What is pleurodynia
Pleurodynia also called Bornholm disease, Devil’s grippe, epidemic myalgia or epidemic pleurodynia 1), is defined as the sudden occurrence of excruciating lancinating pleuritic chest pain or epigastric pain attacks, commonly associated with fever, malaise, and headaches. Pleurodynia is an uncommon complication of coxsackievirus B infection 2). Coxsackievirus B is an RNA Enterovirus, which usually causes an asymptomatic or brief upper respiratory tract or gastroenteric infection. Classically, commonest strains causing epidemic pleurodynia have been Coxsackievirus B3 and Coxsackievirus A9 and the disease pre-sentation is almost indistinguishable 3). In rare cases, other severe complications of coxsackievirus B infection develop, including aseptic meningitis, pericarditis, orchitis and even as convulsions in infants 4). Coxsackievirus B in particularhave been notoriously associated with myocarditis and pericarditis 5). There have been large case series described in pediatric popula-tions with proteiform appearances 6). However, less frequently cases of pleurodynia secondary to other enteroviruses have been reported (e.g., echovirus types 1, 6, 8, 9 and 19 and further less frequentlywith coxsackievirus A types 4, 6, 9 and 10) 7).
Data on association of epidemic pleurodynia with coxsackievirus B5 is scanty. Bain et al., in 1961 8), for the first time described epidemic pleurodynia, benign pericarditis and aseptic meningitis which was attributable to coxsackievirus B5. In general, respiratory complaints are more common with type B virus infections as compared to type A viral strains. Also coxsackievirus type B strains result is more number of hospitalizations 9).
Coxsackievirus B virus infection occurs most commonly in children younger than 15 years; half of these patients are younger than 5 years, and 30% are younger than 1 year 10). Coxsackievirus B infection is rare in patients older than 60 years. However, pleurodynia most commonly affects adults infected with the virus, with fewer than 10% of cases occurring in patients younger than 20 years. Of the 372 prospectively studied children aged 4-18 years with nonpolio enteroviral infections, only 3% developed pleurodynia. In contrast, 30 of the 78 mostly adult patients with coxsackievirus B-associated cardiac disease had pleurodynia. Therefore, the location of pain is believed to be predominantly thoracic in adults and abdominal in children.
Most cases require admission to the hospital due to non-diagnostic work up in the outpatient settings. Careful history often reveals high attack rate amongst the close contacts and family members 11).
The prognosis is good, with complete recovery with supportive care in most cases 12), however rare complications may include disseminated intravascular coagulopathy, myocarditis, respiratory failure, and hepatic necrosis with coagulopathy 13). No deaths are reported as a direct result of pleurodynia. Symptomatic and expectant management usually suffices, intercostal 2% Xylocaine injections diluted in normal saline have alsobeen tried with success in some cases 14).
Pleurodynia vs Pleurisy
Pleurisy is an inflammation of the lining of the lungs and chest (the pleura) that leads to chest pain when you take a breath or cough. The pleura is a membrane that consists of two large, thin layers of tissue. One layer wraps around the outside of your lungs. The other layer lines the inside of your chest cavity.
Between the layers of tissue is a very thin space called the pleural space. Normally this space is filled with a small amount of fluid—about 4 teaspoons full. The fluid helps the two layers of the pleura glide smoothly past each other as you breathe in and out.
Pleurisy occurs if the two layers of the pleura become irritated and inflamed. Instead of gliding smoothly past each other, they rub together every time you breathe in. The rubbing can cause sharp pain.
Pleurisy may develop when you have lung inflammation due to infection, such as a viral infection, pneumonia, or tuberculosis.
It may also occur with:
- Asbestos-related disease
- Certain cancers
- Chest trauma
- Blood clot (pulmonary embolus)
- Rheumatoid arthritis
The main symptom of pleurisy is pain in the chest. This pain often occurs when you take a deep breath in or out, or cough. Some people feel the pain in the shoulder.
Deep breathing, coughing, and chest movement make the pain worse.
Pleurisy can cause fluid to collect inside the chest. As a result, the following symptoms can occur:
- Shortness of breath
- Rapid breathing
- Pain with deep breaths
Treatment depends on the cause of the pleurisy. Bacterial infections are treated with antibiotics. Surgery may be needed to drain infected fluid from the lungs. Viral infections normally run their course without medicines.
Taking acetaminophen or ibuprofen can help reduce pain.
Recovery depends on the cause of the pleurisy.
Pleurodynia is an uncommon complication of coxsackievirus B infection. Underlying mechanism for the phenotypical presentation of epidemic pleurodynia are postulated to be secondary to local viral proliferation in the muscles of chest wall, diaphragm and abdominal muscles 15). The most likely port of entry is pharynx, proliferation of virus takes place in lymphatic tissues and reaches muscle by blood stream 16). Lepine et al. 17) first described the muscle involvement with the coxsackievirus B and associated inflammatory lesions with isolation of virus from the muscle tissue. Despite its association with muscle instead of pleura or peritoneum, only minority of cases have revealed elevated levels of serum aspartate aminotransferase or creatinine kinase 18).
The striated muscle is the actual anatomic structure targeted by the coxsackievirus B and is responsible for the attacks of severe chest pain 19). Therefore, the term pleurodynia may be a misnomer because only some patients with the condition actually develop pleuritis (i.e., inflammation of the pleural surface). In patients with pleurodynia, the striated intercostal muscles necrose, which explains the frequent elevations in serum creatine kinase levels 20). Some of the more chronic complications, such as myocarditis, dermato-polymyositis, chronic fatigue syndrome, and possibly, juvenile-onset diabetes type 1, are believed to be immune mediated 21).
The coxsackievirus B virus has an incubation time of 1 week in the gastrointestinal tract and then, through hematogenous dissemination, involves the target organs, most commonly the skeletal muscles but also the central nervous system (i.e., meningitis, encephalitis) and myocardium (i.e, carditis with or without associated pericarditis). Coxsackievirus B can be recovered in the stool or pharynx for up to 2 weeks after the resolution of the symptoms.
Pleurodynia also called Bornholm disease, is defined as the sudden occurrence of lancinating chest pain or abdominal pain attacks, commonly associated with fever, malaise, and headaches. Fever is the commonest presentation, reported in almost 70% of the subjects and the typical“devil’s grip” or thoracic stitch is present in about 40% of the cases 22).
Duration of chest pain may last up to 4 days and duration of pain correlates well with duration of fever 23). Laboratory data other than specific virological studies is usuallynot helpful. Patient may have elevated sedimentation rate similar to ourpatient, but this is found to be non-specific 24). Cases where myocardium or pericardium is involved, it may present with nonspecific EKG changes such as T wave inversion that resolve with resolution of the disease 25). In some case series pulmonary infiltrates with orwithout pleural effusions have been found to be a consistent feature inabout half the cases 26).
To relieve pleurodynia chest pain symptoms, your doctor may recommend:
- Acetaminophen or non-steroidal anti-inflammatory drugs (such as ibuprofen) to control pain.
- Intercostal 2% Xylocaine injections diluted in normal saline have also been tried with success in some cases 27).
With complete recovery with supportive care in most cases 28).
References [ + ]
|1.||↵||Huebner R.J., Risser J.A., Bell J.A., Beeman E.A., Beigelman P.M., Strong J.C. Epidemic pleurodynia in Texas; a study of 22 cases. N. Engl. J. Med. 1953;248(7):267–274|
|2, 10, 19, 20, 21.||↵||Pleurodynia. https://emedicine.medscape.com/article/300049-overview|
|3, 6, 23, 24.||↵||A.Y. Chong, L.H. Lee, H.B. Wong. Epidemic pleurodynia (Bornholm disease) outbreak in Singapore. A clinical and virological study. Trop. Geogr. Med., 27 (2) (1975), pp. 151-159|
|4, 8.||↵||H.W. Bain, L.D. Mc, S.J. Walker. Epidemic pleurodynia (Bornholm disease) due to Coxsackie B-5 virus. The inter-relationship of pleurodynia, benign pericarditis and aseptic meningitis. Pediatrics, 27 (1961), pp. 889-903|
|5.||↵||I. Gaaloul, S. Riabi, R. Harrath, T. Hunter, K.B. Hamda, A.B. Ghzala, et al. Coxsackievirus B detection in cases of myocarditis, myopericarditis, pericarditis and dilated cardiomyopathy in hospitalized patients. Mol. Med. Rep., 10 (6) (2014), pp. 2811-2818|
|7, 28.||↵||M. Leendertse, M. van Vugt, K.S. Benschop, K. van Dijk, R.P. Minnaar, H.W. van Eijk, et al. Pleurodynia caused by an echovirus 1 brought back from the tropics. J. Clin. Virol. Off. Publ. Pan Am. Soc. Clin. Virol., 58 (2) (2013), pp. 490-493|
|9, 13.||↵||C.J. Lee, Y.C. Huang, S. Yang, K.C. Tsao, C.J. Chen, Y.C. Hsieh, et al. Clinical features of coxsackievirus A4, B3 and B4 infections in children. PloS One, 9 (2) (2014), Article e87391|
|11.||↵||J. Prisman, H. Shrand Epidemic myalgia in Cape Town; pleurodynia or Bornholm disease. South Afr. Med. J. Suid-Afrikaanse tydskrif vir geneeskunde, 24 (17) (1950), pp. 309-311|
|12.||↵||Unusual cause of chest pain, Bornholm disease, a forgotten entity; casereport and review of literature. Respiratory Medicine Case Reports 25 (2018) 270–273|
|14, 27.||↵||G.I. Tagarakis, D. Karangelis, F. Tsolaki, M. Dikoudi, T. Koufakis, K. Mouzaki, et al. Bornholm disease–a pediatric clinical entity that can alert a thoracic surgeon. J. Paediatr. Child Health, 47 (4) (2011), p. 242|
|15.||↵||Lal A, Akhtar J, Isaac S, et al. Unusual cause of chest pain, Bornholm disease, a forgotten entity; case report and review of literature. Respir Med Case Rep. 2018;25:270–273. Published 2018 Oct 9. doi:10.1016/j.rmcr.2018.10.005 https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6197799|
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|17.||↵||Lepine P., Desse G., Sautter V. Muscle biopsy with histological examination and isolation of Coxsackie virus in epidemic myalgia (Bornholm disease) Bulletin de l’Academie nationale de medecine. 1952;136(5–6):66–69|
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|22.||↵||T. Johnsson. Studies on the etiology of Bornholm disease (epidemic pleurodynia). II. Epidemiological observations. Archiv fur die gesamte Virusforschung, 5 (4) (1954), pp. 401-412|
|25.||↵||N.H. Swann. Epidemic pleurodynia, orchitis, and myocarditis in an adult due to Coxsackie virus, group B, type 4. Ann. Intern. Med., 54 (1961), pp. 1008-1013|
|26.||↵||W.T. Huang, P.I. Lee, L.Y. Chang, C.L. Kao, L.M. Huang, C.Y. Lu, et al. Epidemic pleurodynia caused by coxsackievirus B3 at a medical center in northern Taiwan. J. Microbiol. Immunol. Infect. Wei mian yu gan ran za zhi., 43 (6) (2010), pp. 515-518|