Ombrédanne–Armingeat syndrome

Isolated postoperative hyperthermic deaths had been reported earlier, but Louis Ombrédanne published the first comprehensive overview in 1922. His pupil Jean Armingeat subsequently undertook a detailed clinical investigation under Ombrédanne’s mentorship, culminating in his 1929 doctoral thesis and their joint report that year.

The syndrome has subsequently been cited as an early description of malignant hyperthermia (MH). However, the historical cases were heterogeneous and included episodes occurring with local anaesthesia or without anaesthesia.

Ombrédanne–Armingeat syndrome is best regarded as an historical antecedent of perioperative hyperthermic syndromes rather than an earlier synonym for modern malignant hyperthermia.

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History

1914 – Nantes otolaryngologists Victor Texier and Louis Levesque published Considérations cliniques sur 300 cas d’antrotomie. Armingeat, following Canuyt and Buzon, later identified one of their 1914 cases as the earliest published case of postoperative pallor–hyperthermia. The specific observation has not yet been identified with certainty within the surviving 1914 material.

1922 – At the Montreal congress, Louis Ombrédanne presented the first travail d’ensemble on the matter. Essentially the first clinical synthesis of the syndrome, describing the characteristic postoperative combination of progressive hyperthermia, pallor, respiratory acceleration and rapid deterioration in infants

1923 – In the first edition of his Précis clinique et opératoire de chirurgie infantile, Ombrédanne warned surgeons of the syndrome. He further defined its clinical features and discussed possible pathogenetic mechanisms.

1924–1927 – A growing otorhinolaryngology literature documented similar deaths, particularly after infant mastoid surgery. Le Mée, André Bloch, Georges Canuyt, Moreau and others refined the clinical description while proposing infectious, anaesthetic, endocrine and neurological mechanisms; the cause remained unresolved.

1928 – René-Marius-Etienne Buzon, a pupil of Georges Canuyt in Strasbourg, completed his thesis Contribution à l’étude de la mort rapide des nourrissons opérés d’antrite mastoïdienne extériorisée. His analysis compared outcomes before and after changes in mastoid operative technique and helped consolidate the earlier ENT literature.

1929 – Jean Armingeat completed his doctoral thesis Le Syndrome Paleur et Hyperthermie Chez les Nourrissons Operés. Armingeat had spent two years as an intern under Ombrédanne at the Enfants-Malades, and the investigation was undertaken on Ombrédanne’s advice. He analysed several hundred postoperative temperature curves, and explored competing pathogenetic explanations. Ombrédanne served as president of the thesis jury.

1929 – Ombrédanne and Armingeat published Le syndrôme paleur et hyperthermie chez les nourissons opérés. The intial report title was to be “rapid and unexpected death of infants following surgical interventions“. However in in preparing the report, they realised that the cases constituted a recognisable “syndrome pâleur et hyperthermie mortel”.

They characterised a syndrome arising typically within 6–12 hours after surgery, with progressive fever, striking pallor, extreme tachycardia and sometimes abrupt cardiovascular collapse. They emphasised that postoperative fever alone did not constitute the syndrome and that the combination with pallor and systemic deterioration was central.

1929 – In a analysis of anaesthetic choice (De l’influence de l’anesthésique employé), Ombrédanne argued that neither ether nor chloroform alone explained the syndrome. He documented cases after general anaesthesia, local anaesthesia and without anaesthesia. He concluded that the choice of anaesthetic played only a limited role, although anaesthesia might help trigger an attack.

1932 – Ombrédanne devoted a specific section to Le syndrome pâleur-hyperthermie chez les nourrissons opérés in the third edition of his Précis clinique et opératoire de chirurgie infantile (p.25).

1960 – Michael Denborough and colleagues recognised malignant hyperpyrexia as a familial peri-anaesthetic disorder after investigating a family with multiple anaesthetic deaths. Subsequent work established inherited susceptibility and ultimately the modern concept of malignant hyperthermia.

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Eponymythology

Ombrédanne–Armingeat syndrome should not be equated directly with modern malignant hyperthermia. The historical syndrome described a heterogeneous group of postoperative events in infants characterised by pallor, hyperthermia, tachycardia and rapid deterioration. Importantly, Ombrédanne documented similar events after local anaesthesia and even in the absence of anaesthesia, and in 1929 argued that the particular anaesthetic agent was not the principal cause.

Later authors retrospectively linked these reports with malignant hyperthermia, sometimes crediting Ombrédanne with an early description of MH. Modern malignant hyperthermia, however, emerged as a distinct familial anaesthetic-susceptibility disorder through the work of Denborough and colleagues. Ombrédanne–Armingeat syndrome is therefore better regarded as a historical antecedent within the broader history of perioperative hyperthermic syndromes, rather than an earlier name for MH.

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Associated Persons

• Louis Ombrédanne (1871-1956)
• Jean Armingeat (1897–1945)
• Georges Canuyt (1888-1975)
• René-Marius-Etienne Buzon (1903-
• Michael Denborough (1929–2014)

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References

Historical references

• Texier V, Levesque L. Trois cents trépanations de l’apophyse mastoïde pour mastoïdite aiguë. Gazette médicale de Nantes. 1914.
• Texier, Levesque. Considérations cliniques sur 300 cas d’antrotomie. Revue hebdomadaire de laryngologie, d’otologie et de rhinologie 1914
• Büzon R. Contribution à l’étude de la mort rapide des nourrissons opérés d’antrite mastoïdienne extériorisée. Thèse : Médecine : Strasbourg : 1928 Thesis
• Canuyt G, Terracol J. La mort rapide des nourrissons et les interventions sur I’antre mastoidien. Revue de Laryngologie, d’Otologie et de Rhinologie, 1925; 46(13): 437-443
• Canuyt G. La mort rapide des nourrissons opérés d’antrite mastoidienne extériorisée. Technique opératoire de l’intervention mastoidienne chez le nourrisson. 1928
• Armingeat J. Le Syndrome Paleur et Hyperthermie Chez les Nourrissons Operés. 1929
• Ombrédanne L, Armingat J. Le syndrôme paleur et hyperthermie chez les nourissons opérés. Gazette des hôpitaux, 1929; 88(102): 1561–1574
• Ombrédanne L. De l’influence de l’anesthésique employé dans la genèse des accidents post-opératoires de pâleur-hyperthermie observés chez les nourrissons. 1929;10:617–618.
• Ombrédanne L. Le syndrome pâleur-hyperthermie chez les nourrissons opérés. Précis de chirurgie infantile, 1932; 3e: 25

Eponymous term review

• Maclean K, Nickson C. Malignant Hyperthermia. CCC