Bornholm disease

Description

• The self-limiting illness usually lasts 3-5 days and causes fever and sharp, spasmodic pain in the chest wall or abdomen
• Associated with coxsackie B virus and most commonly seen in children and adolescents
• Sometimes preceded by a moderate to severe headache, nausea, vomiting, and pharyngitis
• Pleurodynia is a diagnosis of exclusion – it can mimic more serious diseases such as pneumonia, myocardial infarction and an acute abdomen which need to be ruled out

Also known as: epidemic muscular rheumatism, pleurodynia, myalgia, and myositis or eponymously as Bornholm disease, Sylvest-Bing disease, Bamble disease or just as the ‘Devil’s grip’

Epidemics

Local outbreaks with high infection rates within affected households have occurred, and major epidemics can take place every 10 to 20 years.

Numerous epidemics have been reported in the medical literature, particularly in the Scandinavian countries. Epidemic myalgia, or pleurodynia, was first recognized by Finsen in Iceland in 1856; and first published in 1872 by Daae. The first description in the American medical literature was by Dabney in 1888.

History of Bornholm disease

In 1856 and again in 1865, Jøn Constant Finsen (1826-1885) in Iceland observed outbreaks of an epidemic disease which he termed muscular rheumatism of the chest, or pleurodynia. In 1874 He published his findings as as ‘Pleurodyne‘ in his dissertation for the medical doctorate at the University of Copenhagen ‘Observations regarding the disease conditions in Iceland‘

1872 – A. Daae recorded an epidemic occurrence of ‘acute muscular rheumatism‘ starting in Bamble, Norway on May 13, 1872 with 290 cases and incubation period of 72 hours.

As a rule the patient has a stitch in one side of the chest, most often without any precursory ailment, but sometimes after an attack of chills; the stitch is often accompanied by pains in the back, shoulders, epigastrium and abdomen and these pains are described sometimes as oppressive, sometimes as shooting or aching; less fre­quently they are felt at the back of the head, neck, arms, legs and even in the fingers. There is considerable diffi­culty in moving the affected parts, especially the chest, therefore the respiration is laboured, sometimes to such an extent that the patient feels as if he were being strangled

Daae 1872

District attorney C. Homann recorded in the same journal the series of 474 cases (346 patients out of a population 1,947 in Bamble). He recorded the epidemic to have run ‘an uninterrupted course, being transmitted by infection from patient to patient for a period of three summer months‘.

Repeated epidemics which occurred in Norway were notified in the obligatory annual reports to the Health Department under the heading ‘Bamble disease‘. Areas particularly affected have been eponymously affiliated: ‘Bamble‘, ‘Skien‘ and ‘Drangedal‘

1888 – On Tuesday, June 5, 1888, William C. Dabney (1849-1894), professor of obstetrics and practice of medicine in the University of Virginia, Charlottesville, recorded a case of pleurodynia, the first to be published in North America.

[The child] had been taken suddenly a few hours before with violent pain in the left side of the chest nearly over the region of the heart. His temperature was 103°F and the skin extremely hot and pungent to the touch…His chief complaint was of the pain in the chest, which was excruciating and aggravated by the slightest movement, or by drawing a long breath. Nothing abnormal could be discovered about the thoracic organs, however, on physical examination.

Dabney 1888

Dabney described 29 cases in all, and “saw at least as many more subsequently, but, being extremely busy with the final examinations of the medical students, did not take notes of them.” The Virginia epidemic was notable for its occurrence in children, including one of Dr. Dabney’s, and its familial distribution.

The pain was by far the most striking feature…In character it was usually sharp and lancinating, and was much more violent when the patient attempted to move or to draw a deep breath. (So agonizing was this pain that it was nicknamed the “devil’s grip” by a sufferer from the disease in Rappahannock County, Virginia, and this name became a common one there afterward, as I was told by Dr. W. F. Cooper.) There was slight tenderness over the seat of pain in every case, but it was not nearly so marked as the pain itself. The seat of the pain was usually in the left side of the chest just below the nipple but in some of the cases there was pain in the opposite side, or in the shoulder of the opposite side; and in a few of the cases, especially in children, there was pain in the abdomen, usually in the epigastric region

Dabney 1888

Epidemics of the disease were reported in several states of America in 1923-1925. Various names were applied to these epidemics including epidemic transient diaphragmatic spasm, epidemic pleurodynia, and epidemic diaphragmatic pleurodynia.

1930 – Ernst Sylvest (1880 – 1972), whilst holidaying on Bornholm Island, Denmark witnessed and described the historia morbi of 23 fisherman in Melsted and Gudhjem. He published a paper which gave the case records of his 23 patients. Sylvest considered the most prominent symptom was pain in the abdominal muscles on inspiration. He presumed the condition to be a myositis localised especially to the muscles of the upper abdominal, intercostals, chest wall and diaphragm. As far as he was aware, the disease had been described in Norway by Daae…but had no Danish name. He considered the name ‘Myositis Epidemica‘ not suitable for laymen, and suggested that in Denmark at least, the disease should be called “den Bornholmske Syge” – Bornholm disease.

1930 – Voss published a short note in the same journal as the Sylvest monograph – ‘Myositis epidemica, Bornholm disease, called Taarbæk disease 33 years ago‘. Voss had described a similar epidemic (100 cases) in Taarbæk on Zealand, Denmark in 1897. He had called the disease ‘Rheumatismus muscularis intercostalis febrilis epidemicus‘…

Following Sylvest’s description in 1930, epidemics and sporadic cases of “Bornholm disease ” were reported from all parts of the world. From 1930 to 1934 over 10,000 cases were reported from Denmark alone.

1933 – English general practitioner, Will Pickles (1885-1969) carefully recorded observations on an epidemic in Wensleydale, Yorkshire. Pickles was the first person to describe the disease in detail in Britain and noted the absence of an effective treatment, though ‘hot applications are much appreciated’. Pickles was the first to use the name Bornholm disease in British Medical reports

1948 – Gilbert Dalldorf and G M Sickles isolated a group of viruses associated with ‘poliomyelitis. The pair reviewed samples from two children who experienced limb weakness and were initially diagnosed with poliomyelitis in Coxsackie, a rural town in the state of New York. They made suspensions using faeces from these children and inoculated them to nursing mice, which caused the animals to die. They concluded that the virus causing this disease was different from the one causing poliomyelitis and called it coxsackievirus.

It has become desirable to name those viruses that are pathogenic for suckling mice and hamsters, and that have recently been associated with ‘poliomyelitis’…I have felt that the disease should not be named until something is known of the anatomic lesions in man…however, a provisional designation is needed and it is suggested that the agent be called ‘Coxsackie virus‘, since the’ first recognized human cases were residents of that New York village. Since a number of viruses may be involved, the term ‘Coxsackie group of viruses‘ seems especially suitable.

Dalldorf 1949

1949 – Following the isolation of the Coxsackie group of viruses, three laboratory workers contracted infections while studying it. Curnen et al, found the similarity of the patients’ symptoms to those of Bornholm disease gave rise to the speculation that the coxsackievirus might be the cause of epidemic pleurodynia.

1950 – The viral aetiology of the disease was established by GM Findlay and Elsie Howard, the agent initially isolated found to be Coxsackievirus B1.

Associated Persons

• Jøn Constant Finsen (1826-1885)
• William Cecil Dabney (1849-1894)
• Ejnar Sylvest (1880-1972)
• William Norman Pickles (1885-1969)
• Gilbert Dalldorf (1900-1979)

Alternative names

• Epidemic Myalgia; epidemic pleurodynia and viral myositis
• Norway: Bamble disease (1872), Dragendal disease (1873), Skien disease (1878)
• USA: Devil’s grip (1888)
• Denmark: Taarbæk disease (1897), Den Bornholmske Syge, Bornholm disease (1930); Sylvest-Bing’s disease
• International Bornholmian disease
• …and myriad other synonyms

Controversies

Many prior description of Epidemic pleurodynia/Myositis epidemica. Despite historical references to earlier description of the disease, the name Bornholm disease has remained…

Eponymic chest pain syndromes

• Bornholm disease
• Tietze syndrome
• Syndrôme de Huchard or Texidor Twinge
• Da Costa syndrome
  

References

Historical references

• Daae A. Epidemi i Drangedal af akut Muskelreumatisme, udbredt ved Smitte [Epidemic in Drangedal of acute muscular rheumatism spread by infection] Norsk magazin for lægevidenskaben 1872; 2: 409-413
• Daae A. Om den smitsomme Muskelreumatisme og deres udbredning hovedsagelig i Drangedal’ [The infectious muscular rheumatism and its spread chiefly in Drangedal], Norsk magazin for lægevidenskaben 1872; 2: 529-542
• Homann C. Om en i Kragero Lagedistrikt herskende smitsom Febersygdom [On an infectious febrile disease occurring in the medical district of Kragero], Norsk magazin for lægevidenskaben 1872, 2, 542-55.
• Finsen J. Revmatisme i Brystets Muskler, Pleurodyne. In: Iagttagelser angaaende Sygdomsforholdene i Island: Afhandling for den medicinske Doktorgrad ved Københavns Universitet. 1874: 145-150
• Dabney WC. Account of an Epidemic Resembling Dengue Which Occurred in and Around Charlottesville and the University of Virginia in June, 1888, American Journal of the Medical Sciences. 1888; 96: 488-494
• Epidemic Diaphragmatic Pleurodynia “Devil’s Grip”. Can Med Assoc J. 1924 Nov; 14(11): 1112-3.
• Sylvest E. En Bornholmsk Epidemi. Myositis epidemica [A Bornholm epidemic. Epidemic Myositis], Ugeskrift for Læger 1930; 92: 798-801.
• Voss, A., ‘Myositis epidemica. Den bornholmske syge hed for 33 aar siden den taarbtkske syge’ [Myositis epidemica. Bornhohm disease, called Taarbaek disease 33 years ago], Ugeskrift for Læger 1930; 92: 843-44.
• Pickles WN. “Bornholm” disease: account of a Yorkshire outbreak. Br Med J. 1933; 2(3800): 817-8.
• Sylvest E. Den Bornholmske Syge – Myalgia epidemica. [Bornholm disease-Myalgia epidemica], Doctoral thesis, Copenhagen, 1933.

Epidemic Pleurodynia articles and outbreaks

• Editorial: Epidemic myalgia, or pleurodynia. JAMA. 1934; 102(6): 460-461.
• MacDonald RR, Hewell B, Cooper ML. Epidemic myalgia, or pleurodynia clinical and bacteriologic studies. Am J Dis Child. 1937; 53(6):1425-1434
• Jamieson WM, Prinsley DM. Bornholm Disease in the Tropics. Br Med J. 1947; 2(4514): 47–50
• Ord AFT. Bornholm Disease. Br Med J. 1948; 2(4585): 916
• Hopkins JH. Bornholm disease. Br Med J. 1950; 1(4664): 1230-2
• Warin JF, Davies JB, Sanders FK, Vizoso AD. Oxford epidemic of Bornholm disease, 1951. Br Med J. 1953 Jun 20;1(4824):1345-51.
• Patz IM, Measroch V, Gear J. Bornholm disease, pleurodynia or epidemic myalgia; an outbreak in the Transvaal associated with Coxsackie virus infection. S Afr Med J. 1953 May 9; 27(19): 397-402.
• Pickles WN. Sylvest’s disease (Bornholm disease). N Engl J Med. 1954 Jun 17;250(24):1033-6
• Pickles WN. Bornholm Disease. The Practitioner 1955; 174(1039-1044): 85-86
• Williams WO. Myalgia in Bornholm disease. Proc R Soc Med. 1959 Jun;52(6):477-8.
• Spencer FJ. The Devil and William Dabney. An Epidemiological Postscript. JAMA. 1966; 195(8): 645-8.
• Vogelsang TM. The occurrence of Bamble Disease (epidemic pleurodynia) in Norway. Med Hist. 1967 Jan; 11(1): 86–90

Coxsackie articles

• Dalldorf G, Sickles GM. An Unidentified, Filtrable Agent Isolated From the Feces of Children With Paralysis. Science. 1948; 108(2794): 61-2.
• Dalldorf G. The Coxsackie group of viruses. Science. 1949 Dec 2;110(2866):594.
• Curnen EC, Shaw EW, Melnick JL. Disease resembling nonparalytic poliomyelitis associated with a virus pathogenic for infant mice. J Am Med Assoc. 1949 Nov 26; 141(13): 894-901.
• Dalldorf G. The Coxsackie viruses. Bull N Y Acad Med. 1950; 26(5): 329-35
• Findlay GM, Howard EM. Coxsackie Viruses and Bornholm Disease. Br Med J. 1950; 1(4664): 1233–1236
• Dalldorf G. The William Henry Welch lecture; From clostridium Welchii to the Coxsackie viruses: changing microbiology. J Mt Sinai Hosp N Y. 1952; 19(2): 396-410.
• Dalldorf G. Bornholm disease. Br Med J. 1953; 2(4830): 287-8.