们将回顾患者的临床病程,以及对Lhx3突变及其相关表型的综述。学习要点:描述早期未得到充分治疗的垂体激素缺乏症的不寻常表现垂体转录因子Lhx3的新型突变导致的联合垂体激素缺乏症描述Lhx3突变所致联合垂体激素缺乏症的临床表型

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/63d7/6280131/bbfa69f0e0e8/EDM18-0079fig1.jpg

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