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 Table of Contents  
CASE REPORT
Year : 2018  |  Volume : 7  |  Issue : 12  |  Page : 75-78

Idiopathic peripheral lower-limb gangrene in infancy


1 Department of Orthopaedics and Trauma, University of Abuja Teaching Hospital, Gwagwalada, Nigeria
2 Department of Surgery, National Hospital, Abuja, Nigeria

Date of Web Publication19-Nov-2018

Correspondence Address:
T D Sough
Department of Orthopaedics and Trauma Surgery, University of Abuja Teaching Hospital, Gwagwalada, Abuja
Nigeria
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/nnjcr.nnjcr_18_18

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  Abstract 


Idiopathic peripheral lower-limb gangrene in infancy is a rare condition with attendant difficulty in making adequate or accurate diagnosis, as there are no clear-cut causes of the condition as the name suggests. The predisposing factors are important to consider as a guide for diagnosis and treatment, but timely intervention to restore the normal physiological status of the patient and reverse progression of ischemia is much more important. This is a presentation of two cases that were similarly managed for peripheral lower-limb idiopathic gangrene. Both cases were male infants. The conservative approach adopted in both cases yielded remarkable results.

Keywords: Gangrene, idiopathic, infancy, lower limb, peripheral


How to cite this article:
Sough T D, Madubueze C C. Idiopathic peripheral lower-limb gangrene in infancy. N Niger J Clin Res 2018;7:75-8

How to cite this URL:
Sough T D, Madubueze C C. Idiopathic peripheral lower-limb gangrene in infancy. N Niger J Clin Res [serial online] 2018 [cited 2020 Oct 19];7:75-8. Available from: https://www.mdcan-uath.org/text.asp?2018/7/12/75/245785




  Introduction Top


Peripheral idiopathic lower-limb gangrene is rare and, as the name suggests, the causes are very difficult to pinpoint.[1],[2] Both prenatal and postnatal predisposing factors should be carefully evaluated during the clinical analysis and appropriate investigations should be carried out to determine specific causes. In the available literature, maternal diabetes and premature rupture of the membranes producing a “dry” labor have been cited as the predisposing causes.[2] And more recently, arterial thrombosis and embolism have been seen, most frequently among infants cared for in an intensive care nursery where venous puncture, arterial puncture, angiography, and indwelling catheters are commonly used.[2],[3],[4],[5] Treatment should be commenced as soon as possible and in our case, supportive treatment was enough to stop progression of the condition.[4],[6],[7]


  Case Reports Top


Case 1

A 6-week-old male infant from the suburb of Abuja in the Federal Capital Territory-Abuja, Nigeria, was presented with a 5-day history of gradual swelling and bluish discoloration of both feet. He had a history of treatment for a febrile illness characterized by high-grade fever, vomiting, diarrhea, cough, and respiratory difficulty, at a private health facility a couple of weeks prior to presentation to us. The mode of treatment at the health center was said to be mainly with oral administration of medications. There was no history of intravenous or intramuscular injections, and he was said to have had improvement in general well-being before the onset of the present symptoms.

There was no history of trauma (burns) or convulsions. Traditional medications were not administered. There was no history suggestive of child abuse. The mother is not diabetic. The history of pregnancy was uneventful. The baby was delivered at home by a traditional birth attendant. The cord was severed with a razor blade and tied with a weaving thread. There was no history suggestive of asphyxia. Postnatal days were said to be uneventful before the onset of the febrile illness.

Examination revealed an active but febrile (38.8°C) baby with bilateral gangrene of both forefeet: 2/3rd of the distal right foot, while the left up to the one-half of the distal foot [Figure 1] and [Figure 2]. The cardiovascular and respiratory systems appeared to be within clinically acceptable physiologic limits. There were no lesions in the upper limbs. Peripheral pulses were felt down to the ankle joints. There were also no abnormal findings in the abdomen and chest. Investigations revealed the following: HIV I and II negative, hemoglobin of 12.7%, white blood cell (WBC) count of 13,800, platelets of 272,000/mL, and erythrocyte sedimentation rate of 153/h, echocardiogram was normal, and blood culture yielded no growth.
Figure 1: (a) Gangrene of the feet, Case 1. (b) The anterior side of the feet on the 1st day of admission

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Figure 2: Systemic symptoms subsided as gangrenous toes gradually becoming dry

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Intravenous fluids, intravenous antibiotics, and vitamin supplements were given. The response from hydration and systemic antibiotics was good, as vital signs returned to physiological levels in 72 h. Elevation of both limbs was done with dry dressing of gangrenous areas. The overall response was encouraging, as the gangrenous portions of the feet gradually dried up with demarcation from the normal tissues. At this time, systemic symptoms had subsided and the patient was clinically stable by the 2nd week on admission. Attempts to amputate the gangrenous portions of the feet were not supported by the mother who refused signing consent. Conservative approach was continued with elevation and watchful waiting, with bedside debridement resulting in spontaneous amputation (falling off of the dry toes and bedside removal of the phalanges involved) [Figure 3] and healing of the stump, 6–8 weeks after admission. The patient was discharged home in satisfactory clinical state. He was seen at the 1st follow-up clinic 3 weeks postdischarge with no problem. He was however lost to follow-up subsequently.
Figure 3: (a) Healing of the feet at 6–8 weeks on admission (b) The feet healing well

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Case 2

A 4-week-old, full-term male infant, from the neighboring state of Nasarawa, Nigeria, was admitted by the pediatricians with febrile symptoms in the neonatal unit of the teaching hospital. While he was on admission, he was noticed to have gradual swelling and change in color of the right foot without obvious cause. The discoloration developed into gangrene of the forefoot.

He was a second child of a 27-year-old mother whose pregnancy was not supervised and delivery was conducted by the traditional birth attendance. The patient was being fed with breastmilk and was said to be gaining weight steadily. There was no history of trauma. Traditional medications were not administered. The mother is not diabetic. There was no history suggestive of asphyxiation at birth.

On examination, he was febrile (38.7°C). There was a bluish discoloration of the right foot, with gangrene of all the toes advancing to the forefoot [Figure 4]. The overall cardiorespiratory systems appeared stable. Investigations revealed the following: HIV I and II negative, hemoglobin of 13.2%, WBC count of 14,100, platelets of 472,000/mL, and blood culture yielded no growth.
Figure 4: Case 2 with right unilateral foot gangrene

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He was managed on intravenous fluids, intravenous antibiotics, limb elevation, and watchful waiting for gangrenous portion to demarcate so that amputation could be done. This mode of treatment was also adequate and stopped progression of the condition. The gangrene finally demarcated and phalanges removed and foot healed well. He was discharged after 5 weeks on admission. Follow-up of this patient has been a challenge. He was lost to follow-up since then.


  Discussion Top


Peripheral gangrene has been described in literature by many authors over the years. The possible causes or predisposing factors and associated factors have also been elucidated by various authors. A number of cases reported in the literature identified some predisposing factors such as sepsis, trauma, congenital heart disease and vascular occlusion, and maternal diabetes, among others.[3],[4],[6],[8],[9],[10] Peripheral gangrene associated with renal vein thrombosis in infancy has also been reported.[11] Collagen diseases, such as polyarteritis nodosa as well as rheumatoid arthritis, is reported as being implicated.[12],[13] Apart from the report of association of these pathologic conditions with peripheral extremity gangrene, none of them has been established as the cause of the condition. Some observed a high mortality rate in those patients with polyarteritis nodosa.[13],[14] The report of idiopathic peripheral limb gangrene (IPLG) in a Nigerian adolescent male by Adogu and Abengowe[13] showed association with a fulminating gangrene of the extremities following a febrile diarrheal illness with associated abdominal pains, polyarthropathy, and altered hemostatic function, suggesting autoimmune disease.

The only obvious identifiable cause in our two patients was the febrile illness both were treated of, even though detailed investigations could not be achieved to rule out other associated causes. The first patient had symptoms of diarrhea and one may be tempted to admit that dehydration could predispose to peripheral gangrene of the feet,[13] but he was not brought in a dehydrated condition as such. Clinically, the predisposing factor was identified to be sepsis. Traditional medications and harmful traditional practices such as putting the feet of a convulsing child in fire in an attempt to stop the convulsion have been presented in the burns unit severally, but being trauma, these most often resolve without total gangrene of the feet. The history of convulsion must be elicited in patients presenting with feet gangrene and whether there were attempts at these practices. That was not the case in our patients.

Management of these patients followed the same pattern and was largely expectant; since at presentation, there was established gangrene of both forefeet in the first case and evolving gangrene of the right foot in the second case in a similar fashion. Both of them had history of febrile illness prior to onset of foot-gangrene. The affected lower limbs in both patients were elevated, intravenous fluids were administered, and parenteral antibiotics were also given to control infection, while keeping a watch at the vital signs to monitor progression. A remarkable improvement observed in the general condition of the patients became the driving force to keep treatment conservative. Systemic symptoms such as fever were noticed to have reduced significantly, within 72 h of intravenous administration of antibiotics in the first case and the second case had a much more stable course as he was already on medications when we saw him. The vital signs returned to near-normal physiological levels within a short time, giving an indication that systemic response was well controlled. Expectedly, the gangrenous portions dried up and subsequently spontaneous amputation from the line of demarcation occurred with satisfactory healing of the stump.[4],[8] Treatment until the 1960s was primarily supportive. It consisted of adequate hydration, close clinical monitoring, and measures to prevent sepsis and further trauma to the extremity. Other measures, such as warming of the extremity, anticoagulation, vasodilators, hyperbaric oxygen, and lumbar sympathetic blocks, have all been tried with varying results.[13],[15],[16] It is however not necessary to subject the patient to procedures that are technically demanding, if there is clinical improvement on the supportive therapy. We believe that once there is no further ischemia, the demarcation zone appears and, whether surgery is rushed or not, amputation is the final sequel.


  Conclusion Top


A conservative and expectant approach to management may be of benefit in many of these cases.

Declaration of patient consent

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

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Noyez JF, Sinzobahamvya N, Kalangu K. Peripheral gangrene in African children: A clinical report of twelve cases. Acta Orthop Belg 1996;62:207-11.  Back to cited text no. 1
    
2.
Kothari PR, Gupta A, Kulkarni B. Neonatal lower extremity gangrene. Indian Paediatr 2005;42:1156-8.  Back to cited text no. 2
    
3.
Gross RE. Arterial embolism and thrombosis in infancy. Am J Dis Children 1945;70:61-73.  Back to cited text no. 3
    
4.
Lloyd A, Kemball M, Fraser G. Peripheral gangrene in infancy and childhood. Br Med J 1967;1:468-70.  Back to cited text no. 4
    
5.
Wiseman NE, Briggs JN, Bolton VS. Neonatal arterial occlusion with ischemic limb gangrene. J Pediatr Surg 1977;12:707-10.  Back to cited text no. 5
    
6.
Okoko BJ, Ota MO, Arowolo JO, Whittle HC. Peripheral gangrene complicating Salmonella typhi septicaemia in a Gambian infant. J Trop Pediatr 2001;47:250-1.  Back to cited text no. 6
    
7.
Heller H, Alvari G. Gangrene of the extremities in the newborn. Am J Dis Children 1941;62:133-40.  Back to cited text no. 7
    
8.
Farrar MJ, Bennet GC, Wilson NI, Azmy A. The orthopaedic implications of peripheral limb ischaemia in infants and children. J Bone Joint Surg Br 1996;78:930-3.  Back to cited text no. 8
    
9.
Manios SG, Kanokoudi F, Miliaras-Vlachakis M. Gangrene of lower extremities in a newborn infant associated with intravascular coagulation (recession of gangrene after heparin therapy). Helv Paediatr Acta 1972;27:187-92.  Back to cited text no. 9
    
10.
Turpie AG, Forbes CD, McNicol GP. Idiopathic gangrene in African children. Br Med J 1967;3:646-8.  Back to cited text no. 10
    
11.
Valderrama E, Gribetz I, Strauss L. Peripheral gangrene in a newborn infant associated with renal and adrenal vein thrombosis. Report of a case in an offspring of a diabetic mother. J Pediatr 1972;80:101-3.  Back to cited text no. 11
    
12.
Cummings JK, Taleisnik J. Peripheral gangrene as a complication of rheumatoid arthritis. Report of a case and review of the literature. J Bone Joint Surg Am 1971;53:1001-6.  Back to cited text no. 12
    
13.
Adogu AA, Abengove CU. Idiopathic peripheral gangrene in Nigeria. J Natl Med Assoc 1993;85:560-2.  Back to cited text no. 13
    
14.
Roberts FB, Fetterman GH. Polyarteritis nodosa in infancy. J Pediatr 1963;63:519-29.  Back to cited text no. 14
    
15.
Onalo R, Ogala WN, Lawal YZ, Chom ND, Odogu O, Ige SO, et al. Congenital gangrene of the extremities in a newborn. Niger J Clin Pract 2011;14:245-8.  Back to cited text no. 15
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16.
Quddusi AI, Nizami N, Razzaq A, Husain S. Bilateral gangrene of lower limbs in a neonate. J Coll Physicians Surg Pak 2014;24 Suppl 2:S119-20.  Back to cited text no. 16
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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