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Unilateral upper extremity ischemia in a neonate – A rare complication of prematurity Agrawal AC, Iyer R D, Sakale HS, Garg AK

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 Table of Contents
 
CASE REPORT



Year : 2022  |  Volume
: 14 
Issue : 1  |  Page : 105-107

Unilateral upper extremity ischemia in a neonate – A rare complication of prematurity

Alok Chandra Agrawal, R Dinesh Iyer, Harshal Suhas Sakale, Ankit Kumar Garg

Department of Orthopaedics, All India Institute of Medical Sciences, Raipur, Chhattisgarh, India


Date of Submission 17-Mar-2022
Date of Acceptance 07-Apr-2022
Date of Web Publication 15-Jun-2022





Correspondence Address:
Dr. R Dinesh Iyer
Flat No. C-003, Definer Kingdom Apartments, Near Budhigere Cross, Mandur Panchayat, Bengaluru – 560 049, Karnataka
India
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Source of Support: None, Conflict of Interest: None

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DOI: 10.4103/jotr.jotr_29_22

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Acute limb ischemia in the neonatal period is a rarely reported complication of prematurity and needs a high index of suspicion for early diagnosis and a multidisciplinary approach for its management. Here, we present a case of a preterm baby that developed unilateral upper limb ischemia in the neonatal period and discussed the problems faced in the management of such cases. A 24-day-old male baby was referred to the orthopedics department because of bluish-black discoloration of the right hand and forearm and no active movement in the wrist and hand. The baby was preterm, delivered at 29 weeks of gestation lower-segment cesarean section with a birth weight of 900 g to a healthy mother with no preexisting illness. Although the line of demarcation was just below the elbow joint and conventional wisdom would dictate us to go for a transhumeral amputation, we opted for a below elbow amputation in a bid to save the elbow joint as we could have revised the amputation at a later date if needed. Neonatal acute limb ischemia has been rarely reported and needs a high index of suspicion. Preterm and low birth weight babies are more prone to it. Treatment of such patients depends on the cause of gangrene. Amputation at such young age is psychologically disturbing for the parents. However, it is usually associated with good functional outcomes as the child has not yet learned the use of a limb or developed cortical plasticity in the brain. All attempts should be made to preserve as much joint and physis as possible to have a functional joint with better prosthetic fitting.

Keywords: Amputation, limb ischemia, neonatal gangrene, prematurity

How to cite this article:
Agrawal AC, Iyer R D, Sakale HS, Garg AK. Unilateral upper extremity ischemia in a neonate – A rare complication of prematurity. J Orthop Traumatol Rehabil 2022;14:105-7



How to cite this URL:
Agrawal AC, Iyer R D, Sakale HS, Garg AK. Unilateral upper extremity ischemia in a neonate – A rare complication of prematurity. J Orthop Traumatol Rehabil [serial online] 2022 [cited 2022 Jul 2];14:105-7. Available from: https://www.jotr.in/text.asp?2022/14/1/105/347362

  Introduction   Top

Limb ischemia in a newborn is a rare entity with devastating outcomes. In the majority of cases, an exact etiology is not known and is still classified as idiopathic. Some of the common causes are antenatal compression of the vessel due to intrauterine positioning, vasospasm due to traumatic delivery, and coagulopathy secondary to prematurity and sepsis.[1],[2] Most of the literature related to the disease is in case reports or small series because of the few and sporadic cases seen, and no studies focus on its management.[3],[4],[5],[6] Detecting early ischemic changes in a neonate is a challenge in itself, and its management requires a multidisciplinary approach involving the neonatologist, orthopedic surgeon, and vascular surgeons.

  Case Report   Top

A 24-day-old male baby was referred to the orthopedics department because of bluish-black discoloration of the right hand and forearm and no active movement in the wrist and hand. The baby was preterm, delivered at 29 weeks of gestation by lower-segment cesarean section with a birth weight of 900 g to a healthy mother with no preexisting illness. After birth, the baby developed respiratory distress and was intubated on day 2. The baby was managed in the neonatal intensive care unit with intravenous fluids and antibiotics. Peripheral blood examination revealed the baby had associated anemia and thrombocytopenia and was diagnosed with neonatal sepsis with respiratory distress. On the 5th day, he was extubated and put on continuous positive airway pressure and gradually weaned off oxygen support. The baby was not accepting oral feeds, and the orogastric tube was used for the same. Ophthalmic examination revealed that the baby had retinopathy of prematurity (ROP) (Stage 2).

However, on the 23rd day, the attending pediatrician noted a sluggish movement of the right hand with bluish-black discoloration of the fingers. On the next day, it was noted that the discoloration started extending up to the hand with the complete absence of finger movements. At this stage, an orthopedics’ opinion was sought. On examination, the right upper limb was cold below the elbow, radial and ulnar pulsations were not palpable, with no movements of fingers or wrist. Capillary refill was absent. On pinprick, there was sluggish bleeding and no reflex withdrawal to nociceptive stimulus. Low-molecular-weight heparin was started, and the limb was kept warm. The parents were counseled regarding the poor prognosis of the limb and the need for amputation. However, since the gangrenous changes were still progressive, we waited for the line of demarcation to form. On day 28, when the discoloration did not progress any further and the line of demarcation formed just below the elbow joint, we planned an amputation [Figure 1]. Although the line of demarcation was just below the elbow joint and conventional wisdom would dictate us to go for a transhumeral amputation, we opted for a below elbow amputation in a bid to save the elbow joint as we could have revised the amputation at a later date if needed.

Figure 1: (a) Clinical picture of the baby showing ischemic changes of the right upper limb extending up to the elbow joint. (b and c) Intraoperative pictures showing the level of amputation. (d) The clinical picture of the amputated limb

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The postoperative recovery of the baby was uneventful. He eventually underwent bilateral laser photocoagulation for ROP. The child is now 2 years old, weighing 8 kg and height of 83 cm, is active and playful with no other complaints. On examination, the stump below the elbow joint is about 5 cm in length. However, there is a weakness of the triceps due to the smaller lever arm, which is expected to improve as the child grows and the length of the stump increases [Figure 2] and [Figure 3].

Figure 2: Two-year follow-up radiographs of both elbows for comparison showing growth of the distal humeral and proximal ulna stump

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  Discussion   Top

Limb ischemia in preterm babies is gradually being recognized as an important part of the “prematurity syndrome” which involves almost all the systems. Due to the paucity of data, we do not know the incidence of the disease. Although all cases do not have a specific underlying cause explaining the occurrence of limb ischemia, there are three-leading causes of the ischemia that must be differentiated. The first one is due to compression, which is because of intrauterine position of the fetus or entanglement of the umbilical cord around the limb. In such cases, ischemic signs can appear soon after birth[7],[8] and frequently results in auto amputation of the limb. The next cause is due to vasospasm, which may occur especially during a traumatic delivery. Usually, the spasm resolves, but if the child is not resuscitated or develops hypothermia, the vasospasm may be prolonged enough to cause permanent damage to the limb. The third cause is postnatal due to coagulopathy, sepsis, preterm birth, and low birth weight. The risk is much higher in preterm and low birth weight babies due to the immature hemostasis system.

Iatrogenic thrombosis of vessels causing ischemia is one of the most common causes but underreported. It has been reported that such thrombotic events can be caused by seemingly regular procedures such as peripheral arterial cannulation or drawing arterial samples,[9],[10] which are usually inevitable during in-hospital care of a preterm baby. The use of prophylactic anticoagulants may warrant consideration in such patients to prevent such untoward incidences.

Dilemmas faced during the management of such cases include the timing of surgery (early vs. delayed), type of surgery (debridement and limb salvage vs. amputation), and level of amputation if done. Based on our experience and the existing literature, the cause of ischemia gives a direction to the management. If it is due to sepsis and the limb has developed wet gangrene, it is wise to operate early to reduce the chances of systemic complications. However, in an ischemic limb, surgery can be delayed until a line of demarcation is formed, as sometimes, due to vascular regeneration and formation of collaterals, the demarcation line is more distal to what can be anticipated based on Doppler or angiography studies. In children, it should always be tried to preserve the joint and epiphysis so that the stump can grow with age, which gives both a psychological advantage to the child and better prosthetic fitting. In our patient, we preserved the elbow joint even though the demarcation level was reaching up to the joint. This surgery has given the child a working elbow joint and a stump for a prosthesis.

  Conclusion   Top

Neonatal acute limb ischemia has been rarely reported and needs a high index of suspicion. Preterm and low birth weight babies are more prone to it. Treatment of the patient depends on the cause of gangrene. Amputation at such young age is psychologically disturbing for the parents. However, it is usually associated with better outcomes than older children, as the child has not yet learned the use of a limb or developed cortical plasticity in the brain. Attempts should be made to preserve as much of the joint and the physis as possible so that the child has a functional joint with better prosthetic fitting.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient’s parents have given their consent for his images and other clinical information to be reported in the journal. The patient’s parents understand that name and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 

  References   Top


1.
Bonny-Obro R, Aké YL, Midekor-Gonebo K, Kouassi-Dria AK, Ouattara SJ, Sounkere-Soro M. et al. Neonatal acute limb ischemia, Journal of Pediatric Surgery Case Reports, 2021;66:10.1016/j.epsc.2021.101784.  Back to cited text no. 1
    
2.
Kothari PR, Gupta A, Kulkarni B. Neonatal lower extremity gangrene. Indian Pediatr 2005;42:1156-8.  Back to cited text no. 2
    
3.
Cerbu S, Bîrsăşteanu F, Heredea ER, Iacob D, Iacob ER, Stănciulescu MC, et al. Acute limb ischemia in neonates: Etiology and morphological findings – Short literature review. Rom J Morphol Embryol 2018;59:1041-4.  Back to cited text no. 3
    
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De Carolis MP, Bersani I, Piersigilli F, Rubortone SA, Occhipinti F, Lacerenza S, et al. Peripheral nerve blockade and neonatal limb ischemia: Our experience and literature review. Clin Appl Thromb Hemost 2014;20:55-60.  Back to cited text no. 4
    
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Abdelrazeq S, Alkhateeb A, Saleh H, Alhasan H, Khammash H. Intrauterine upper limb ischemia: An unusual presentation of fetal thrombophilia – A case report and review of the literature. Case Rep Pediatr 2013;2013:670258.  Back to cited text no. 5
    
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Salawu ON. Neonatal lower limb gangrene: Avoidable causes noticed in North-West Nigeria. J Orthop Traumatol Rehabil 2017;9:53-5.  Back to cited text no. 7
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8.
Arshad A, McCarthy MJ. Management of limb ischaemia in the neonate and infant. Eur J Vasc Endovasc Surg 2009;38:61-5.  Back to cited text no. 8
    
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Sadat U, Hayes PD, Varty K. Acute limb ischemia in pediatric population secondary to peripheral vascular cannulation: Literature review and recommendations. Vasc Endovascular Surg 2015;49:142-7.  Back to cited text no. 9
    
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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. 10
    

 
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