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The study and description of obstetrical paralysis began in the 18th century and came from various countries in Europe. Although the first description in the literature came from England, most of the work done in the 19th century came from French authors. The name of the pathological condition was given by Duchenne de Boulogne in the second part of the 19th century. The surgical treatment was developed essentially in the United States at the beginning of the 20th century but was abandoned during the 1930s, only to be begun again in France in 1977. The French contributions are exposed, but they are only a part of the vast international interest for this condition.
The history of obstetrical palsy starts in the world literature with the English obstetrician Smellie1 (Fig. 1). In his Treatise of Midwifery (Fig. 2), Smellie offers what has been considered as the first description of this palsy:
The prolonged compression was responsible for a paralysis of both arms which lasted for several days and disappeared after frictions.
There has been controversy about the etiology of this very transient paralysis, however, and there are many who consider the first description to have been given by Jacquemier2 in 1846 in his book Traité des accouchements:
a few days after birth of a strong and well constituted baby, born after a long and difficult labour, I was asked to examine the child who presented a difference between the two upper limbs. The two arms were equally developed but the right was more inert and the shoulder looked less muscular. When raised, the arm fell immediately. The movements of the arm and forearm were normal. I did not prescribe any treatment and after 15 to 20 days the harmony between the two arms was re-established. I supposed that the paralysis was due to the compression of the axillary nerve against the humeral shaft. (p. 785)
a child of 3325 gr was born after a long (24 hours) and difficult labour. The mother had a severe attack of eclampsia and forceps had to be used. The child was born in a state of apparent death but was reanimated after half an hour. When I saw him, (36 hours after birth), he was breathing normally. He had a left facial paralysis and a paralysis of the upper arm of the same side. The left upper limb was flail, the forearm in pronation, the fingers semi-flexed. There was no spontaneous movements although the sensation was preserved. There was an eschar on the left side of the neck, produced by a branch of the forceps which had penetrated deeply and compressed the brachial plexus. The child became weaker and died after eight days. Blood was found in all the tissues surrounding the brachial plexus. In the infraclavicular area the nerves looked normal. The facial nerve had also an haematoma. (p. 147)
Then, during the second part of the 19th century, several cases were described. Depaul4 (Fig. 4), in 1867, described a case of complete palsy in a child born after the use of forceps. The patient had started his recovery after 3 weeks, but follow-up was not possible. Depaul discussed in detail the previous descriptions by Smellie, Jacquemier, and Danyau and concluded that there were two types of paralysis: those provoked by the forceps, which were more severe, and those resulting from compression, which had a better prognosis. Gueniot5 describes in 1868 a similar case, in which he used the forceps under the leadership of Depaul5 (Fig. 5). Gueniot added that the complete paralysis was diagnosed by the mother only on the 11th day! In the discussion following the presentation, Blot and Depaul5 argue about the gravity of this lesion and about the perspectives of recovery. However the next important step came also from a French scientist: Guillaume Duchenne de Boulogne (Fig. 6).
Guillaume Benjamin Amand Duchenne was born on September 17, 1806. He was descended from a family of fishermen, traders, and sea captains who had resided in the harbor city of Boulogne-sur-Mer in Northern France since the first half of the 18th century. Despite his father's efforts to induce him to follow the family seafaring tradition, it was Duchenne's love of science that prevailed. Duchenne went to a local college at Douai, where he received his baccalauréat at the age of 19 years. From 1827, aged 21 years, he studied medicine under teachers like René-Théophile-Hyacinthe Laënnec (1781–1826), Baron Guillaume Dupuytren (1777–1835), François Magendie (1783–1855), and Léon Cruveilhier (1791–1874). He graduated in medicine in Paris in 1831 and, probably influenced by Dupuytren, presented his Thèse de médecine, which was a monograph on burns.
In Paris Duchenne met with a rather cool reception, being ridiculed for his provincial accent and his coarse manners. He was known under the name of Duchenne de Boulogne to avoid confusion with Édouard Adolphe Duchesne (1894–1869), who was a fashionable society physician. Having arrived alone and without funds, Duchenne set to work in charity clinics and hospitals and gained his livelihood from private practice, which presumably became adequate to supply his limited needs. He seemed to live only for his patients and his scholarship, pursuing his clinical neurological studies in a very unorthodox but effective fashion. He was described perhaps somewhat romantically as a strange mariner-like figure who every morning used to haunt one or two major Parisian teaching hospitals to study the most interesting cases, and who then made them an object of his detailed electrotherapeutic studies.
Duchenne was a diligent investigator and was meticulous at recording clinical histories. When necessary he would follow his patients from hospital to hospital to complete his studies. In this way he achieved an exceptionally rich and exquisite research record that was far superior to that available to a single clinician and hospital physician.
Duchenne was a lonely figure at the wards of the Paris hospitals, mocked by the interns and rebuffed by the senior medical staff, whom he called the monarchs of the ward. However, he demonstrated an enormous personal courage and a singleness of purpose and soon obtained a reputation as an outstanding neurologist, thus achieving some degree of academic recognition. The absence of daily hospital routines gave him the time and opportunity to develop his interest in muscular diseases, electrodiagnostics, and electrical stimulation. However unambitious Duchenne may have been, he was frequently involved in disputes concerning forms of diseases that he had discovered, and he was never short of enemies and protagonists.
Toward the end of his life Duchenne became established and popular with, among others, Jean-Martin Charcot (1825–1893) at the Hôtel de Salpêtrière. Charcot was among Duchenne's friends, and they held each other in considerable esteem. Duchenne's clinical ability was such that the great Charcot dubbed him “The Master.” Despite his lack of recognition in France, Duchenne at this stage of his career had become an international celebrity.
More than any other person of his day Duchenne was responsible for developing the technique of the meticulous neurological examination, and he took an early interest in electrophysiology. In 1835 he tried electric stimulation of a patient with neuralgia, despite the facts that the scientist who had previously tried this had been scared off, and that there was a lot of charlatanry around this practice.
Duchenne built his own machine for neuromuscular stimulation and developed the technique of using surface electrodes. Previous electric stimulation had caused a lot of tissue damage. The power source, portable for bedside use, consisted of a coal–zinc battery. Exactly how the stimulation was performed is not known, but it resulted in either repeated contractions or tetanus.
Duchenne discovered that external electrical stimulation could cause muscle movements and initially used this stimulation as a form of therapy, but he then appreciated its possibilities as a diagnostic method. Duchenne employed this technique to analyze the mechanism of facial expression, which research was published and illustrated by many striking photographs. He was one of the first scientists to use photographs to illustrate disease processes.
Duchenne was able to make a distinction between indirect stimulation via the nerve and direct stimulation of the muscle. He also performed electrostimulation of the rectum and the bladder in incontinence and of uterus in amenorrhea. The first results were described in a report to the Académie de Médecine in Paris in 1848.
It was largely Duchenne who introduced the painstaking and at times flamboyant physical examination and interrogation, which has become the stamp of all modern neurologists. Duchenne laid the foundation for Robert Remak (1815–1865) and Hugo Wilhelm von Ziemssen's (1829–1902) subsequent work in physical diagnostics. It therefore strange that the French school remained so discouraging to the great advances achieved in this field in Germany. Duchenne himself ended up in an aggressive dispute with Remak.
Duchenne was probably the first person to use the biopsy procedure to obtain tissue from a living patient for microscopic examination. This aroused a deal of controversial discussion in the lay press concerning the morality of examining living tissues. To perform histopathological diagnostics Duchenne constructed a biopsy needle, which made possible percutaneous muscle biopsies without anesthesia:
I have tried to create a sort of anatomy of the living (anatomie vivante). I have defined precisely the isolated and individual action of every muscle using exact methods. It is not for me to judge the value of these investigations, but I dare say that if my results are corroborated, muscular physiology will have a whole new look.
Later Duchenne's research interest turned more toward pathology and, eventually, to the pathological anatomy of the nervous system. In this field he also achieved significant results.
In the first two editions of De l'Electrisation localisée Duchenne renders exact clinical descriptions of the natural course of polio. By a series of observations and deductions he proved that acute poliomyelitis (previously called “paralysie essentielle de l'enfance,” localization unknown) was a disease of motor nerve cells in the spinal cord. He also clarified the various forms of lead poisoning and revealed their electrical reactions.
Duchenne also made prognostic evaluations on the basis of the contractional capacity of the muscle in connection with electric stimulation and suggested—without having any histological founding—that the profound paralysis of poliomyelitis must be caused by a lesion that he located in the anterior horn cells of the spinal cord. This hypothesis proved correct when André Victor Cornil (1837–1898) in 1863 and Charcot in 1870 demonstrated histological changes.
According to Ernest-Charles Lasègue (1816–1883) and Isidor Straus (1845–1896), Duchenne was “of medium height, thickset, active in movement and slow in speech,” with a faint provincial accent, and he closely resembled his father, who had received the Croix de la Légion d'Honneur from Napoleon for valor as a sea captain in the French–English wars.
Although Duchenne was given no official recognition by the Académie de Médecine and the Institut de France, he was made an honorary or corresponding member of academies in Rome, Madrid, Stockholm, St. Petersburg, Geneva, and Leipzig, to his great satisfaction.
During the obstetrical maneuvers, the delivery may be very difficult, especially when there is a breech presentation or after version and the obstetrician is forced in order to extract the body when the head is out, to use his fingers as a hook under the armpits. This may result in traction or compression of the nerves resulting in paralyses. It may happen even with the most experienced obstetricians. (p. 358)
He then makes a very fine and precise review of the previous published cases, noting, for instance, that the case described by Blot was probably the description of a spastic paralysis and not an obstetrical palsy.
Duchenne then described four personal cases that represent the first precise exposition of the upper paralysis:
In the upper limb, the forearm is extended, the hand in pronation, due to internal rotation of the humerus. The proximal part of the arm is smaller with wasting of the muscles. The biceps, the deltoid, the supraspinatus, the brachialis are paralyzed. The cutaneous sensation is maintained in all children. In all cases the hand is normal. (p. 359)
This description is not only a very accurate report of four cases of avulsion injury of the upper roots after a breech presentation but it created the term paralysie obstetricale (obstetrical palsy), as well as proposing a traumatic etiology: The modern era of obstetrical paralysis had started.
However, the treatment suggested for all these cases by Duchenne was simply electrical stimulation, which, of course, gave him some satisfactory results.
Duchenne goes on in another chapter to describe subacromial obstetrical paralysis, which resembles closely a complete palsy associated with the late sequelae of a shoulder imbalance: posterior subluxation of the humeral head (Fig. 8).
The description phase was almost complete—it was time now for understanding and physiopathology.
One of the great names of this century was Claude Bernard7,8,9 (Fig. 9). Born in 1813, the son of a winemaker of the Beaujolais area, Bernard became a medical intern in Paris in 1837. In 1855, he became professor at the College de France and held the chair of physiology. In 1852 and 1858, he made his major contributions on the sympathetic system and its relations to the eye pupil, although Pourfour du Petit had, in 1727, already described the relationship between the intercostal nerves and the eyes.10
Horner,11 a German ophthalmologist, published in 1869 his description of the ptosis sign. Although the sign is called in many countries the Claude-Bernard-Horner sign, and in others it is just known as Horner's sign, it should be called Klumpke's sign. Mrs. Klumpke is, in fact, the author who linked with accuracy the sign with the avulsion of the T1 root.
Augusta Marie Klumpke (Fig. 10) was born October 15, 1859, in San Francisco, the daughter of a prominent businessman. Her parents were separated, and in 1871 her mother moved to Germany with her six children. Two years later they moved to Geneva, where Augusta attended tutorials for girls as well as courses in chemistry and the natural sciences at the academy of Lausanne. Augusta wanted to study medicine, and as there was no faculty of medicine in Lausanne, the family moved to Paris
Despite the resistance of the dean of the faculty, Alfred Vulpian (1826–1887), Augusta was able to enter the school of medicine. In 1882 she won entrance to an externship, a hospital position without residence at the hospital. During her second and third years, Augusta was connected to Vulpian's clinic. It was only a result of intervention by the minister of education, the physiologist Paul Bert (1830–1886), that Augusta in 1887 became the first woman in France to be appointed interne des hôpitaux. She received her doctor's degree in 1889 with the thesis Des polynévrites en général et des paralysies et atrophies saturnines en particulier—a 295-page work.
One of those who had noticed Klumpke's outstanding abilities, while still a student, was Jules Dejerine, who wrote of her: “Elle a toutes les qualités possibles.” They married in 1886.
Augusta Dejerine-Klumpke's participation in her husband's writings was important, but mostly anonymous. She was a skilled illustrator who drew up schematic diagrams and showed unexampled dexterity when slicing microscopic preparations from the central nervous system. Under her own name and with her colleagues, she published a large number of neurological articles.
During the First World War and subsequent following years she was a pioneer in the treatment and rehabilitation of the large number of soldiers afflicted by wounds of the nervous system, and especially of the spinal cord.
This researcher made a crucial contribution in the history of brachial plexus when, in her paper on Contribution à l'étude des paralysies radiculaires du plexus brachial12 (Fig. 11), in 1885, she described with precision complete paralyses, which was different from what was known as the superior palsies called Erb palsies or Duchenne-Erb palsies. Sixteen cases were described and reviewed in detail, and her clinical description has never been matched. She not only describes but explains the signs of lower root avulsion:
This interpretation is the only one which explains the vaso-motor anomalies in the face and the anaesthesia of the medial aspect of the arm. For us, the lesion of the communicating ramus of the first dorsal nerve can explain the ocular anomalies and the lower roots paralyses. Our experiments have shown that the avulsion of the brachial plexus roots will provoke the oculo-pupilar anomalies only if the communicating branch of the first dorsal nerve is injured. (p. 789)
In 1897, Fieux13 (Fig. 12) made a theoretical study of the mechanical factors involved in the algorithm of lesions involved in the brachial plexus injury. He then confirmed his theories using rabbits. His ideas have been widely accepted and are still completely valid (Fig. 13).
In 1897, Guillemot14 published a very interesting paper on 12 cases of obstetrical palsies. All the cases were breech presentations, and all were from the same midwife! Most of them are bilateral cases. This description of the breech cases has been confirmed a century later (Guetjens, Sloof).
In 1898, one of the last important papers of the 19th century was published by Duval and Guillain15 (Fig. 14). In their paper, they exposed an excellent anatomical study of the roots, and the work of Fieux was confirmed (Fig. 15). Duval and Guillain state that the different lesions observed in traumatic brachial plexus can be explained by the anatomical dispositions of the roots.
The turn of the century was another crucial moment in obstetrical paralysis, as surgical repair of the plexus becomes an option. This time the advance comes in England, where, in 1903, Kennedy described three cases improved by suture, and in Germany, where Spitzy and Lange described a few cases that the history moved. Wyeth and Sharp in the United States published in 1917 a very large series (193 cases) of patients who had been operated on. In 1920, Taylor operated on 70 patients who were 3 months of age, with 3 deaths. It was necessary to wait until 1930 (Lauwers16), however, for the first report on surgical repair of the plexus (nine cases) in the French literature. In all these cases, the most common procedure was a neurolysis, and rarely a suture.
This was the last surgical report on obstetrical paralysis for almost half a century. During most of the 20th century, the surgical approach had been forgotten because of the difficulties of surgery, the risks of anesthesia in babies, the lack of knowledge about the grafts, and the average results after operation.
There are then no indications of surgical repair of the plexus and the treatment should be directed to the sequelae. He describes very well the trumpet sign (named after the way of holding the trumpet in a particular unit of the French army) (Fig. 17). He also very well describes the shortening during growth (Fig. 18).
In 1946, André-Thomas,18 a pediatric neurologist, published a paper on obstetrical paralysis that is the best and extensive clinical study on the subject since Duchenne de Boulogne (Fig. 19). At this time, however, the general feeling expressed by the author was that
Are there any indications for surgery in the early phase: we think not! (p. 182)
This remained the situation until 1977, when the surgical approach was renewed (Gilbert 1980).19
During all these years, despite the lack of interest in direct plexus repair, the secondary operations were extensively practiced and developed. Tendon transfers to the shoulder were published by Bertrand in 1960,20 Lacheretz in 1962,21 Merle d'Aubigné in 1947,22 and Rigault in 1970,23 although some authors were still using rotational osteotomies (Chigot 1958).24
At the French congress of orthopedics in 1972, the resulting report made a state-of-the-art review of obstetrical paralysis (Mallet 1972,25 Arthuis 1972,26 Dubousset 1972,27,28,29 Isch 1972,30,31,32 Castaing 1972,33 Lacheretz 1972,34,35 Masse 1972,36,37,38 Rigault 1972,39,40 and Faysse 197241) and proposed a universally used scale of evaluation of the shoulder (Fig. 20). In the same report, Dubousset presented an evaluation scale for the hand (1972).42
Medial rotation contracture of the shoulder had been treated for a long time by Sever (1927)43 by tenotomy of the subscapularis muscle and by capsulotomy.
Saloff Coste44 in 1966 proposed the section of the muscle in front of the scapula without capsulotomy. Carlioz and Brahimi45 refined the operation and proposed the complete des insertion of the muscle from the scapula (1971) (Fig. 21). Since that time, we have used this operation without modification.
In October 1977, the first child was operated on. She was a 4-year-old child with very bad sequelae of a complete paralysis. The brachial plexus was repaired by grafts, but even though the patient reinnervated several muscles of the upper extremity, she was not able to use them.
Since 1977, more than 3500 children have been examined for obstetrical paralysis and have been explored for brachial plexus repair. These patients have been followed and their results assessed for more than 20 years, for some of them (Gilbert).46,47,48,49,50,51 Our recommendation for surgical treatment at 3 months in case of lack of biceps recovery has not varied and is followed by most teams in the world. We believe in an aggressive and complete repair of the plexus in complete paralysis (Haerle 2004),52 with osteotomy of the clavicle and promising results in the hand.
The secondary procedures have been extensively used and reviewed: Sub-scapularis release (Gilbert et al. 1988),53 latissimus dorsi transfers (Pagnotta et al.),54 supination contracture (Allende and Gilbert),55 wrist and finger extension defects (Duclos and Gilbert 1995).56
More than 600 secondary transfers, releases, or osteotomies have been done. In addition to our center, there is one other active center (Dr. Romana—Hopital Trousseau) and several smaller units.
Our main achievement has been to slowly change the mentalities in France but also in several countries and to make the medical community concerned about the obstetrical lesions. The surgical treatment is now the standard indication in most pediatric textbooks.