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一名毛发色素减退儿童的反复住院:免疫缺陷病初现端倪。

Recurrent Hospitalisation in a Child With Hypopigmented Hair: Inborn Errors of Immunity Emerge.

作者信息

Kundavaram Rajkumar, Kadiri Harish, Khurana Ujjawal, Chaudhary Narendra K, Dhingra Bhavna, Bargir Umair, Malik Shikha

机构信息

Pediatrics, All India Institute of Medical Sciences, Bhopal, IND.

Pathology and Laboratory Medicine, All India Institute of Medical Sciences, Bhopal, IND.

出版信息

Cureus. 2024 Dec 29;16(12):e76554. doi: 10.7759/cureus.76554. eCollection 2024 Dec.

Abstract

Griscelli syndrome is a rare autosomal recessive disorder characterised by pigmentary dilution of skin and hair, recurrent skin and pulmonary infections, neurological manifestations, and immunodeficiency. We present a four-month-old female child with hypopigmented silvery hair and a history of recurrent hospitalisations for respiratory illness. The child was extensively evaluated for inborn errors of immunity (IEI), and the final diagnosis of type 2 Griscelli syndrome was made only after genetic testing. Antibiotic and antifungal prophylaxis was initiated, and the child is currently in good health. The family was counselled regarding haematopoietic stem cell transplant (HSCT) as the only curative option and the need for prenatal testing in further pregnancies. This case emphasises the need for a high index of clinical suspicion in diagnosing IEI and also highlights the limited therapeutic options and the role of genetic counselling.

摘要

格里塞利综合征是一种罕见的常染色体隐性疾病,其特征为皮肤和毛发色素稀释、反复的皮肤和肺部感染、神经学表现以及免疫缺陷。我们报告一名4个月大的女童,其毛发呈色素减退的银色,并有因呼吸道疾病反复住院的病史。对该儿童进行了广泛的先天性免疫缺陷(IEI)评估,最终仅在基因检测后才确诊为2型格里塞利综合征。开始了抗生素和抗真菌预防治疗,该儿童目前健康状况良好。已就造血干细胞移植(HSCT)作为唯一的治愈选择以及进一步妊娠时进行产前检测的必要性向其家人提供了咨询。该病例强调了在诊断IEI时需要高度的临床怀疑指数,同时也突出了有限的治疗选择以及遗传咨询的作用。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/e737/11774631/73b3ccf84261/cureus-0016-00000076554-i01.jpg

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本文引用的文献

1
Diagnostic and therapeutic caveats in Griscelli syndrome.格雷塞利综合征的诊断和治疗注意事项。
Scand J Immunol. 2021 Jun;93(6):e13034. doi: 10.1111/sji.13034. Epub 2021 Mar 20.
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Pediatric CNS-isolated hemophagocytic lymphohistiocytosis.儿童中枢神经系统孤立性噬血细胞性淋巴组织细胞增生症。
Neurol Neuroimmunol Neuroinflamm. 2019 Apr 8;6(3):e560. doi: 10.1212/NXI.0000000000000560. eCollection 2019 May.
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Seizure as the presenting manifestation in Griscelli syndrome type 2.癫痫发作作为2型格里塞利综合征的首发表现。
Pediatr Neurol. 2015 May;52(5):535-8. doi: 10.1016/j.pediatrneurol.2015.01.010. Epub 2015 Jan 26.
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Cerebellar involvement of Griscelli syndrome type 2.2型格里塞利综合征的小脑受累情况。
BMJ Case Rep. 2014 Oct 14;2014:bcr2014206703. doi: 10.1136/bcr-2014-206703.
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Griscelli syndrome.格里塞利综合征
J Am Acad Dermatol. 2006 Aug;55(2):337-40. doi: 10.1016/j.jaad.2005.11.1056.

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