Waddell W R
Ann Surg. 1975 Mar;181(3):299-302. doi: 10.1097/00000658-197503000-00009.
A female patient with Gardner's syndrome was treated with delta1-testololactone (200 mg daily) because of growth of a large desmoid tumor in the pelvis and lower abdomen and a tumor in a scar from a previous laparotomy. There was also pain and swelling of the left leg. An immediate effect of the drug therapy was complete relief of pain followed shortly thereafter by disappearance of the edema of the leg. After two months, the numerous sebaceous cysts were less prominent. The gross measurements of the diameter of the pelvic and lower abdominal tumor clearly demonstrated tumor shrinkage following therapy. Small polyps scattered over the rectal mucosa and numerous osteomata were not demonstrably affected. After one year of treatment with delta1-testololactone, a laparotomy for partial small bowel obstruction was necessary. Obstruction was caused by the involvement of small bowel mesentery and the bowel itself in a contracted residuum of dense fibrous tissue. Substitution of theophylline and chlorothiazide for the testololactone in Januray 1974 was followed by further diminution of the measurable abdominal and pelvic desmoids. All of these compounds synergize the action of 3',5'-adenosine monophosphate and at least the latter two may function by inhibiting the action of 3',5'-adenosine monophosphate diesterase.
一名患有加德纳综合征的女性患者，因盆腔和下腹部的巨大硬纤维瘤以及既往剖腹手术瘢痕处的肿瘤生长，同时伴有左腿疼痛和肿胀，接受了δ1-睾内酯（每日200毫克）治疗。药物治疗的即时效果是疼痛完全缓解，随后不久腿部水肿消失。两个月后，大量皮脂腺囊肿不再那么明显。盆腔和下腹部肿瘤直径的粗略测量清楚地显示治疗后肿瘤缩小。散布在直肠黏膜上的小息肉和众多骨瘤未显示出明显受影响。在用δ1-睾内酯治疗一年后，因部分小肠梗阻需要进行剖腹手术。梗阻是由小肠系膜和肠本身卷入致密纤维组织的收缩残留物所致。1974年1月用茶碱和氯噻嗪替代睾内酯后，可测量的腹部和盆腔硬纤维瘤进一步缩小。所有这些化合物都能增强3',5'-单磷酸腺苷的作用，至少后两种化合物可能通过抑制3',5'-单磷酸腺苷二酯酶的作用发挥功能。
