Bornholm disease

Nature – Acute, self-limited enteroviral myositis (predominantly Coxsackie B) with abrupt, severe, pleuritic chest wall or upper abdominal pain (± low grade fever/sweats). Sometimes preceded by a moderate to severe headache, nausea, vomiting, and pharyngitis

Epidemiology – All ages affected, though children predominate; community epidemics every 10–20 years reported.

Aetiology – Coxsackie B enteroviruses (multiple serotypes) with skeletal/diaphragmatic muscle tropism. Isolation during outbreaks and rising neutralising/complement-fixing antibodies in convalescence

Course – Typically 3–5 days, sometimes with short relapses. Benign with full recovery. Manage with analgesia and reassurance; test only to rule out dangerous mimics.

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’

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History of Bornholm disease

1717 – Waldschmidus (Wilhelm Ulrich Waldschmidt, praes.; Christian Stephan Scheffel, resp.) provided
the earliest medical thesis on an epidemic “convulsive” disorder in Holstein, widely cited in later Nordic literature as a proto-Bornholm description.

1735 – Georg Detharding (praes.) & Jørgen/Georg Hannaeus (resp.) published Copenhagen disputation on Eyderost (Eiderstedt) epidemic fevers, aka Stoppelfieber (“stubble fever”) linked in later reviews as an early pleurodynia cluster.

1856, 1865, Jøn Constant Finsen (1826-1885) observed two Icelandic epidemics of “muscular rheumatism of the chest with pleurodynia,” published in his 1874 MD dissertation as Revmatisme i Brystets Muskler, Pleurodyne

1872 – Andreas Daae (1838–1910) 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. He described a contagion with classic stitch-like chest pain and benign course.

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…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) recorded the Charlottesville (Virginia) outbreak popularising the term “devil’s grip”, the first published account 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.

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. He determined the epidemic myalgia was characterised by:

• Prevalence in warmer summer months
• Incubation 3-4 days; epidemic with multiple close contact cases especially within same family
• Can occur at any age; most common in children
• Acute onset
• Main symptom is chest pain (or upper abdomen), which is usually severe and unilateral
• Minimal systemic upset with rare exception of headache, sweating and mild fever
• Symptoms last 2-3 days but duration is unpredictable, may relapse and last up to a month
• Disease is benign with no sequelae
• Epidemic disease, simple to diagnose

Sylvest considered the most prominent symptom was chest and upper abdominal muscle pain on inspiration secondary to a myositis of the muscles of the upper abdominal, intercostals and chest wall. He was aware the disease had been described in Norway by Daae…but had no Danish name. He considered ‘Myositis Epidemica‘ unsuitable for laymen, and suggested “den Bornholmske Syge“

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 (1900-1979) and G M Sickles a new enterovirus from children with paralysis in in Coxsackie, a rural town in the state of New York. They inoculated faecal suspensions from the children into 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. “…disease resembling non-paralytic poliomyelitis associated with a virus pathogenic for infant mice” and speculated that the coxsackievirus might be the cause of epidemic pleurodynia.

1950s – The viral aetiology of the disease was established by G.M. Findlay (1890–1979) and Elsie Howard, in their paper Coxsackie Viruses and Bornholm Disease with isolation of Coxsackievirus B1 (1951). Thordarson et al (1953) in the Reykjavík epidemic (225 cases) with isolation of coxsackie virus from patients with epidemic pleurodynia

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Associated Persons

• Jøn Constant Finsen (1826-1885)
• Andreas Daae (1838–1910)
• William Cecil Dabney (1849-1894)
• Ejnar Sylvest (1880-1972)
• William Norman Pickles (1885-1969)
• Gilbert Dalldorf (1900-1979)

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Alternative names

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

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Controversies

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

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Eponymic chest pain syndromes

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

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References

Historical references

• Waldschmidt WU. Dissertatio Medica De Morbo Epidemio Convulsivo, Per Holsatiam Grassante, Oppido Raro, Memoriæ Celebrati Iubilæi Lutherani Secundi Dicata. 1717
• LVI. Georgii Dethading et Georgii Hannaei. Disputatio de Febribus Eyderostadiensibus epidemicis, vulgo Stoppelfiebern. Haffniae. 1735. (Von den Eyderstädtschen epidemischen Fiebern. S. 249.)
• 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
• Finsen J. Revmatisme i Brystets Muskler, Pleurodyne In: Lagttagelser angaaende Sygdomsforholdene i Island : Afhandling for den medicinske Doktorgrad ved Københavns Universitet. København : C. A. Reitzel, 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.
• Rector JM. Acute epidemic myalgia or pleurodynia: clinical course and diagnosis of the disease in children. Am J Dis Child. 1935; 50(5): 1095–1100
• MacDonald RR, Hewell B, Cooper ML. Epidemic myalgia, or pleurodynia clinical and bacteriologic studies. Am J Dis Child. 1937; 53(6):1425-1434
• Ronald J. Bornholm Disease. Journal of The Royal Naval Medical Service 1942; 28(2): 144-149.
• 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
• Huang WT, Lee PI, Chang LY, Kao CL, Huang LM, Lu CY, Chen JM, Lee CY. Epidemic pleurodynia caused by coxsackievirus B3 at a medical center in northern Taiwan. J Microbiol Immunol Infect. 2010 Dec;43(6):515-8.

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
• Aumonier FJ. Structural changes in the skeletal muscles of suckling mice following infection with a Coxsackie virus of the A group. J R Microsc Soc. 1952 Dec;72(4):218-22.
• 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.
• Thordarson ÓT, Sigurdsson B, Grímsson H. Isolation of coxsackie virus from patients with epidemic pleurodynia. JAMA. 1953;152(9):814–815