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CASE REPORT
Year : 2014  |  Volume : 9  |  Issue : 3  |  Page : 276-277
 

Idiopathic brachial neuritis in a child: A case report and review of the literature


1 Department of Pediatrics, All India Institute of Medical Sciences, Bhopal, Madhya Pradesh, India
2 Department of Neurology, All India Institute of Medical Sciences, Bhopal, Madhya Pradesh, India

Date of Web Publication23-Dec-2014

Correspondence Address:
Girish Chandra Bhatt
Department of Pediatrics, All India Institute of Medical Sciences, Saket Nagar, Bhopal, Madhya Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/1817-1745.147593

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   Abstract 

Brachial neuritis is a rare disease in children, affecting mainly the lower motor neurons of the brachial plexus and/or individual nerves or nerve branches. We report a case of idiopathic brachial plexus neuritis in a 2³-year-old female child admitted with acute respiratory distress and given antibiotic therapy following which she developed weakness of the left hand. She was diagnosed as a case of idiopathic brachial plexus neuritis and was given supportive care. Although, the association with antibiotic therapy in this case could be incidental, indeed it is intriguing and requires further studies.


Keywords: Autoimmune, brachial plexus neuritis, child


How to cite this article:
Jain S, Bhatt GC, Rai N, Bhan BD. Idiopathic brachial neuritis in a child: A case report and review of the literature. J Pediatr Neurosci 2014;9:276-7

How to cite this URL:
Jain S, Bhatt GC, Rai N, Bhan BD. Idiopathic brachial neuritis in a child: A case report and review of the literature. J Pediatr Neurosci [serial online] 2014 [cited 2019 Oct 23];9:276-7. Available from: http://www.pediatricneurosciences.com/text.asp?2014/9/3/276/147593



   Introduction Top


Brachial neuritis (BN) (also known as neuralgic amyotrophy or  Parsonage-Turner syndrome More Details) is a rare disease in children, affecting mainly the lower motor neurons of the brachial plexus and/or individual nerves or nerve branches. BN exists in an inherited and an idiopathic form. The inherited form is autosomal dominant and has been linked to mutations in the SEPT9 gene on chromosome 17q. The idiopathic form is currently assumed to be autoimmune in origin. The exact etiology of idiopathic BN is not known, but upper respiratory tract infections, immunizations, trauma were antecedent event in many cases. [1],[2] To our knowledge, only one case of BN due to antibiotic is reported. Here, we present a case of brachial plexus neuritis and its association with administration of antibiotic therapy, thus implicating toward unidentified diverse etiologies of this rare disease.


   Case Report Top


A 2½-year-old female with reactive airway disease since 6 month of age was admitted in the hospital with acute onset of respiratory distress. She was treated with oxygen therapy, nebulization with salbutamol and budecort, hydrocortisone and piperacillin/tazobactam through peripheral venous catheter. She recovered well in hospital and was discharged after 3 days. On 4 th day, cefoperazone and sulbactum injection was given to her intramuscularly on left thigh. After that, parents noticed that the baby was unable to move her left hand. Before administration of antibiotic, baby's left hand was fully functional as mother remembered normal hand movements while dressing the baby. On examination, her left hand was adducted and internally rotated. Limb was hypotonic with free movements across wrist, elbow, and shoulder. Power was grade zero in all the muscle groups. Biceps, triceps, and supinator jerks were not elicited. Touch, temperature, and pain sensations were intact. The rest of the physical examination was within normal limits, and no dysmorphism was present. There was no family or previous history of a similar complaint.

Her blood counts were within normal limit. Serum IgE level was 345.58 IU/ml. Her magnetic resonance imaging (MRI) (cervical spine) showed hyperintense signal involving brachial plexus component arising from C6 to D1 nerve roots in intercostoclavicular triangle as well as interscalene triangle on left side s/o BN.

High-resolution computed tomography chest revealed spiculated subpleural parenchymal densities in right upper zone with fissural thickening in the right mid and left lower zones with patchy parenchymal haziness in both lung fields at places.

Patient was not treated with steroids initially, may be because of unawareness of doctors at periphery. She was subjected to regular physiotherapy. On 3-month follow-up, there was no improvement in her condition. Her arm was adducted with grade zero power in all the muscle groups. Reflexes could not be elicited. Further follow-up was not possible as the patient did not turnup.


   Discussion Top


Idiopathic neuralgic amyotrophy (INA) is a rare neurological disorder in children and only few cases are reported. In adults, the typical clinical course starts with acute, severe, aching, unilateral shoulder, and proximal arm pain lasting from days to a few weeks. When the pain abates, painless shoulder girdle and arm weakness develops, more prominent in the upper plexus muscles. [3] Pain is less frequent in children, and its absence by no means excludes the diagnosis. [4]

Idiopathic neuralgic amyotrophy is considered to be autoimmune in origin, [5] but its exact mechanism and etiology is unknown. Various antecedent events to BN are reported in adult patients, but only one case in association with antibiotic is published. Finstad et al. reported one case of INA in 22 years male with cystic fibrosis after vancomycin administration. [6] Al Masri et al. reported a case of tacrolimus associated BN in pediatric renal transplant recipient. [7]

Diagnostic tests such as MRI, although not confirmatory, are required to rule out other conditions. MRI typically shows increased signal intensity related to neurogenic edema on T2 images. Electrophysiological studies are not needed in typical cases, and they usually show denervation of affected and some clinically unaffected muscles. [8] Cervical spine disease, rotator cuff disease, entrapment neuropathies, and idiopathic hypertrophic BN are differentials of INA.

Treatment of INA usually involves a combination of corticosteroids, analgesics, immobilization, and physical therapy. Oral steroids have been recommended by some, but there is poor literature evidence to support its efficacy. Oral prednisone given the 1 st month after onset may shorten the duration of symptoms. [9] In a retrospective study by Naito et al., intravenous immunoglobulin and methylprednisolone pulse therapy showed improvement in 9 out of 10 patients. [10] Further studies need to be performed to establish efficacy of treatment with corticosteroids or other immune-modulating therapies. Acupuncture and transcutaneous electrical nerve stimulation are adjuncts to medications.

Different studies show contradictory results when it comes to the prognosis. A study by van Alfen et al. states that children have a less favorable recovery, but when they fully recover they do so in a shorter period. [4] However, Høst et al. reviewed the literature and identified 58 pediatric cases of INA. They found that out of 58 patients, 63% made a full recovery, 25% made a partial, and 13% made no recovery. [11] The mean recovery time was found to be 11.1 months. Thus, they indicated better prognosis in children than previously anticipated. Further studies should be conducted to chalk out the course of the disease in children.

Thus, idiopathic brachial plexus neuritis is a rare disease in children with diverse etiology. Although, the association with antibiotic therapy in this case could be incidental, indeed it is intriguing and requires further studies.

 
   References Top

1.
Kelkar P, Parry GJ. Brachial plexus disorders. Neurologic Therapeutics: Principles and Practice. London: Martin Dunitz Publishers; 2003.  Back to cited text no. 1
    
2.
van Alfen N, van Engelen BG. The clinical spectrum of neuralgic amyotrophy in 246 cases. Brain 2006;129:438-50.  Back to cited text no. 2
    
3.
Misamore GW, Lehman DE. Parsonage-Turner syndrome (acute brachial neuritis). J Bone Joint Surg Am 1996;78:1405-8.  Back to cited text no. 3
    
4.
van Alfen N, Schuuring J, van Engelen BG, Rotteveel JJ, Gabreëls FJ. Idiopathic neuralgic amyotrophy in children. A distinct phenotype compared to the adult form. Neuropediatrics 2000;31:328-32.  Back to cited text no. 4
    
5.
Suarez GA. Immune brachial plexus neuropathy. Peripheral Neuropathy. Philadelphia: Elsevier Saunders; 2005. p. 2299-308.  Back to cited text no. 5
    
6.
Finstad K, Guajardo JR, Scoville C. Neuralgic amyotrophy associated with antibiotic therapy. Ann Pharmacother 2008;42:1344-7.  Back to cited text no. 6
    
7.
Al Masri O, Fathallah W, Quader S. Recovery of tacrolimus-associated brachial neuritis after conversion to everolimus in a pediatric renal transplant recipient - Case report and review of the literature. Pediatr Transplant 2008;12:914-7.  Back to cited text no. 7
    
8.
Subramony SH. AAEE case report 14: Neuralgic amyotrophy (acute brachial neuropathy). Muscle Nerve 1988;11:39-44.  Back to cited text no. 8
    
9.
van Alfen N, van Engelen BG, Hughes RA. Treatment for idiopathic and hereditary neuralgic amyotrophy (brachial neuritis). Cochrane Database Syst Rev 2009;CD006976.  Back to cited text no. 9
    
10.
Naito KS, Fukushima K, Suzuki S, Kuwahara M, Morita H, Kusunoki S, et al. Intravenous immunoglobulin (IVIg) with methylprednisolone pulse therapy for motor impairment of neuralgic amyotrophy: Clinical observations in 10 cases. Intern Med 2012;51:1493-500.  Back to cited text no. 10
    
11.
Høst C, Skov L. Idiopathic neuralgic amyotrophy in children. Case report, 4 year follow up and review of the literature. Eur J Paediatr Neurol 2010;14:467-73.  Back to cited text no. 11
    




 

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