线粒体DNA缺失疾病的临床异质性:皮尔逊综合征的诊断挑战。

American Journal of Medical Genetics Pub Date : 2000-11-27
F Lacbawan, C J Tifft, N L Luban, S M Schmandt, M Guerrera, S Weinstein, M Pennybacker, L J Wong
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摘要

线粒体DNA (mtDNA)疾病的临床表现是多种多样的。通常,最初的症状不符合特定的疾病,很难做出诊断。我们描述了一个病人谁提出了大细胞性贫血。广泛的生化和临床检查未能提供大细胞性贫血的病因。患者在6年的治疗过程中出现步态问题、运动不耐受、间歇性呕吐、身材矮小、皮肤问题和复发性感染。8岁时,她患有伴有共济失调和吞咽困难的脑病。乳酸升高、双侧基底节区钙化和红色纤维不均匀导致mtDNA突变分析。在肌肉活检中发现了一个新的4.4 kb的缺失,从核苷酸位置10,560到核苷酸位置14,980。在血液、颊细胞和毛囊中也存在相同的异质mtDNA缺失,但在母亲的血液中没有,这与患者的散发性突变一致。这个病例强调了在多系统症状的患者中考虑mtDNA紊乱的重要性,这些症状不能通过特定的诊断来解释。
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Clinical heterogeneity in mitochondrial DNA deletion disorders: a diagnostic challenge of Pearson syndrome.

The clinical presentation of mitochondrial DNA (mtDNA) disorders is quite diverse. Very often, the initial symptoms do not fit a specific disease, and diagnosis is difficult to make. We describe a patient who presented with macrocytic anemia. Extensive biochemical and clinical work-up failed to provide an etiology for the macrocytic anemia. The patient over the course of 6 years developed gait problems, exercise intolerance, episodic vomiting, short stature, dermatological problems, and recurrent infection. At age 8 years she had encephalopathy with ataxia and dysphagia. The presence of elevated lactate, bilateral basal ganglia calcification, and ragged red fibers led to mtDNA mutational analysis. A novel 4.4-kb deletion from nucleotide position 10,560 to nucleotide position 14, 980 was identified in muscle biopsy. The same heteroplasmic mtDNA deletion was present in blood, buccal cells, and hair follicles, but not in mother's blood, consistent with sporadic mutation in the patient. This case emphasizes the importance of considering mtDNA disorder in patients with multisystemic symptoms that cannot be explained by a specific diagnosis.

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