Tishler P V, Redline S, Ferrette V, Hans M G, Altose M D
Brockton/West Roxbury Veterans Affairs Medical Center, Brockton, MA 02401-5596 USA.
Am J Respir Crit Care Med. 1996 Jun;153(6 Pt 1):1857-63. doi: 10.1164/ajrccm.153.6.8665046.
We studied the relationship of sudden unexpected infant death/apparent life-threatening events (ALTE) to obstructive sleep apnea (OSA) in 74 index probands who had either sleep-laboratory-confirmed OSA or a clinical diagnosis of OSA requiring treatment, 62 matched control probands, and their spouses and first- and second-degree relatives. Sleep was monitored in the home overnight, and OSA was defined by respiratory disturbance indices (number of apneas/hypopneas per hour of sleep) corrected for normal increases with age. Information on sudden unexpected infant death/ALTE was obtained by questionnaire and was corroborated. For living relatives, data were obtained by questionnaire, examination, or study (cephalometric radiographs, ventilatory responsiveness to hypercapnia and hypoxia). Eight index families had 10 infants with sudden unexpected infant death/ALTE; two control families had three infants with sudden death (p = 0.11). All told, 91 of the 136 families (index plus control) included members with OSA, and all 10 infant death/ALTE families were among these (versus zero of 45 families with no OSA; p = 0.03). The sudden infant death/ALTE families had a greater frequency of two or more members with OSA (p = 0.06), reported more respiratory disease or allergy, were more frequently brachycephalic (p = 0.05), and had a smaller mean posterior nasal spine-basion distance (p = 0.0001) and ratio of anterior mandibular/anterior maxillary dental height (p < 0.05). Ventilatory responses to hypoxia were reduced in members of families with OSA (p = 0.008), with a trend toward the greatest blunting in subjects from families with OSA plus sudden unexpected infant death/ALTE. Thus, OSA in adults and sudden unexpected infant death/ALTE in their biologic relatives appear to be related. Familial factors influencing this association may include the degree of the predilection for OSA, liability for respiratory illness or allergy, dimensions of the oral-pharyngeal airway, and ventilatory response to hypoxia.
我们研究了74名索引先证者（这些先证者要么经睡眠实验室确诊为阻塞性睡眠呼吸暂停（OSA），要么经临床诊断为需要治疗的OSA）、62名匹配的对照先证者及其配偶、一级和二级亲属中，婴儿猝死/明显危及生命事件（ALTE）与阻塞性睡眠呼吸暂停（OSA）之间的关系。在家中对睡眠进行整夜监测，并根据随年龄正常增加而校正的呼吸紊乱指数（每小时睡眠中的呼吸暂停/低通气次数）来定义OSA。通过问卷调查获取婴儿猝死/ALTE的信息，并进行了核实。对于在世的亲属，通过问卷调查、检查或研究（头影测量X线片、对高碳酸血症和低氧的通气反应性）获取数据。8个索引家庭中有10名婴儿发生婴儿猝死/ALTE；2个对照家庭中有3名婴儿猝死（p = 0.11）。总体而言，136个家庭（索引家庭加对照家庭）中有91个家庭的成员患有OSA，所有10个婴儿死亡/ALTE家庭都在这些家庭之中（而45个无OSA的家庭中为零；p = 0.03）。婴儿猝死/ALTE家庭中，有两名或更多成员患有OSA的频率更高（p = 0.06），报告的呼吸道疾病或过敏更多，更频繁地为短头畸形（p = 0.05），平均后鼻棘-鼻根距离更小（p = 0.0001），下颌前/上颌前牙高度比更小（p < 0.05）。OSA家庭成员对低氧的通气反应降低（p = 0.008），在患有OSA加婴儿猝死/ALTE家庭的受试者中，这种钝化趋势最为明显。因此，成年人的OSA与其生物学亲属中的婴儿猝死/ALTE似乎有关联。影响这种关联的家族因素可能包括对OSA的易患程度、对呼吸道疾病或过敏的易感性、口咽气道的尺寸以及对低氧的通气反应。
