Maternal and fetal outcomes of intraplacental choriocarcinoma complicated by fetomaternal hemorrhage: a systematic review

Figures & data

Table 1. PRISMA flow diagram of study selection.

Table 2. Joanna Briggs Institute Critical Appraisal Checklist for case reports.

	Title	Reference	D1	D2	D3	D4	D5	D6	D7	D8
1	Fetomaternal hemorrhage: a clue to intraplacental choriocarcinoma and neonatal malignancy	Simões et al. [14]	Yes	Yes	Yes	Unclear	Unclear	Yes	Yes	Yes
2	Fetomaternal hemorrhage caused by an intraplacental choriocarcinoma	Jeong et al. [15]	Yes	Yes	Yes	Unclear	Unclear	Yes	Yes	Yes
3	Hémorragie fœtomaternelle sur choriocarcinome placentaire	Touboul et al. [16]	Yes	Yes	Yes	Unclear	Yes	Yes	Yes	Yes
4	Intraplacental choriocarcinoma with fetomaternal hemorrhage: a case study and literature review	Takahashi et al. [17]	Yes	Yes	Yes	Yes	Yes	Yes	Yes	Yes
5	Intraplacental choriocarcinoma and fetomaternal haemorrhage and maternal disseminated intravascular coagulopathy in a term pregnancy: a case report	Hookins and Vatsayan [18]	Yes	Yes	Yes	Yes	Unclear	Yes	Yes	Yes
6	Intraplacental choriocarcinoma coexisting with fetomaternal hemorrhage: case report, chemotherapy management, and literature review	She et al. [11]	Yes	Yes	Yes	Yes	Yes	Yes	Yes	Yes
7	Fetomaternal hemorrhage caused by intraplacental choriocarcinoma: a case report and review of literature in Japan	Koike et al. [19]	Yes	Yes	Yes	Unclear	Yes	Yes	Yes	Yes
8	Massive feto-maternal hemorrhage: an early presentation of women with gestational choriocarcinoma	Lam et al. [20]	Yes	Yes	Yes	Unclear	Yes	Yes	Yes	Yes
9			Yes	Yes	Yes	Yes	Yes	Yes	Yes	Yes
10	Massive fetomaternal hemorrhage caused by an intraplacental choriocarcinoma: a case report	Henningsen et al. [21]	Yes	Yes	Yes	Yes	Yes	Yes	Yes	Yes
11	Prenatal findings in a case of massive fetomaternal hemorrhage associated with intraplacental choriocarcinoma	Aso et al. [8]	Yes	Yes	Yes	Yes	Yes	Yes	Yes	Yes
12	Term pregnancy with choriocarcinoma presenting as severe fetal anemia and postpartum hemorrhage	Peng et al. [22]	Yes	Yes	Yes	Unclear	Yes	Yes	Yes	Yes
13	Intraplacental choriocarcinoma with fetomaternal transfusion	Takai et al. [23]	Yes	Yes	Yes	Unclear	Yes	Yes	Yes	Yes
14	Intraplacental choriocarcinoma: systematic review and management guidance	Jiao et al. [24]	Yes	Yes	Yes	Unclear	Yes	Yes	Yes	Yes
15
16	Infantile choriocarcinoma with idiopathic massive fetomaternal hemorrhage	Chou et al. [25]	No	No	Yes	Unclear	Yes	Yes	Yes	Yes
17	Intraplacental choriocarcinoma associated with pregnancy continuum: a diagnostic dilemma	Datta et al. [10]	Yes	Yes	Yes	Unclear	Unclear	Yes	Yes	Yes
18	Intraplacental choriocarcinoma as an unexpected cause of intrauterine death at term	Nagel et al. [26]	Yes	Yes	Yes	Yes	Yes	Yes	Yes	Yes
19	Intraplacental choriocarcinoma in twin pregnancy causing fetomaternal haemorrhage and single twin demise: case report	Schepisi et al. [27]	Yes	Yes	Yes	Unclear	Yes	Yes	Yes	Yes

Table 3. Characteristics of the cases with IC and FMH included in the review.

	Description of the case
	Mother												Fetus
Source of data	Age	Gravida/ para	Presentation	Size of tumor	Site of diagnosis	Time of diagnosis after delivery	HCG at diagnosis (IU/L)	Metastasis	Treatment	Outcome	Gestational age	Delivery	Symptoms	CTG	EFW (g)	Gender	Apgar score (1/5 min)	K–B test	Metastasis	Treatment	Outcome	HGB (g/dL)
Takai et al. [23]	35	G2P0	Asymptomatic	20 mm	Placental examination	1–3 days	NA	–	HCG monitoring	Alive	37	Emergency CS	Fetal distress, IUGR (known), neonatal anemia Hb 3.0	Pathological	1878	Male	6/7	Positive for FMH	–	Blood transfusion	Discharged	3
Lam et al. [20]	27	G1	Chest pain 3 weeks after VD	NA	NA	3 weeks	1,200,000	Lung	Chemotherapy	Alive	39	VD	Neonatal anemia Hb 5.8, heart failure	Normal	3160	NA	9/NA	Positive for FMH	–	Blood transfusion	Discharged	5.8
	30	G2P1	Asymptomatic, heavy bleeding 7 weeks after VD	10 mm	D&C, retrospective placental examination	7 weeks	986	Uterus	Chemotherapy	Alive	35	VD	Intrauterine death	–	3000	NA	–	Positive for FMH	–	–	Died	–
Chou et al. [25]	NA	NA	NA	Grossly normal	NA	NA	NA	–	HCG monitoring	Alive	40	Emergency CS	Fetal distress, neonatal anemia	Pathological	3020	Male	5/9	NA	Liver	Blood transfusion; hepatic segmentectomy	Died after rupture of hepatic tumor	5.9
Koike et al. [19]	31	G1	Asymptomatic	NA	Placental examination	1–3 days	NA	–	HCG monitoring	Alive	39	Emergency CS	Fetal distress, IUGR, neonatal anemia Hb 5.1	Pathological	2272	Female	5/6	Positive for FMH	–	Blood transfusion	Discharged	Negative
Nagel et al. [26]	30	G2P0 previous hydatiform mole	Vaginal bleeding 3 months after delivery	Grossly normal	Retrospective placental examination	3 months	11.311 (three months postpartum)	–	Chemotherapy	Alive	40	VD	Intrauterine death	–	3275	Male	–	Positive for FMH	–	–	Died
Takahashi et al. [17]	30	G1	Reduced fetal movements	20 mm	Placental examination	1–3 days	NA		HCG monitoring	Alive	38 + 2	Emergency CS	Fetal distress, IUGR, neonatal anemia Hb 5.1	Pathological	2272	Female	5/6	NA	–	Blood transfusion	Discharged	5.1
Henningsen et al. [21]	47	P7	Reduced fetal movements	30 mm	Retrospective placental examination	1–3 days	510 (17 days postpartum)	–	HCG monitoring	Alive	35 + 6	Emergency CS	Fetal distress, neonatal anemia Hb 2.1	Pathological	2800	Female	0/7	NA	–	Blood transfusion	Discharged	2.1
Aso et al. [8]	27	G2P1	Reduced fetal movements	15 mm	Placental examination	1–3 days	90,600	–	Chemotherapy	Alive	37	Emergency CS	Fetal distress, neonatal anemia Hb 3.6, hypovolemic shock	Pathological	2450	Male	7/7	NA	–	Blood transfusion	Died	3.6
Datta et al. [10]	23	G2P1	Reduced fetal movements, persistent vaginal bleeding 4 weeks post-partum, headache  + chest pain 2 months later	NA	Retrospective placental examination	2 months	766,433	Lung	Chemotherapy	Alive	36	CS	Fetal distress, neonatal anemia Hb 5.0	Pathological	2925	NA	8/10	Positive for FMH	–	NA	Discharged	5
Touboul et al. [16]	34	G1	Asymptomatic	13 mm	Placental examination	1–3 days	NA	–	HCG monitoring	Alive	41	Operative vaginal delivery	Asymptomatic, neonatal anemia Hb 7.6	Non reassuring	3820	NA	8	Positive for FMH	–	NA	Discharged	NA
Jeong et al. [15]	27	G1	Reduced fetal movements	NA	Placental examination	4 days	195 (18 days post-partum)	–	HCG monitoring	Alive	31 + 4	Emergency CS	Fetal cardiomegaly and ascites, fetal distress	–	2020	NA	3/5	NA	–	Blood transfusion, mechanical ventilation	Died	NA
Peng et al. [22]	34	G3P2	Reduced fetal movements, Vaginal bleeding 2 weeks post CS	NA	D&C	2 weeks	538,312	Uterus; lung	Chemotherapy	Alive	37	Emergency CS	Oligoamnios, fetal distress, neonatal anemia Hb 3.7	Pathological	2650	Male	2/3	NA	–	Blood transfusion	Discharged	3.7
Jiao et al. [24]	32	G5P4	Asymptomatic	20 mm	Placental examination	NA	NA	–	HCG monitoring	Alive	40	VD	Intrauterine death	–	NA	NA	–	Positive for FMH	–	–	Died
	32	G1P0	Asymptomatic	<10 mm	Placental examination	NA	NA	–	HCG monitoring	Alive	40	Emergency CS	Fetal distress, neonatal anemia	Pathological	NA	Female	NA	NA	–	Blood transfusion	Discharged	NA
She et al. [11]	21	G1	Reduced fetal movements	20 mm	Placental examination	6 days	31,280 (6 days postpartum) 42,269 (95 days postpartum)	Uterus, lung	–	Died	35	Emergency CS	Fetal anemia diagnosed with US, neonatal anemia Hb 2.6	Pathological	2170	Male	8/8	Positive for FMH	–	Blood transfusion	Discharged	30.4
Hookins and Vatsayan [18]	31	G1	Reduced fetal movements, DIC after CS, vagina bleeding 3 weeks after CS	11 mm	D&C	3 weeks	1,000,000 (3 weeks postpartum)	Uterus, right adnexa	Chemotherapy	Alive	38 + 1	Emergency CS	Fetal distress, neonatal anemia Hb 4.7	Pathological	3050	Male	8/7	Positive for FMH	–	NA	Discharged	NA
Simões et al. [14]	35	G2P1	Vaginal bleeding after VD	NA	D&C	2 months	66.780	Lung	NA	Alive	39	VD	Neonatal distress, anemia Hb 3.6	Non reassuring	3200	Female	7/9	Not performed	–	Blood transfusion, antibiotic therapy	Discharged	Negative
Schepisi et al. [27]	34	G1P0, D/D twin pregnancy	Preterm labor, twin A fetal distress	15 mm	Placental examination	1–3 days	18 (1 month post-partum)	–	HCG monitoring	Alive	35	Emergency CS	Twin A: fetal distress, neonatal death, neonatal anemia Hb 2.1	Pathological	NA	NA	NA	NA	–	–	Died	NA
													Twin B: SGA, no complication at birth	–	NA	NA	NA	NA	–	–	Discharged	NA

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Data availability statement

The authors confirm that data supporting the findings of this study are available within the article.