Table of Contents  
CASE REPORT
Year : 2013  |  Volume : 23  |  Issue : 1  |  Page : 24-25

Congenital gangrene of the right forearm and hand in a neonate of a diabetic mother


Department of Surgery, Nnamdi Azikiwe University Teaching Hospital, Nnewi, Anambra State, Nigeria

Date of Acceptance24-Sep-2013
Date of Web Publication14-Feb-2014

Correspondence Address:
Chima C Ihegihu
Department of Surgery, Nnamdi Azikiwe University Teaching Hospital, P. O. Box 2333, Nnewi, Anambra State
Nigeria
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/1116-5898.127110

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  Abstract 

This is a case report of a day-old 5.6 kg female baby delivered by cesarean section of a known diabetic and hypertensive mother who presented with gangrene of the right forearm and hand. The diagnosis of congenital gangrene in this neonate was purely clinical and maternal diabetes associated with hypertension an etiological possibility.

Keywords: Congenital, diabetic, gangrene, mother, neonate


How to cite this article:
Ihegihu CC. Congenital gangrene of the right forearm and hand in a neonate of a diabetic mother. Niger J Surg Sci 2013;23:24-5

How to cite this URL:
Ihegihu CC. Congenital gangrene of the right forearm and hand in a neonate of a diabetic mother. Niger J Surg Sci [serial online] 2013 [cited 2020 Oct 31];23:24-5. Available from: https://www.njssjournal.org/text.asp?2013/23/1/24/127110


  Introduction Top


Congenital neonatal gangrene is an uncommon disaster caused by an intra-uterine vascular catastrophe. Less than 100 cases have been reported in the literature. [1] Etiology is mostly unknown, [2] but in poorly controlled diabetic mothers, altered hemosthetic mechanisms may be responsible. [3] The increased tendency of infants of diabetic mothers to form venous and arterial [3] thrombi has been firmly established in the literature even though the reason for this has not been fully elucidated. Other known causes of congenital gangrene are compression by encircling umbilical cord, [2] oligohydramnios, [4] intrauterine trauma, [5] congenital constriction band, [6] direct pressure from maternal pelvis [7] and compound presentation. [8] Pregnancy hypertension was also associated in some previously described cases. [9]


  Case Report Top


A day-old female baby was referred to the orthopedic unit on account of discoloration and blistering of the right forearm and hand noticed at birth. The baby weighed 5.6 kg, delivered by cesarean section to a 38-year-old known diabetic woman who was also hypertensive. She was gravida 4, para 1 + 2 (alive 1). At presentation, her blood pressure was 190/130 mmHg, fasting blood sugar was 6.1 mmol/l, packed cell volume was 0.20, retroviral screening was negative and her urine analysis was normal.

At delivery, the infant was moderately asphyxiated, macrosomic, pink all over except for the right forearm and hand which were discolored with blisters. Apgar scores were 5 and 7 at 1 and 5 min respectively.

On examination at presentation, the right forearm and hand were bluish black in color, cold to touch with excoriation of the skin. The forearm and the hand was flail, insensate and reflexes were absent. There was no active movement of the fingers and capillary refill was absent. Both radial and brachial pulses were absent [Figure 1]. Nothing abnormal was detected in the other systems.
Figure 1: Congenital gangrene of the right fore-arm and hand in a neonate

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A diagnosis of congenital gangrene of the right forearm and the hand was made. The parents of the child were duly educated about the child's condition and informed that surgical above elbow amputation was the inevitable treatment to save the child's life.

The laboratory results were as follows; packed cell volume 0.55, total white cell count 12.7 × 10 9 /L (neutrophils 84%, eosinophils 16%), anisocytosis+, macrocytosis+, microcytosis+, poikilocytosis + and polychromasia++. Retroviral screening was negative and plain radiographs of the forearm and hand did not show any bone involvement. By the 3 rd day, the parents requested for discharge against medical advice and did not consent to an above elbow amputation.


  Discussion Top


Of all the etiological factors that may be responsible for the gangrene in this neonate, only diabetes in the mother can reasonably be implicated though no investigations conclusively confirmed it. Hypertension may be a risk factor. The increased incidence of venous and arterial thrombosis in infants of diabetic mothers has been well-established in the literature. The sources of embolization may be the renal vein, inferior vena cava or the placenta. [9] Due to financial constraints investigations needed for confirmation of diagnosis such as prothrombin time, activated partial thrombin time, maternal anticardiolipin antibodies associated, computerized tomography, magnetic resonance imaging, Doppler and duplex ultrasound, which could be used to detect renal vein and inferior vena cava thrombosis including level of vascular obstruction in the limb were not done. A pathologic review of the placenta could not also be carried out because it had been discarded. Demonstration of placental thrombi may have confirmed the etiology of the gangrene in this neonate. In a previous study, [9] autopsy findings demonstrated that 37.5% of fetuses with a significant placental thrombi had associated somatic thrombi. Refusal of surgery also denied us the benefit of pathologic examination of the amputated limb.


  Conclusion Top


The diagnosis of congenital gangrene in this child was purely clinical and maternal diabetes with associated hypertension an etiological possibility. More awareness needs to be created about this devastating condition and more work done to establish the etiopathogenesis. This will help to establish a treatment protocol, which may improve outcome in this condition.

 
  References Top

1.Sinclair C, Murray PM, Terkonda SP. Combined intrauterine vascular insufficiency and brachial plexus palsy: A case report. Hand (N Y) 2008;3:135-8.  Back to cited text no. 1
    
2.Turnpenny PD, Stahl S, Bowers D, Bingham P. Peripheral ischaemia and gangrene presenting at birth. Eur J Pediatr 1992;151:550-4.  Back to cited text no. 2
    
3.Van Allen MI, Jackson JC, Knopp RH, Cone R. In utero thrombosis and neonatal gangrene in an infant of a diabetic mother. Am J Med Genet 1989;33:323-7.  Back to cited text no. 3
    
4.Johnson D, Rosen JM, Khoury M, Stevenson D. Infarction of the upper limbs associated with oligohydramnios and intrauterine compression. J Hand Surg Am 1988;13:408-10.  Back to cited text no. 4
    
5.Nagai MK, Littleton AG, Gabos PG. Intrauterine gangrene of the lower extremity in the newborn: A report of two cases. J Pediatr Orthop 2007;27:499-503.  Back to cited text no. 5
    
6.Rajoo RD, Mennen U. Congenital constriction band associated with pseudarthrosis and impending gangrene. A case report. S Afr J Surg 1991;29:27-9.  Back to cited text no. 6
    
7.Hensinger RN. Gangrene of the newborn. A case report. J Bone Joint Surg Am 1975;57:121-3.  Back to cited text no. 7
    
8.Tebes CC, Mehta P, Calhoun DA, Richards DS. Congenital ischemic forearm necrosis associated with a compound presentation. J Matern Fetal Med 1999;8:231-3.  Back to cited text no. 8
    
9.Kraus FT, Acheen VI. Fetal thrombotic vasculopathy in the placenta: Cerebral thrombi and infarcts, coagulopathies, and cerebral palsy. Hum Pathol 1999;30:759-69.  Back to cited text no. 9
    


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Abstract
Introduction
Case Report
Discussion
Conclusion
References
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