O'Grady Michael J, Monavari Ahmad A, Cotter Melanie, Murphy Nuala P
Department of Paediatrics, Midland Regional Hospital, Mullingar, Ireland.
National Centre for Inherited Metabolic Disorders, Children's University Hospital, Dublin, Ireland.
BMJ Case Rep. 2015 Feb 26;2015:bcr2014208514. doi: 10.1136/bcr-2014-208514.
A fatigued 8-year-old boy was found to have sideroblastic anaemia (haemoglobin 7.8 g/dL) which over time became transfusion dependent. Subtle neurological dysfunction, initially manifesting as mild spastic diplegia, was slowly progressive and ultimately led to wheelchair dependence. Elevated plasma lactate and urinary 3-methylglutaconate led to a muscle biopsy which confirmed partial complex IV deficiency. PCR in leucocytes and muscle was negative for mitochondrial DNA (mtDNA) deletions. Faltering growth prompted an insulin tolerance test which confirmed growth hormone sufficiency and adrenal insufficiency. Plasma renin was elevated and adrenal androgens were low, suggesting primary adrenal insufficiency. Glucocorticoid and mineralocorticoid replacement therapy was initiated. A renal tubular Fanconi syndrome and diabetes mellitus developed subsequently. Sideroblastic anaemia and primary adrenal insufficiency, both individually and collectively, are associated with mtDNA deletion; however, absence of the same does not exclude the possibility that sideroblastic anaemia and primary adrenal insufficiency are of mitochondrial origin.
一名8岁疲劳男孩被诊断为铁粒幼细胞性贫血(血红蛋白7.8 g/dL),随着时间推移,该病发展为依赖输血。轻微的神经功能障碍最初表现为轻度痉挛性双瘫,呈缓慢进展,最终导致依赖轮椅。血浆乳酸和尿3 - 甲基戊二酸水平升高,促使进行肌肉活检,结果证实存在部分复合物IV缺乏。白细胞和肌肉的PCR检测显示线粒体DNA(mtDNA)缺失为阴性。生长发育迟缓促使进行胰岛素耐量试验,结果证实生长激素充足但存在肾上腺功能不全。血浆肾素升高,肾上腺雄激素水平低,提示原发性肾上腺功能不全。遂开始糖皮质激素和盐皮质激素替代治疗。随后出现肾小管性范科尼综合征和糖尿病。铁粒幼细胞性贫血和原发性肾上腺功能不全,无论是单独还是共同出现,都与mtDNA缺失有关;然而,未检测到mtDNA缺失并不能排除铁粒幼细胞性贫血和原发性肾上腺功能不全源于线粒体的可能性。