Guidi L, Zeppilli P, Sassara M, Ghirlanda G
G Ital Cardiol. 1981;11(8):1151-9.
Pure acute Pandysautonomia (PAP), first described in 1969 by Young, is a rare but well defined clinical entity, in which the isolate involvement of Autonomic Nervous System (ANS) gives rise to a characteristic clinical admixture of symptoms and signs all due to the alteration of parasympathetic and sympathetic control of neurovegetative functions. The most prominent clinical features are: 1) postural hypotension without changes in heart rate leading to orthostatic syncopal or near-syncopal episodes, 2) abnormalities of intestinal tract and urinary bladder motility, leading to constipation alternating with diarrhea and urinary retention, 3) loss of sweating, 4) ocular abnormalities. We report a case of a 17 year old man with PAP, in whom almost complete resolution of clinical symptoms and signs of ANS involvement was observed within 9 months from the onset of illness. In our case, besides an accurate clinical and laboratory follow-up, particular attention was paid to investigate the cardiac responses to autonomic reflex tests and pharmacological interventions (atropine and beta-blocking drugs administration) during the entire course of illness. During the acute phase, complete vagal and almost complete beta-sympathetic "denervation" of the heart leading to disabling postural hypotension, fixed resting tachycardia and total unresponsiveness to Valsalva manoeuvre, were observed. PAP is a rare condition which must be known from cardiologists for a more complete knowledge of the etiology and pathophysiological mechanisms of postural syncopal attacks and must be distinguished from the less favourable progressive autonomic failure of the Shy-Drager syndrome.
纯急性全自主神经功能不全(PAP)于1969年由扬首次描述,是一种罕见但定义明确的临床实体,其中自主神经系统(ANS)的孤立受累导致一系列特征性的临床症状和体征混合出现,所有这些均归因于副交感神经和交感神经对神经植物性功能控制的改变。最突出的临床特征为:1)体位性低血压,心率无变化,导致直立性晕厥或接近晕厥发作;2)肠道和膀胱运动异常,导致便秘与腹泻交替以及尿潴留;3)出汗丧失;4)眼部异常。我们报告一例17岁患PAP的男性病例,该患者在发病后9个月内,自主神经系统受累的临床症状和体征几乎完全消退。在我们的病例中,除了进行准确的临床和实验室随访外,在疾病全过程中还特别关注研究心脏对自主神经反射试验和药物干预(给予阿托品和β受体阻滞剂)的反应。在急性期,观察到心脏完全迷走神经和几乎完全的β交感神经“去神经支配”,导致严重的体位性低血压、固定的静息心动过速以及对瓦尔萨尔瓦动作完全无反应。PAP是一种罕见疾病,心脏病专家必须了解它,以便更全面地认识体位性晕厥发作的病因和病理生理机制,并且必须将其与预后较差的夏伊-德雷格综合征进行鉴别,后者为进行性自主神经功能衰竭。