Odaman Al Isik, Oymak Yesim, Hazan Filiz, Gursoy Semra, Ozturk Tulay, Bag Ozlem, Gozmen Salih, Karakaya Nurgul, Karapinar Tuba Hilkay
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.
Sisli Etfal Hastan Tip Bul. 2022 Mar 28;56(1):161-165. doi: 10.14744/SEMB.2021.88964. eCollection 2022.
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.
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。