Division of Headache, Department of Neurology, NYU Langone Health, New York, NY, USA.
Headache. 2021 Jul;61(7):1132-1135. doi: 10.1111/head.14174. Epub 2021 Jul 29.
To report two cases of hemicrania continua (HC) in a mother and daughter.
HC is a rare primary headache disorder belonging to the family of trigeminal autonomic cephalalgias (TACs). Unlike migraine, familial cases of TACs are rare, and we know relatively little of their inheritance pattern and genetic mechanisms.
We present a mother and daughter with HC. We compare the similarities and differences between this family and the first report of familial HC and discuss the implications for future studies.
Both the mother and daughter presented with a constant, side-locked headache of moderate intensity, with episodic exacerbations of more severe pain that are associated with ipsilateral cranial autonomic activation. After negative workup, both patients were started on indomethacin and achieved absolute response at different doses, confirming HC.
Our report further corroborates other reports of familial TACs that TACs are primary headaches possibly attributable to genetic factors, albeit detailed mechanisms remain elusive. Nevertheless, whether clinical presentation and treatment responses would be substantially different between sporadic and familial HCs remain unclear.
报告一例母女共患连续性偏头痛(HC)病例。
HC 是一种罕见的原发性头痛疾病,属于三叉神经自主神经性头痛(TACs)家族。与偏头痛不同,TACs 的家族性病例较为罕见,我们对其遗传模式和遗传机制知之甚少。
我们报告了一例母女共患 HC 的病例。我们比较了这个家族与首例家族性 HC 报告之间的异同,并讨论了对未来研究的影响。
母亲和女儿均表现为持续的、单侧锁定的中度头痛,伴有同侧颅自主神经激活的更严重疼痛的阵发性加重。经过阴性检查,两名患者均开始使用吲哚美辛治疗,且在不同剂量下均获得了绝对缓解,从而确诊为 HC。
我们的报告进一步证实了其他 TACs 家族性病例的报告,即 TACs 可能是原发性头痛,归因于遗传因素,但详细的机制仍不清楚。然而,散发性和家族性 HC 之间的临床表现和治疗反应是否会有很大差异仍不清楚。