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Parsonage-Turner syndrome following post-exposure prophylaxis.

Fransz DP, Schönhuth CP, Postma TJ, van Royen BJ - BMC Musculoskelet Disord (2014)

Bottom Line: No currently available tests can provide a definite confirmation or exclusion of PTS.Supplementary administration of oral prednisolone could shorten the duration of pain.Although the outcome is typically preferable, a substantial amount of patients are left with some residual paresis and functional impairment.

View Article: PubMed Central - HTML - PubMed

Affiliation: Department of Orthopaedic Surgery, VU University Medical Center, PO Box 7057, 1007 MB Amsterdam, The Netherlands. c.schonhuth@vumc.nl.

ABSTRACT

Background: The 'Parsonage-Turner syndrome' (PTS) is a rare but distinct disorder with an abrupt onset of shoulder pain, followed by weakness and atrophy of the upper extremity musculature, and a slow recovery requiring months to years. To our best knowledge, this is the first case describing symptoms and signs of PTS following the administration of a post-exposure prophylaxis (PEP) regimen against possible human immunodeficiency virus (HIV) and hepatitis B virus (HBV) infection.

Case presentation: A 25-year-old Caucasian man presented with pain and unilateral scapular winging following PEP against possible HIV and HBV infection. Although atrophy and weakness were observed for the right supraspinatus muscle, a full range of motion was achievable. Neurological examination, plain radiography of the right shoulder and electromyography showed no additional abnormalities. The patient was diagnosed with post-vaccination PTS and treated non-operatively. During the following 15 months the scapular winging receded and full muscle strength was regained.

Conclusion: Parsonage-Turner syndrome is a rare clinical diagnosis. The precise pathophysiological mechanism of PTS remains unclear, but it seems to involve an interaction between genetic predisposition, mechanical vulnerability and an autoimmune trigger. An immunological event, such as - in this case - a vaccination as part of PEP treatment, can trigger the onset of PTS. The clinical presentation is distinctive with acute severe pain followed by patchy paresis, atrophy and sensory symptoms that persist for months to years. No currently available tests can provide a definite confirmation or exclusion of PTS. Routine blood examination, electromyography (EMG), and computed tomography (CT) or magnetic resonance imaging (MRI) serve mainly to exclude other disorders. The recovery can be quite lengthy, non-operative treatment is the accepted practice. Supplementary administration of oral prednisolone could shorten the duration of pain. Although the outcome is typically preferable, a substantial amount of patients are left with some residual paresis and functional impairment.

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Clinical presentation after 15 months. A, B &C. Complete recovery of scapular winging and no residual atrophy. Photographs were taken fifteen months after initial presentation.
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Figure 3: Clinical presentation after 15 months. A, B &C. Complete recovery of scapular winging and no residual atrophy. Photographs were taken fifteen months after initial presentation.

Mentions: A 25-year-old Caucasian man was referred to our outpatient clinic, two months after initial presentation at the Emergency Room because of accidental exposure to bloodborne pathogens. In accordance with the Dutch post-exposure prophylaxis guidelines, post-exposure prophylaxis (PEP) was indicated against possible infection with human immunodeficiency virus (HIV) and hepatitis B virus (HBV)[9]. The patient immediately received 500 IU of human hepatitis B immunoglobulin intramuscularly into the right shoulder, and hepatitis B vaccination according to the vaccine dose schedule, at 0, 1 and 6 months. Additionally, the patient received 4 weeks of atazanavir 400 mg with combivir 450 mg (lamivudine/zidovudine) twice daily. Flu-like symptoms occurred in the first week, which subsequently diminished. Following the second intramuscular vaccine dose, the patient complained of neck pain, with radiating pain towards his right shoulder. The patient noticed a deviated position of his right scapula. During the next weeks the pain gradually subsided, but the scapular deviation persisted. No subjective signs of muscle weakness or sensory symptoms were present.On physical examination, the patient had evident scapular winging (Figures 1A-C). Atrophy and weakness were observed for the right supraspinatus muscle. However, a full range of motion was achievable. Thorough neurological examination did not indicate any further deficits. Plain radiography of the right shoulder showed no abnormalities. Electromyography (EMG) recorded two months after initial presentation, showed no neuromuscular abnormalities of the long thoracic or suprascapular nerve.The clinical diagnosis ‘Parsonage-Turner syndrome’ was made. The patient received non-operative treatment. The scapular winging receded during the following 15 months (Figures 2A-C and3A-C). Full muscle strength was regained. The HIV and HBV serology proved to be negative.


Parsonage-Turner syndrome following post-exposure prophylaxis.

Fransz DP, Schönhuth CP, Postma TJ, van Royen BJ - BMC Musculoskelet Disord (2014)

Clinical presentation after 15 months. A, B &C. Complete recovery of scapular winging and no residual atrophy. Photographs were taken fifteen months after initial presentation.
© Copyright Policy - open-access
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC4126347&req=5

Figure 3: Clinical presentation after 15 months. A, B &C. Complete recovery of scapular winging and no residual atrophy. Photographs were taken fifteen months after initial presentation.
Mentions: A 25-year-old Caucasian man was referred to our outpatient clinic, two months after initial presentation at the Emergency Room because of accidental exposure to bloodborne pathogens. In accordance with the Dutch post-exposure prophylaxis guidelines, post-exposure prophylaxis (PEP) was indicated against possible infection with human immunodeficiency virus (HIV) and hepatitis B virus (HBV)[9]. The patient immediately received 500 IU of human hepatitis B immunoglobulin intramuscularly into the right shoulder, and hepatitis B vaccination according to the vaccine dose schedule, at 0, 1 and 6 months. Additionally, the patient received 4 weeks of atazanavir 400 mg with combivir 450 mg (lamivudine/zidovudine) twice daily. Flu-like symptoms occurred in the first week, which subsequently diminished. Following the second intramuscular vaccine dose, the patient complained of neck pain, with radiating pain towards his right shoulder. The patient noticed a deviated position of his right scapula. During the next weeks the pain gradually subsided, but the scapular deviation persisted. No subjective signs of muscle weakness or sensory symptoms were present.On physical examination, the patient had evident scapular winging (Figures 1A-C). Atrophy and weakness were observed for the right supraspinatus muscle. However, a full range of motion was achievable. Thorough neurological examination did not indicate any further deficits. Plain radiography of the right shoulder showed no abnormalities. Electromyography (EMG) recorded two months after initial presentation, showed no neuromuscular abnormalities of the long thoracic or suprascapular nerve.The clinical diagnosis ‘Parsonage-Turner syndrome’ was made. The patient received non-operative treatment. The scapular winging receded during the following 15 months (Figures 2A-C and3A-C). Full muscle strength was regained. The HIV and HBV serology proved to be negative.

Bottom Line: No currently available tests can provide a definite confirmation or exclusion of PTS.Supplementary administration of oral prednisolone could shorten the duration of pain.Although the outcome is typically preferable, a substantial amount of patients are left with some residual paresis and functional impairment.

View Article: PubMed Central - HTML - PubMed

Affiliation: Department of Orthopaedic Surgery, VU University Medical Center, PO Box 7057, 1007 MB Amsterdam, The Netherlands. c.schonhuth@vumc.nl.

ABSTRACT

Background: The 'Parsonage-Turner syndrome' (PTS) is a rare but distinct disorder with an abrupt onset of shoulder pain, followed by weakness and atrophy of the upper extremity musculature, and a slow recovery requiring months to years. To our best knowledge, this is the first case describing symptoms and signs of PTS following the administration of a post-exposure prophylaxis (PEP) regimen against possible human immunodeficiency virus (HIV) and hepatitis B virus (HBV) infection.

Case presentation: A 25-year-old Caucasian man presented with pain and unilateral scapular winging following PEP against possible HIV and HBV infection. Although atrophy and weakness were observed for the right supraspinatus muscle, a full range of motion was achievable. Neurological examination, plain radiography of the right shoulder and electromyography showed no additional abnormalities. The patient was diagnosed with post-vaccination PTS and treated non-operatively. During the following 15 months the scapular winging receded and full muscle strength was regained.

Conclusion: Parsonage-Turner syndrome is a rare clinical diagnosis. The precise pathophysiological mechanism of PTS remains unclear, but it seems to involve an interaction between genetic predisposition, mechanical vulnerability and an autoimmune trigger. An immunological event, such as - in this case - a vaccination as part of PEP treatment, can trigger the onset of PTS. The clinical presentation is distinctive with acute severe pain followed by patchy paresis, atrophy and sensory symptoms that persist for months to years. No currently available tests can provide a definite confirmation or exclusion of PTS. Routine blood examination, electromyography (EMG), and computed tomography (CT) or magnetic resonance imaging (MRI) serve mainly to exclude other disorders. The recovery can be quite lengthy, non-operative treatment is the accepted practice. Supplementary administration of oral prednisolone could shorten the duration of pain. Although the outcome is typically preferable, a substantial amount of patients are left with some residual paresis and functional impairment.

Show MeSH
Related in: MedlinePlus