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一例以血小板减少和肌张力减退为表现的婴儿恶性骨硬化症新生儿病例:氯离子电压门控通道7基因的新突变

A Neonatal Case of Infantile Malignant Osteopetrosis Presenting with Thrombocytopenia and Hypotonicity: A Novel Mutation in Chloride Voltage-Gated Channel 7 Gene.

作者信息

Odaman Al Isik, Oymak Yesim, Hazan Filiz, Gursoy Semra, Ozturk Tulay, Bag Ozlem, Gozmen Salih, Karakaya Nurgul, Karapinar Tuba Hilkay

机构信息

Department of Pediatric Hematology and Oncology, Dr. Behçet Uz Child Disease and Pediatric Surgery Training and Research Hospital, Izmir, Turkey.

Department of Pediatric Medical Genetics, Dr. Behçet Uz Child Disease and Pediatric Surgery Training and Research Hospital, Izmir, Turkey.

出版信息

Sisli Etfal Hastan Tip Bul. 2022 Mar 28;56(1):161-165. doi: 10.14744/SEMB.2021.88964. eCollection 2022.

DOI:10.14744/SEMB.2021.88964
PMID:35515972
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC9040309/
Abstract

Autosomal recessive osteopetrosis is also known as infantile malignant osteopetrosis (IMO). The clinical course is often serious and if left untreated, it is fatal in the 1st year of life. Diagnosis is challenging and often delayed or misdiagnosed. Herein, we present an infant girl who was diagnosed with IMO during evaluations for her hypotonicity and thrombocytopenia. A novel mutation of the chloride voltage-gated channel 7 (CLCN7) gene was also reported. A 10-day-old female patient was referred to our hospital for evaluation of hypotonicity. Her physical examination was normal, other than hypotonicity. Laboratory analysis revealed thrombocytopenia and hypocalcemia. In the progress, while she was followed in outpatient clinic, hepatosplenomegaly was detected at the age of 3 months. IMO was suspected with the findings of hepatosplenomegaly, cytopenia, hypocalcemia, difficulty of obtaining bone marrow, peripheral smear findings, and hearing loss. The X-ray of the bones was consistent with IMO. A novel pathogenic homozygous c.1504>T (p.Arg502Trp) mutation in CLCN7 gene was revealed. IMO is a rare disorder and it is important to differentiate this entity for better clinical outcome. The presence of neurological and hematological findings, organomegaly, hearing loss, and vision disorders must attract attention to IMO.

摘要

常染色体隐性遗传性骨硬化症也被称为婴儿恶性骨硬化症(IMO)。其临床病程通常较为严重,若不进行治疗,在出生后第一年往往会致命。诊断具有挑战性,常常延迟或误诊。在此,我们报告一名在因肌张力减退和血小板减少症接受评估期间被诊断为IMO的女婴。还报道了氯化物电压门控通道7(CLCN7)基因的一种新突变。一名10日龄的女性患者因肌张力减退被转诊至我院进行评估。除肌张力减退外,其体格检查正常。实验室分析显示血小板减少和低钙血症。在后续过程中,她在门诊接受随访时,3个月大时被检测出肝脾肿大。根据肝脾肿大、血细胞减少、低钙血症、获取骨髓困难、外周血涂片检查结果以及听力丧失等表现,怀疑为IMO。骨骼X线检查结果与IMO相符。发现CLCN7基因存在一种新的致病性纯合c.1504>T(p.Arg502Trp)突变。IMO是一种罕见疾病,为了获得更好的临床结果,区分这一疾病实体很重要。神经学和血液学表现、器官肿大、听力丧失以及视力障碍的存在必须引起对IMO的关注。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8176/9040309/434be159785a/SEMB-56-161-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8176/9040309/38c129143e7b/SEMB-56-161-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8176/9040309/434be159785a/SEMB-56-161-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8176/9040309/38c129143e7b/SEMB-56-161-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8176/9040309/434be159785a/SEMB-56-161-g002.jpg

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本文引用的文献

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Bone. 2020 Jan;130:115144. doi: 10.1016/j.bone.2019.115144. Epub 2019 Nov 6.
2
Novel CLCN7 mutations cause autosomal dominant osteopetrosis type II and intermediate autosomal recessive osteopetrosis.新型 CLCN7 突变导致常染色体显性骨硬化症 II 型和中间型常染色体隐性骨硬化症。
Mol Med Rep. 2019 Jun;19(6):5030-5038. doi: 10.3892/mmr.2019.10123. Epub 2019 Apr 3.
3
One Disease, Many Genes: Implications for the Treatment of Osteopetroses.
一种疾病,多个基因:对骨硬化症治疗的启示
Front Endocrinol (Lausanne). 2019 Feb 19;10:85. doi: 10.3389/fendo.2019.00085. eCollection 2019.
4
Novel compound heterozygous mutations in intermediate autosomal recessive osteopetrosis.中间型常染色体隐性骨硬化症中的新型复合杂合突变。
Hum Genome Var. 2017 Aug 17;4:17036. doi: 10.1038/hgv.2017.36. eCollection 2017.
5
Diagnosis and Management of Osteopetrosis: Consensus Guidelines From the Osteopetrosis Working Group.成骨不全症的诊断与管理:成骨不全症工作组的共识指南。
J Clin Endocrinol Metab. 2017 Sep 1;102(9):3111-3123. doi: 10.1210/jc.2017-01127.
6
A novel mutation and a known mutation in the CLCN7 gene associated with relatively stable infantile malignant osteopetrosis in a Chinese patient.中国一名患者中与相对稳定的婴儿恶性骨硬化症相关的CLCN7基因的一种新突变和一种已知突变。
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7
Radiologic resolution of malignant infantile osteopetrosis skeletal changes following hematopoietic stem cell transplantation.
Pediatr Blood Cancer. 2015 Sep;62(9):1645-9. doi: 10.1002/pbc.25524. Epub 2015 Mar 27.
8
Standards and guidelines for the interpretation of sequence variants: a joint consensus recommendation of the American College of Medical Genetics and Genomics and the Association for Molecular Pathology.序列变异解读的标准与指南:美国医学遗传学与基因组学学会和分子病理学协会的联合共识推荐
Genet Med. 2015 May;17(5):405-24. doi: 10.1038/gim.2015.30. Epub 2015 Mar 5.
9
Osteopetrosis: genetics, treatment and new insights into osteoclast function.骨硬化症:遗传学、治疗方法及破骨细胞功能的新见解。
Nat Rev Endocrinol. 2013 Sep;9(9):522-36. doi: 10.1038/nrendo.2013.137. Epub 2013 Jul 23.
10
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