DOI: http://dx.doi.org/10.18203/2349-3291.ijcp20194538

Intrauterine upper limb thrombosis: an unusual presentation

Baraturam B. Bhaisara, Charusheela S. Korday, Minal R. Wade, Vikram V. Yaragatti, Anjali M. Bhatawdekar, Anupama V. Mauskar

Abstract


Intrauterine thrombosis with extremity ischemia presenting at birth in a newborn is a rare event. A 29 year old mother, 2nd gravida with one first trimester spontaneous abortion delivered a 33week gestation male preterm baby. On Examination, the entire left upper limb was ischemic and edematous with an absent flow on Doppler USG. Low molecular weight heparin (LMWH) was started after which gradually the limb turned pink with good volume pulsations. Thrombophilia mutation studies revealed the heterozygous state for the MTHFR (C677T) mutation only in the mother. Prompt diagnosis and early treatment has a favourable outcome in cases of intra-uterine thrombo-embolism.


Keywords


Hypercaogulable state, Intrauterine thrombosis, Low molecular weight heparin, Methylenetetrahydrofolate reductase, Neonate, Upper limb ischemia

Full Text:

PDF

References


Armstrong AP, Page RE. Intrauterine vascular deficiency of the upper limb. J Hand Surg. 1997;22(5):607-11.

Letts M, Blastorah B, Al-Azzam S. Neonatal gangrene of the extremities. J Pediatr Orthopaed. 1997 May 1;17(3):397-401.

Arshad A, McCarthy MJ. Management of limb ischaemia in the neonate and infant. European J Vasc Endovasc Surg. 2009 Jul 1;38(1):61-5.

Johnson D, Rosen JM, Khoury M, Stevenson D. Infarction of the upper limbs associated with oligohydramnios and intrauterine compression. J Hand Surg. 1988 May 1;13(3):408-10.

Nagai MK, Littleton AG, Gabos PG. Intrauterine gangrene of the lower extremity in the newborn: a report of two cases. J Pediatr Orthopaed. 2007 Jul 1;27(5):499-503.

Broadbent RS. Recipient twin limb ischemia with postnatal onset. J Pediatr. 2007 Feb 1;150(2):207-9.

Nowak-Göttl U, Duering C, Kempf-Bielack B, Sträter R. Thromboembolic diseases in neonates and children. Pathophysiology haemostasis and thrombosis. 2003;33(5-6):269-74.

Khriesat WM, Al-Rimawi HS, Lataifeh IM, Al-Sweedan S, Baqain E. Intrauterine upper limb ischemia associated with fetal thrombophilia: a case report and review of the literature. Acta Haematologica. 2010;124(1):1-4.

Jones DB, Lourie GM, Peljovich AE. Intrauterine vascular deficiency secondary to methylenetetrahydrofolate reductase deficiency: 2 case reports. American J Orthoped (Belle Mead, NJ). 2006 Apr;35(4):183-5.

Alioglu B, Ozyurek E, Tarcan A, Atac FB, Gurakan B, Ozbek N. Heterozygous methylenetetrahydrofolate reductase 677C-T gene mutation with mild hyperhomocysteinemia associated with intrauterine iliofemoral artery thrombosis. Blood Coagulation Fibrinolysis. 2006 Sep 1;17(6):495-8.

Carr MM, Al-Qattan M, Clarke HM. Extremity gangrene in utero. J Hand Surgery. 1996 Oct;21(5):652-5.

Kenny D, Tsai-Goodman B. Neonatal arterial thrombus mimicking congenital heart disease. Archives of Disease in Childhood-Fetal and Neonatal Edition. 2007 Jan 1;92(1):F59-61.

Andrew M, Monagle PT, Brooker L. Thromboembolic complications during infancy and childhood. PMPH USA; 2000.

Khriesat WM, Al-Rimawi HS, Lataifeh IM, Al-Sweedan S, Baqain E. Intrauterine upper limb ischemia associated with fetal thrombophilia: a case report and review of the literature. Acta Haematolo. 2010;124(1):1-4.

Uva MS, Serraf A, Lacour-Gayet F, Bruniaux J, Sidi D, Kachaner J, et al. Aortic arch thrombosis in the neonate. Annals Thoracic Surg. 1993 Apr 1;55(4):990-2.

Ricciardelli E, Morgan RF, Lin KY. In utero brachial artery thrombosis: limb salvage with postnatal urokinase infusion. Annals Plastic Surgery. 1995 Jan;34(1):81-3.

Sharathkumar AA, LaMear N, Pipe S, Parikh S, Russell M, Simon A, et al. Management of neonatal aortic arch thrombosis with low-molecular weight heparin: a case series. J Pediatr Hematol Oncol. 2009 Jul 1;31(7):516-21.

Edstrom CS, Christensen RD. Evaluation and treatment of thrombosis in the neonatal intensive care unit. Clinics in Perinatol. 2000 Sep;27(3):623-41.

Streif W, Goebel G, Chan AK, Massicotte MP. Use of low molecular mass heparin (enoxaparin) in newborn infants: a prospective cohort study of 62 patients. Archives of Disease in Childhood-Fetal and Neonatal Edition. 2003 Sep 1;88(5):F365-70.

Michaels LA, Gurian M, Hegyi T, Drachtman RA. Low molecular weight heparin in the treatment of venous and arterial thromboses in the premature infant. Pediatr. 2004 Sep 1;114(3):703-7.

Malowany JI, Monagle P, Knoppert DC, Lee DS, Wu J, McCusker P, et al. Enoxaparin for neonatal thrombosis: a call for a higher dose for neonates. Thrombosis Research. 2008 Jan 1;122(6):826-30.

Malowany JI, Knoppert DC, Chan AK, Pepelassis D, Lee DS. Enoxaparin use in the neonatal intensive care unit: experience over 8 years. Pharmacotherapy: J Human Pharmacol Drug Therapy. 2007 Sep;27(9):1263-71.

Nagai MK, Littleton AG, Gabos PG. Intrauterine gangrene of the lower extremity in the newborn: a report of two cases. J Pediatr Orthopaed. 2007 Jul 1;27(5):499-503.

Young G, Albisetti M, Bonduel M, Brandao L, Chan A, Friedrichs F, Goldenberg NA, et al. Impact of inherited thrombophilia on venous thromboembolism in children: a systematic review and meta-analysis of observational studies. Circulation. 2008 Sep 8;118(13):1373-82.