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有症状的门克斯病的铜替代治疗:伦理考量。

Copper-replacement treatment for symptomatic Menkes disease: ethical considerations.

作者信息

Sheela S R, Latha M, Liu P, Lem K, Kaler S G

机构信息

Indira Gandhi Co-operative Hospital, Kadavanthra, Cochin, Kerala, India.

出版信息

Clin Genet. 2005 Sep;68(3):278-83. doi: 10.1111/j.1399-0004.2005.00496.x.

Abstract

We describe a child with classical Menkes disease with a novel ATP7A mutation, intractable seizures, severe hypotonia and developmental delay, hypopigmentation of the skin and hair, and failure to thrive, who was treated with daily subcutaneous copper histidine injections for 2(1/2) years, beginning at 15 months of age. He became seizure-free and pigmentation of his skin and hair darkened, but he continued to have severe developmental delays. His condition remains stable 8 months after stopping treatment. We review the ethical aspects of offering copper treatment for Menkes disease infants diagnosed after neurological symptoms become manifest. These include (1) the prospect for any benefits, (2) the potential risks and discomforts, (3) the parents' wishes with respect to treatment, (4) the family's understanding of the treatment's potential futility, (5) the family's understanding of the investigational nature of this treatment, (6) the potential for treatment to have an adverse impact on unaffected family members, (7) whether the ultimate decision regarding treatment should rest with health care providers or with the patient's parents, and (8) the duration of treatment. The ethical issues encountered in providing possibly futile treatment in this difficult disorder seem relevant to other pediatric medical conditions as well.

摘要

我们描述了一名患有经典型门克斯病的儿童,其携带一种新的ATP7A突变,有难治性癫痫、严重肌张力低下和发育迟缓、皮肤和头发色素减退以及生长发育不良,从15个月大开始接受为期2年半的每日皮下注射组氨酸铜治疗。他不再发作癫痫,皮肤和头发色素沉着加深,但仍有严重的发育迟缓。停止治疗8个月后,他的病情保持稳定。我们回顾了在神经系统症状出现后诊断出的门克斯病婴儿提供铜治疗的伦理问题。这些问题包括:(1)任何获益的可能性;(2)潜在风险和不适;(3)父母对治疗的意愿;(4)家庭对治疗可能无效的理解;(5)家庭对该治疗试验性质的理解;(6)治疗对未受影响家庭成员产生不利影响的可能性;(7)关于治疗的最终决定应由医疗保健提供者还是患者父母做出;(8)治疗持续时间。在这种疑难病症中提供可能无效的治疗时遇到的伦理问题似乎也与其他儿科疾病相关。

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