Familial Blue Rubber Bleb Nevus Syndrome in Pregnancy with Spinal Epidural Involvement

Overview

Journal Case Rep Obstet Gynecol

Publisher Wiley

Specialty Gynecology & Obstetrics

Date 2013 Jun 14

PMID 23762681

Citations 5

Authors

Daigo Ochiai

Kei Miyakoshi

Kazumi Yakubo

Tatsuro Fukuiya

Yasunori Yoshimura

Affiliations

Soon will be listed here.

Abstract

Blue rubber bleb nevus syndrome (BRBNS) is a rare vascular disorder characterized by multiple venous malformations (VMs) of the skin, gastrointestinal tract, and other organs. To date, several cases of sporadic BRBNS involving various parts of the pregnant woman's body have been reported; however, BRBNS in pregnancy with spinal epidural involvement has not been reported. Here, we describe the clinical features and management of familial BRBNS in pregnancy. The patient presented with multiple VMs on her head, neck, floor of the mouth, trunk, leg, foot, and vulva and spinal epidural lesions. The patient's mother and sister also exhibited multiple VMs similar lesions, indicating a familial form of BRBNS. Cesarean section under general anesthesia was performed, and a healthy male neonate was delivered. The mother's postoperative course was uneventful and her VMs decreased in size after delivery. Physicians should consider the possibility of systemic diseases and familial inheritance in cases of VMs.

Citing Articles

Delivery management of pregnant blue rubber bleb nevus syndrome patients: a case report and literature review.

Nagahama M, Chigusa Y, Shiraki A, Takei Y, Yamaguchi A, Mizota T Nagoya J Med Sci. 2024; 86(3):507-513.

PMID: 39355358 PMC: 11439602. DOI: 10.18999/nagjms.86.3.507.

A multidisciplinary approach to caring for a pregnant patient with blue rubber bleb nevus syndrome: A case report.

Daviskiba S, Shuman H, Ayyash M, Vadlamudi G, Smietana S, Goyert G Case Rep Womens Health. 2022; 34:e00403.

PMID: 35299692 PMC: 8920868. DOI: 10.1016/j.crwh.2022.e00403.

Blue Rubber Bleb Nevus Syndrome With Multiple Cavernoma-Like Lesions on MRI: A Familial Case Report and Literature Review.

Anwar G, Elma P, Adib J, Ilse M, Alonso G, Ciltlaltepelt S Front Neurol. 2020; 11:176.

PMID: 32318009 PMC: 7154106. DOI: 10.3389/fneur.2020.00176.

Anesthetic considerations for blue rubber bleb nevus syndrome: a case report.

Aizawa M, Ishihara S, Yokoyama T JA Clin Rep. 2020; 5(1):83.

PMID: 32026052 PMC: 6967292. DOI: 10.1186/s40981-019-0304-4.

Pulmonary embolism during pregnancy in a case of blue rubber bleb nevus syndrome.

Liu Y, Jiang Y, Du J Chin Med J (Engl). 2019; 132(12):1499-1501.

PMID: 31205114 PMC: 6629321. DOI: 10.1097/CM9.0000000000000268.

References

Kanai M, Noike M, Masaki C, Kita N, Ashida T, Kobayashi T . Severe gastrointestinal bleeding during pregnancy in a case of blue rubber bleb nevus syndrome. Semin Thromb Hemost. 2005; 31(3):284-9. DOI: 10.1055/s-2005-872433. View

Nirmal D, Rhodes S, Hassanaien M . Pregnancy in blue rubber bleb syndrome: a case report. Am J Obstet Gynecol. 2008; 199(1):e14-5. DOI: 10.1016/j.ajog.2008.01.028. View

Marler J, Mulliken J . Vascular anomalies: classification, diagnosis, and natural history. Facial Plast Surg Clin North Am. 2007; 9(4):495-504. View

Vijay K, Shetty A, Rajasekaran S . Symptomatic vertebral hemangioma in pregnancy treated antepartum. A case report with review of literature. Eur Spine J. 2008; 17 Suppl 2:S299-303. PMC: 2525908. DOI: 10.1007/s00586-008-0592-2. View

Chi J, Manley G, Chou D . Pregnancy-related vertebral hemangioma. Case report, review of the literature, and management algorithm. Neurosurg Focus. 2005; 19(3):E7. DOI: 10.3171/foc.2005.19.3.8. View

Akhaddar A, Oukabli M, En-Nouali H, Boucetta M . Acute postpartum paraplegia caused by spinal extradural capillary hemangioma. Int J Gynaecol Obstet. 2009; 108(1):75-6. DOI: 10.1016/j.ijgo.2009.08.012. View

Boon L, Mulliken J, Vikkula M, Watkins H, Seidman J, Olsen B . Assignment of a locus for dominantly inherited venous malformations to chromosome 9p. Hum Mol Genet. 1994; 3(9):1583-7. DOI: 10.1093/hmg/3.9.1583. View

Terata M, Kikuchi A, Kanasugi T, Fukushima A, Sugiyama T . Association of blue rubber bleb nevus syndrome and placenta previa: report of a case. J Clin Ultrasound. 2012; 41(8):517-20. DOI: 10.1002/jcu.22000. View

Kisu T, Yamaoka K, Uchida Y, Mori H, Nakama T, Hisatsugu T . A case of blue rubber bleb nevus syndrome with familial onset. Gastroenterol Jpn. 1986; 21(3):262-6. DOI: 10.1007/BF02774569. View

10.

Tanaka N, Tsuda M, Samura O, Miyoshi H, Hara T, Kudo Y . Blue rubber bleb nevus syndrome: Report of a patient with hemangiomas of the vaginal portion of the cervix appearing during pregnancy. J Obstet Gynaecol Res. 2007; 33(4):546-8. DOI: 10.1111/j.1447-0756.2007.00568.x. View

11.

Razvi S, Stefak Y, Bird J . Caesarean section for a woman with Von Hippel-Lindau disease. Int J Obstet Anesth. 2009; 18(3):294-5. DOI: 10.1016/j.ijoa.2009.01.005. View

12.

Demiraran Y, Ozgon M, Utku T, Bozkurt P . Epidural anaesthesia for Caesarean section in a patient with von Hippel-Lindau disease. Eur J Anaesthesiol. 2001; 18(5):330-2. DOI: 10.1046/j.0265-0215.2000.00824.x. View

13.

Wada O, Unno N, Matsuoka R, Taketani Y . A case report: pregnancy complicated by blue rubber-bleb nevus syndrome. J Obstet Gynaecol Res. 1999; 25(4):261-3. DOI: 10.1111/j.1447-0756.1999.tb01159.x. View

14.

Sarubbo S, Garofano F, Maida G, Fainardi E, Granieri E, Cavallo M . Spontaneous and idiopathic chronic spinal epidural hematoma: two case reports and review of the literature. Eur Spine J. 2009; 18(11):1055-61. PMC: 2899401. DOI: 10.1007/s00586-009-1175-6. View

15.

McCarthy T, Leighton R, Mushambi M . Spinal anaesthesia for caesarean section for a woman with von Hippel Lindau disease. Int J Obstet Anesth. 2010; 19(4):461-2. DOI: 10.1016/j.ijoa.2010.03.015. View

16.

Munkvad M . Blue rubber bleb nevus syndrome. Dermatologica. 1983; 167(6):307-9. DOI: 10.1159/000249805. View