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Abstract：
BACKGROUND: Women are counseled preconceptionally about the potential risks of rAML progression and chance of complications during and due to pregnancy. However, a systematic search investigating the evidence on which this advice is based does not exist. The aim of this systematic review is to determine the effect of pregnancy on renal angiomyolipoma (rAML) size and risk of haemorrhage in patients with tuberous sclerosis complex (TSC).
METHODS: We searched PubMed, EMBASE, Medline and ClinicalTrials.gov using terms for \"renal angiomyolipoma\" and \"pregnancy\". English-language articles published between January 1st 2000, and December 31st 2020 of which full-text was available were included. The initial search resulted in 176 articles. After the screening process we included 45 case reports and 1 retrospective study. For the retrospective study we assessed the risk of bias using the Newcastle-Ottawa Scale. We included articles about renal AML and pregnancy with and without an established diagnosis of TSC. From these articles we recorded the rAML sizes and rAML complications.
RESULTS: Seven case reports, from a total of 45 case reports, provided follow-up data on renal AML size (these were all cases of renal AML without a known diagnosis of TSC). Of these cases, renal AML size decreased in one patient, was stable in one patient, increased in three patients and fluctuated in two others. Renal AML size of women who suffered a haemorrhage were significantly larger (12.1 ± 4.6 cm) than rAMLs of women who did not suffer a haemorrhage (8.3 ± 3.2 cm). Data from the retrospective study showed no difference in renal complications between the women with and without a history of pregnancy. Haemorrhage occurred in 30% of the women with a history of pregnancy (n = 20) and in 11% in the patients without a history of pregnancy (n = 2), however this retrospective study had methodological limitations.
CONCLUSIONS: The effect of pregnancy on renal AML size and complications in patients with TSC is unclear. More research is needed to determine the risk of pregnancy on TSC-associated kidney disease in TSC patient.
METHODS: We searched PubMed, EMBASE, Medline and ClinicalTrials.gov using terms for \"renal angiomyolipoma\" and \"pregnancy\". English-language articles published between January 1st 2000, and December 31st 2020 of which full-text was available were included. The initial search resulted in 176 articles. After the screening process we included 45 case reports and 1 retrospective study. For the retrospective study we assessed the risk of bias using the Newcastle-Ottawa Scale. We included articles about renal AML and pregnancy with and without an established diagnosis of TSC. From these articles we recorded the rAML sizes and rAML complications.
RESULTS: Seven case reports, from a total of 45 case reports, provided follow-up data on renal AML size (these were all cases of renal AML without a known diagnosis of TSC). Of these cases, renal AML size decreased in one patient, was stable in one patient, increased in three patients and fluctuated in two others. Renal AML size of women who suffered a haemorrhage were significantly larger (12.1 ± 4.6 cm) than rAMLs of women who did not suffer a haemorrhage (8.3 ± 3.2 cm). Data from the retrospective study showed no difference in renal complications between the women with and without a history of pregnancy. Haemorrhage occurred in 30% of the women with a history of pregnancy (n = 20) and in 11% in the patients without a history of pregnancy (n = 2), however this retrospective study had methodological limitations.
CONCLUSIONS: The effect of pregnancy on renal AML size and complications in patients with TSC is unclear. More research is needed to determine the risk of pregnancy on TSC-associated kidney disease in TSC patient.
摘要：
背景：妇女在怀孕期间和由于妊娠引起的rAML进展的潜在风险和并发症的机会方面有先入为主的建议。然而,调查该建议所依据的证据的系统搜索不存在。这项系统评价的目的是确定妊娠对结节性硬化症（TSC）患者肾血管平滑肌脂肪瘤（rAML）大小和出血风险的影响。
方法：我们搜索了PubMed，EMBASE,Medline和ClinicalTrials.gov使用术语“肾血管平滑肌脂肪瘤”和“妊娠”。包括2000年1月1日至2020年12月31日之间发表的英文文章，其中全文可用。最初的搜索结果是176篇文章。筛选过程后，我们纳入了45例病例报告和1例回顾性研究。在回顾性研究中，我们使用纽卡斯尔-渥太华量表评估偏倚风险。我们纳入了关于肾AML和妊娠的文章，有或没有确定的TSC诊断。从这些文章中，我们记录了rAML大小和rAML并发症。
结果：7例病例报告，在总共45例病例报告中，提供了肾脏AML大小的随访数据(这些病例均为无已知TSC诊断的肾脏AML).在这些案件中,一名患者的肾AML大小减少，一名患者病情稳定,三名患者增加，另外两名患者波动。出血妇女的肾AML大小（12.1±4.6cm）明显大于未出血妇女的rAML（8.3±3.2cm）。回顾性研究的数据显示，有和没有妊娠史的妇女之间的肾脏并发症没有差异。有妊娠史的妇女（n=20）发生出血，无妊娠史的妇女（n=2）发生出血，占11％。然而,这项回顾性研究有方法学上的局限性.
结论：妊娠对TSC患者肾脏AML大小和并发症的影响尚不清楚。需要更多的研究来确定TSC患者TSC相关肾脏疾病的妊娠风险。
方法：我们搜索了PubMed，EMBASE,Medline和ClinicalTrials.gov使用术语“肾血管平滑肌脂肪瘤”和“妊娠”。包括2000年1月1日至2020年12月31日之间发表的英文文章，其中全文可用。最初的搜索结果是176篇文章。筛选过程后，我们纳入了45例病例报告和1例回顾性研究。在回顾性研究中，我们使用纽卡斯尔-渥太华量表评估偏倚风险。我们纳入了关于肾AML和妊娠的文章，有或没有确定的TSC诊断。从这些文章中，我们记录了rAML大小和rAML并发症。
结果：7例病例报告，在总共45例病例报告中，提供了肾脏AML大小的随访数据(这些病例均为无已知TSC诊断的肾脏AML).在这些案件中,一名患者的肾AML大小减少，一名患者病情稳定,三名患者增加，另外两名患者波动。出血妇女的肾AML大小（12.1±4.6cm）明显大于未出血妇女的rAML（8.3±3.2cm）。回顾性研究的数据显示，有和没有妊娠史的妇女之间的肾脏并发症没有差异。有妊娠史的妇女（n=20）发生出血，无妊娠史的妇女（n=2）发生出血，占11％。然而,这项回顾性研究有方法学上的局限性.
结论：妊娠对TSC患者肾脏AML大小和并发症的影响尚不清楚。需要更多的研究来确定TSC患者TSC相关肾脏疾病的妊娠风险。
