Hansen Jonas Kjeldbjerg, Møller Hans Ulrik
Department of Pediatrics, Regional Hospital Viborg, Viborg, Denmark.
Department of Pediatric Ophthalmology, Aarhus University Hospital, Aarhus, Denmark.
Neuropediatrics. 2017 Jun;48(3):185-187. doi: 10.1055/s-0036-1597614. Epub 2017 Jan 4.
Tadpole pupil is a rarely encountered phenomenon caused by episodic, segmental iris dilator muscle spasm of short duration (2-15 minutes), occurring in clusters without a known precipitating factor. It has most commonly been described in women aged 28 to 48 years. A few hypotheses on pathogenesis have been discussed but none has been proved. Here, we present an adolescent girl with bilateral tadpole pupil that appeared during physical exercise. This is the first pediatric case of tadpole pupil, not caused by preceding surgery, to be published. Based on (1) this case in which tadpole pupil developed when the norepinephrine level rose during exercise, (2) the high ratio of patients with tadpole pupil who concurrently have or later develop Horner syndrome, in which denervation hypersensitivity is well described, (3) a previous report of a patient with both tadpole pupil and Horner syndrome who had denervation hypersensitivity on pharmacological testing, (4) a 29-year-old man with unilateral tadpole pupil induced by exercise, and (5) a 19-year-old man with bilateral tadpole pupil and possible autonomic neuropathy, we suggest denervation hypersensitivity as a probable pathogenic mechanism causing tadpole pupil. In addition, a suggestion for investigations to be performed in future pediatric cases is provided.
蝌蚪样瞳孔是一种罕见现象，由短暂发作（2 - 15分钟）的节段性虹膜开大肌痉挛引起，呈簇状发作，无已知诱发因素。最常见于28至48岁的女性。关于其发病机制已讨论了一些假说，但均未得到证实。在此，我们报告一名青春期女孩，在体育锻炼期间出现双侧蝌蚪样瞳孔。这是首例非因先前手术导致的蝌蚪样瞳孔儿科病例并已发表。基于：（1）该病例中，运动期间去甲肾上腺素水平升高时出现蝌蚪样瞳孔；（2）蝌蚪样瞳孔患者中同时患有或后来发展为霍纳综合征的比例较高，而在霍纳综合征中去神经超敏反应已有充分描述；（3）先前有一例同时患有蝌蚪样瞳孔和霍纳综合征的患者在药理学测试中有去神经超敏反应的报告；（4）一名29岁男性运动诱发单侧蝌蚪样瞳孔；（5）一名19岁男性双侧蝌蚪样瞳孔且可能存在自主神经病变，我们认为去神经超敏反应可能是导致蝌蚪样瞳孔的发病机制。此外，还为未来儿科病例的检查提供了建议。
