Table 2.

Severe complications occurring during pregnancy or post-partum in women with HHT and published as case report (n = 31)

Complication	Study	Treatment T: Transfusion S: Surgery E: Embolisation D: Drain	Date of events (wg)	Clinical presentation	Term of delivey (wg)	Maternal outcome	Fœtal outcome
Haemothorax	Texier et al. 2018	T + S/HHT–	26	Chest pain	40	Good	Live infant
	Md Noh et al. 2018	E/HHT–	20	Dyspnea	20	Small bowel active hemorrhage	Foetal death
	Raiya et al. 2017	D + E/HHT–	23	Dyspnea, chest pain	40	Good	Live infant
	Jakobi et al. 2001	S/HHT+	26	Severe hypoxemia	40	Hypoxemia PP embolisation	SGA infant
	Adegboyega et al. 1996	D/HHT–	29	Dyspnea Chest pain	40	Discharged no sequelae	Live infant (CS)
	Freixinet et al. 1995	D + S/HHT?	27	Dyspnea	?	Severe mitral regurgitation	Live infant
	Bevelaqua et al. 1992	D + E/HHT–	26	Dyspnea Chest pain	40	Diagnosed 6 wk	Live infant
	Laroche et al. 1992	D + S / HHT +	29	Dyspnea Chest pain	37	Post embolotherapy	Live infant
	Gammon et al. 1990	D + E / HHT -	24	Dyspnea Chest pain	30	Heart failure resolved	Live infant
	Waring et al. 1990	D + E/HHT +	26	Dyspnea	32	Heart failure resolved	Live infant
Hemoptysis	Banerjee et al. 2018		34	Hemoptysis	–	Good	–
	Tandon et al. 2017	reE of PAVM/HHT+	32	Unconscious and hypoxia	37	Good	Live infant (VD)
	Yaniv-Salem et al. 2017	reE of PAVM/HHT+	35	Massive hemoptysis	37	Good	Live infant (VD)
	Wispelaere et al. 1996	−/HHT+	10	Hemoptysis, transient loss of consciousness and tachycardia	Therapeutic abortion	Well, required resection of PAVM	–
Severe Hypoxemia	Worda et al. 2007	−/HHT+	12	Dyspnea, cyanosis	32	Clinically improved	Live infant (CS)
	Jakobi et al. 2001	−/HHT+	25	Hypoxemia IUGR	25	–	Foetal death
	Swinburne et al. 1986	−/HHT+	35	Dyspnea, cyanosis	35	Post partum PAVM surgery Active limited live	Live infant (CS)
Cerebral Ischemic stroke	Swietlik et al. 2008	Craniotom/HHT+	35	Headache and dyspnea Brain abscess + PAVM	35	Post partum Hemothorax requiring re Embolisation	Live infant (CS)
Intracranial hemorrhage	Gillard et al. 1996	CAVM surgery/HHT–	21	Right hemiplegia, aphasia	38.5	Post partum epilepsy, Right hemiparesis	Live infant (CS)
	Neau et al. 1988	Brain surgery/HHT–	30	Right hemiplegia with aphasia violent headache and vomiting	30	Fatal (multiple CAVM, 5 hematomas)	Foetal death (VD)
Pulmonary edema	Euser et al. 2012	HHT+	33	Pre-eclampsia, extensive edema in her legs and face	34	Good	Live infant (CS)
High output heart failure all related to liver AVM	Berthelot et al. 2015	Diuretics/HHT–	25	Dyspnea	33	At day 16 post partum complete regression of congestive signs	Live infant (CS)
	Lai et al. 2010	?/HHT+	36	Rest Dyspnea	36	Good	Live infant (CS)
	Goussous et al. 2009	?/HHT-	29	Right sided heart failure and preterm labor		On post partum day 2: dyspnea and lower extremity edema	? (CS)
	Livneh et al. 1988	Diuretics/HHT+	26	Weakness, dyspnea	35 (?)	At 4 months Post partum Good	Live infant
	Livneh et al. 1988	Diuretics/HHT–	26	Dyspnea	40	At 4 months Post partum no more heart failure	Live infant
Hepatobiliary necrosis	McInroy et al. 1998	Post partum Liver transplantation HHT+	30	Abdominal pain + fever	30	Biliary necrosis, liver transplant postpartum	Live infant
	Bauer et al. 1995	Liver transplant HHT–	–	Abdominal pain dyspnea, Melaena	–	Liver transplant	–
Gastrointestinal bleeding	Hillert et al. 2001	Liver transplantation/HHT+	27	Diffuse abdominal pain cholangitis	29	Good	Live infant (CS)
Branch retinal artery occlusion	Askim et al. 2017	Subcutaneous Heparin + PAVM embolization/HHT–	12	Sudden painless scotoma in left eye	40	Good	Live infant (VD)
Massive intraperitoneal haemorrhage	Sivarani et al. 2010	HHT+	36	High-output cardiac failure	36	Fatal (on post-partum: massive haemorrhage in the intra-peritoneal cavity with multiple AVMs in the gastrointestinal tract)	Live infant (CS)

Legends: HHT- means that the diagnosis was not done before the complication and HHT+ means that the diagnosis was known before the complication. CS Cesarean section, PAVM Pulmonary arteriovenous malformation, SGA Small for gestational age, VD Vaginal delivery