The description of the pathology of human intervertebral

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1 J Neurosurg Spine 19: , 2013 AANS, 2013 Little-known Swiss contributions to the description, diagnosis, and surgery of lumbar disc disease before the Mixter and Barr era Historical vignette Martin Nikolaus Stienen, M.D., 1 Werner Surbeck, M.D., 1 Ulrich Tröhler, M.D., Ph.D., 2 and Gerhard Hildebrandt, M.D. 1 1 Department of Neurosurgery, Kantonsspital St. Gallen; and 2 Institute of Social and Preventive Medicine, University of Bern, Switzerland The understanding of lumbar spine pathologies made substantial progress at the turn of the twentieth century. The authors review the original publication of Otto Veraguth in 1929 reporting on the successful resection of a herniated lumbar disc, published exclusively in the German language. His early report is put into the historical context, and its impact on the understanding of pathologies of the intervertebral disc (IVD) is estimated. The Swiss surgeon and Nobel Prize laureate Emil Theodor Kocher was among the first physicians to describe the traumatic rupture of the IVD in As early as 1909 Oppenheim and Krause published 2 case reports on surgery for a herniated lumbar disc. Goldthwait was the first physician to delineate the etiopathogenes is between annulus rupture, symptoms of sciatica, and neurological signs in his publication of Further publications by Middleton and Teacher in 1911 and Schmorl in 1929 added to the understanding of lumbar spinal pathologies. In 1929, the Swiss neurologist Veraguth (surgery performed by Hans Brun) and the American neurosurgeon Walter Edward Dandy both published their early experiences with the surgical therapy of a herniated lumbar disc. Veraguth s contribution, however, has not been appreciated internationally to date. The causal relationship between lumbar disc pathology and sciatica remained uncertain for some years to come. The causal relationship was not confirmed until Mixter and Barr s landmark paper in 1934 describing the association of sciatica and lumbar disc herniation, after which the surgical treatment became increasingly popular. Veraguth was among the first physicians to report on the clinical course of a patient with successful resection of a herniated lumbar disc. His observations should be acknowledged in view of the limited experience and literature on this ailment at that time. ( Key Words lumbar disc herniation sciatica Theodor Kocher Otto Veraguth William Jason Mixter Joseph S. Barr intervertebral disc The description of the pathology of human intervertebral disc (IVD) diseases and of their surgical therapy is closely related to the experiences and reports of several European and North American physicians of the late 19th and early 20th centuries. It was a time of fast knowledge acquisition and transcontinental exchange of information. As English evolved as the universal scientific language, important contributions made by scientists and physicians who did not master English were often not appreciated internationally. In the field of lumbar spinal pathology and surgery, the Swiss physician Otto Veraguth ( ) and the surgeon Hans Brun ( ) made an early contribution in 1929 that we Abbreviation used in this paper: IVD = intervertebral disc. aim to highlight in this paper. Veraguth s original case report is put into the context of the literature of its time, and its impact on the understanding of pathologies of the IVD is discussed. In 1896 the 1909 Nobel Prize laureate Emil Theodor Kocher ( ) had already pointed out the possibility of traumatic IVD rupture At that time, however, IVD rupture was not associated with sciatica. 15 Despite the early description of the clinical entity sciatica in 1764 by Cotugno, 1 the causal relationship between the herniation of a lumbar disc, compression of a lumbar nerve root, and radicular leg pain remained unrecognized until the This article contains some figures that are displayed in color on line but in black-and-white in the print edition. 767

2 M. N. Stienen et al. beginning of the 20th century. Accordingly, therapeutic strategies could not evolve until the pathophysiological framework of sciatica had been discovered. In 1929, the Swiss neurologist Otto Veraguth of Zürich, Switzerland (Table 1), 20 published a case report involving successful transdural resection of IVD fragments between the fourth and fifth lumbar vertebral bodies in a patient with a conus-cauda syndrome (combined conus medullaris and cauda equina syndromes). 21 The surgery on this patient was performed by the surgeon Hans Brun, a Privatdozent at the University of Zürich. The clinical course and the surgical therapy of this patient are summarized below. Veraguth s Case Report In his Neurologische Skizzen (Neurological sketches), Veraguth reported the cases of 4 patients who were treated surgically at the University Clinic Zurich, Switzerland. 21 Three of these patients had diverse spinal pathologies. In addition to a patient with a cervical rib and a patient with an epidural mass ( Peripachymeningitis spinalis chronica non specifica ) between the third and eighth thoracic vertebral bodies after chronic osteomyelitis, Veraguth described the clinical features and surgical therapy of a 36- year old female. Between 1926 and 1927 this woman had suffered from recurring episodes of lumbar spinal pain, accentuated after mechanical stress. In August 1927 she experienced a further painful episode. A massive exacerbation occurred on September 23, 1927, and over the next 2 days, the pain further increased. Bladder weakness and sensory disturbances in the right buttock developed within 5 days. The family physician noticed perianal hypesthesia and a reduced strength of the anal sphincter; an intraspinal space-consuming lesion was suspected. Plain radiograph studies were normal. On October 13, 1927, a suboccipital spinal tap and instillation of an oily contrast medium were performed (analysis of the CSF was unremarkable). The contrast medium sank down to the region of the fifth lumbar vertebral body, outlining a blockage of spinal fluid at that level (Fig. 1). On October 17, complete paralysis of the bladder and the sphincter muscle was observed and the sensory disturbances were progressive (Fig. 2). The surgeon Hans Brun performed an L4 5 laminectomy and dorsal durotomy in this region without identification of a pathological process. However, subsequent palpation of the ventral dural circumference revealed a resistance of approximately the size of a cherry pit. After a ventral durotomy the lesion was resected. Subsequently the lesion was examined histologically. According to Veraguth s pathologist consultant (presumably Ambrosius von Albertini), the final diagnosis was Extradurales Myxochondrom am unteren Ende des Dural-Sackes (extradural myxochondroma at the terminal end of the dural sac). Fig. 3 is another reproduction from Veraguth s original publication, which displays the histology of the removed lesion, a myxochondroma in the terminology of the time. According to our present interpretation, the figure shows IVD tissue. Postoperatively, the pain and sphincter and bladder weakness resolved completely within 3 weeks after surgery. A mild perianal hypesthesia persisted, however. Veraguth stressed that a precise neurological examination would facilitate the localization and surgical intervention. He pointed out that even small extradural masses in the region of the cauda equina could lead to bladder and sphincter disturbances because the nerve fibers had little room for evasion. However, Veraguth did not establish any connection between the spinal mass and IVD tissue in his case report. Context of Veraguth s Case Regarding descriptions of IVD rupture and herniation of a lumbar disc, we have to go back into the 19th century. Probably the first description of a traumatic IVD rupture was by Rudolf Virchow in ,22 After W.F. Wilkins gave a brief mention without much detail on a case with possible traumatic disc protrusion in 1888, 18 it was the Swiss surgeon Emil Theodor Kocher who published on the topic of ruptured IVD in spinal trauma at the end of the 19th century. 9,10 In his 1896 articles, Kocher described his experiences in the diagnosis and treatment of trauma and diseases of the spine and the spinal cord over 245 pages The data were based on the records of 78 patients from the Berne university surgical clinic from 1872 onward. Kocher divided spinal trauma into 2 groups: TABLE 1: Biographical data of Otto Veraguth ( ) Date Biographical Event May 13, 1870 Born in Chur, Switzerland After attending schools in Chur, studies medicine in Zürich, Switzerland, & Heidelberg, Germany 1895 Receives his M.D. from the University of Zürich Trains in neurology in Paris, neurosurgery in London w/ V. Horsley, and neuroanatomy in Zürich w/ C. von Monakow 1900 Is admitted Privatdozent for neurology at the University of Zürich Private practice in neurology & neurosurgery ( works in Paris w/ P. Marie); works also on physical therapy 1917 Founding member & secretary of the Swiss Neurological Society Professor & director of the Institute for Physical Therapy at the University of Zürich President of the Swiss Neurological Society, 1927 honorary president December 17, 1944 Dies in Zürich 768

3 Lumbar disc surgery at the turn of the 20th century Fig. 1. Illustration from the 1929 report of Veraguth (Fig. 1). The image outlines the location of the suspected lesion (black area) after a suboccipital lipiodol injection, the contrast medium used at that time. 1) partial dislocation trauma, which included contusion and distortion of the vertebral body, isolated fractures of the vertebral arch and the spinous process, isolated dislocation of the facet joints, and isolated fractures of the vertebral body; and 2) total dislocation trauma, including dislocation and compression fractures affecting 1 or both vertebral joints, total dislocation trauma with rupture of the IVD, and total dislocation trauma affecting both facet joints and distraction of the fracture surface of the vertebral body. Kocher provided numerous pathoanatomical reports and extensive discussions of special cases in children and adults. He put an emphasis on the relevance of contusio spinalis (spinal cord contusion) in the genesis of hematomyelia. Partial lesions of the spinal cord in spinal distortion were also mentioned. Graphic illustrations of the segmental peripheral and central neurological deficits were provided for some of the case reports. Kocher also illustrated the impact of spinal lesions on vegetative functions in detail. But even more, he also acted therapeutically as a surgeon and valued the technique of decompressive laminectomy. 8 Thirty-six patients with spinal trauma, 6 with spondylitis, 2 with spinal sarcoma, and 2 with spina bifida were included in his case series. Detailed postmortem findings from the affected spinal region were often provided by his pathologist colleague, Theodor Langhans. The different clinical courses were characterized according to the location of the spinal lesion (cervical, thoracic, or lumbar). Among his most valuable contributions in the field of spine surgery is a figure in the appendix of the first volume of his surgical textbook (Fig. 4), representing an early example of the illustration of the assignment of dermatomes to the specific segments of the spine and the medulla. 9,10 This dermatome map was published before the commonly known illustrations by Charles Sherrington (1898), Head and Campbell (1900), or Otfrid Foerster (1933). 5,7,17 A detailed description of Kocher s contributions to the field of spinal and cranial neurosurgery in chronological order, together with his ideas on valve surgery for epilepsy, has been recently published. 8,19 The credit for the initial description of the surgical treatment of a degenerative herniated lumbar disc with subsequent conus-cauda syndrome probably must be given to the German physician Hermann Oppenheim and the surgeon Fedor Krause. 14 In their 1909 publication they indicated that they had performed an L-4 laminectomy in a patient as early as They chose the spinal approach craniad from the expected stenosis. During an intradural exploration they only found cauda equina fibers spread to the left and right; a tumor was expected but not found. 14 In a second case, an L-3 laminectomy was conducted for conus-cauda syndrome that occurred after physical work in a 43-year-old man. Histopathological analysis confirmed the cartilage origin of the transdurally resected mass. In both cases the pain resolved postoperatively and urinary continence was restored. Oppenheim and Krause noted the symptoms resulting from sudden compression of the cauda equina; they interpreted the findings as tumors ( enchondromas ). Goldthwait 6 and Middleton and Teacher 12 analyzed the pathology of ruptured IVDs more comprehensively in The former established a relationship between the symptoms of sciatica and paraplegia and the underlying disc pathology (the traumatic rupture of the annulus fibrosus) in 2 cases. 6 He also mentioned the indication for surgery for both patients. Lumbar laminectomy for decompression of the cauda equina was performed in 1 patient with prolapsed disc material and subsequent spinal canal stenosis, of whom Goldthwait was in charge (the surgeon was Harvey Cushing). Cushing chose a midline incision from the first lumbar vertebral body down to the sacrum. Then, an intradural inspection of the cauda equina followed without revealing a pathological finding. The disc material was not realized as such intraoperatively 769

4 M. N. Stienen et al. Fig. 2. Illustration from the 1929 report of Veraguth (Fig. 3). The figure illustrates the clinical course of the sensory deficits of the patient from Veraguth s report. Veraguth discriminates between the sensory qualities touch (Berührungshypästhesie), pain (Analgesie) and temperature (Kälteanästhesie = inability to feel cold, Wärmeanästhesie = inability to feel warmth). and thus was not extirpated. 6 Goldthwait did not establish the relationship between annulus rupture and cauda compression until extensive anatomical studies after the event. The special value of his report lies in the fact that he was able to denominate the etiopathogenesis between annulus rupture, symptoms of sciatica, and neurological signs. He did not expect a tumorous lesion, as was still reported in subsequent publications. 3,21 In contrast, Goldthwait assumed early on that sciatica was more often related to pathologies of the IVD. His assumptions were based on the second case of his publication, in which disc tissue was proven to be the cause of conus-cauda compression. He did not provide more details concerning the surgery in his report. He did indicate that the patient was also under the care of the physician Dr. Crosbie and that Fig. 3. Image from the 1929 report of Veraguth (Fig. 4), showing the histology of the resected material. The pathologist, presumably Dr. von Albertini, diagnosed a myxochondroma. According to our present interpretation, the figure shows IVD tissue. he anticipated a more detailed case report by Crosbie. 6 The authors of the present article, however, are not aware of this report. Also in 1911, Middleton and Teacher 12 described traumatic lumbar disc herniation. They were able to demonstrate in autopsy studies that traumatic IVD rupture resulted from heavy lumbar spinal load (lifting heavy weights). 12 Interestingly, the authors also cited Kocher s early contribution on this topic. 9,10 Likewise, Cushing was aware of Kocher s article and cited it in his own report on 2 spinal gunshot injuries. 2 Among other reasons, Cushing s acquaintance with Kocher s article would later lead him to Berne where he worked at Hugo Kronecker s Institute of Physiology on a subject that Kocher had suggested. 8,19 In 1915, the American Charles Elsberg cured a patient suffering from sciatica by resecting a ruptured ligament of subflavum with intraoperative evidence of fourth lumbar nerve root compression. 4 However, we interpret Elsberg s finding as an early description of a hypertrophied ligamentum flavum causing spinal stenosis and thus leg pain. 11 In 1929, the pathologist Christian Georg Schmorl of Dresden, Germany, described the so-called Schmorlnodules, representing, according to him, prolapsed fragments of the nucleus pulposus after traumatic or degenerative rupture of the annulus fibrosus. 16 That same year, the neurosurgeon Walter Dandy of Johns Hopkins, who had been trained by Cushing (among others), published another important paper on this issue. 3 He described the transdural removal of loose cartilage after lumbar laminectomy for cauda equina compression. He suspected this cartilage to have come from prolapsed disc material. 3,21 Five years later the American surgeons William Jason Mixter ( ) and Joseph S. Barr ( ; yet another former trainee of Cushing), finally proved the 770

5 Lumbar disc surgery at the turn of the 20th century Fig. 4. Kocher was among the first physicians to publish an illustration of the dermatomal skin innervation. 9,10 The names on the figure represent patients treated by Kocher and who served as the basis of his observations. This map has remained unknown to most English-speaking neurologists and neurosurgeons, however. Modern dermatome maps are based on the work of Otfrid Foerster published in causal relationship between herniation of a lumbar disc and sciatica. 13 This landmark paper gave birth to an increasing popularity of spine surgery for herniated lumbar discs (Fig. 5). 15 Discussion We have shown that the Swiss neurologist Veraguth and his surgeon colleague Brun (surgery performed in October 1927) were among the first surgeons after Oppenheim and Krause (1909) and before Dandy (who performed surgery a few months later in May 1928) to have resected a herniated lumbar disc. Surely, therefore, Dandy s claim to be the first was mistaken, considering also the operations of Krause, possibly Goldthwait, and others. 11 Dandy was probably acquainted with neither the papers of Oppenheim and Krause nor with Veraguth s paper, published in the Deutsche- and the Schweizerische Medizinische Wochenschrift, respectively. While Krause s and Dandy s contributions have later been valued in the literature on the history of spine surgery, 11,18 Veraguth s publication on this matter was forgotten. Veraguth described the clinical signs of the patient in detail (Fig. 2), correlated them to the radiological signs (Fig. 1), and correctly diagnosed a space-occupying lesion of the lower lumbar spinal segment to be the cause of the ailment. After careful discussion of all therapeutic strategies, the surgeon Brun performed the surgery and the patient s pain disappeared after resection of the lesion. Although a pioneering surgical achievement must be conceded to these early spine surgeons, many were not aware of the etiology of their surgical specimen. One reason could be that the pathophysiological relationship between IVD rupture and sciatica was doubted at that time. Surgeons (and/or their pathologist colleagues) of this generation appear to have tended to interpret the intraoperative findings as neoplasms ( enchondroma according to Oppenheim and Krause, 1909, 14 and Elsberg, 1915; 4 and myxochondroma, according to Veraguth, ). In contrast, Dandy concluded that the loose cartilage he had removed presumably originated from the IVD and mimicked a spinal neoplastic lesion. 3 This issue was finally solved by the Americans Mixter and Barr in After their publication, consensus was reached regarding the origin of the spinal masses. Evidence rapidly accumulated in favor of dislocated disc fragments following a traumatic event. There were still years to go until a deeper understanding of the biology of the IVD, the pathophysi- 771

6 M. N. Stienen et al. ology of degenerative disc disease, and its medicolegal impact would develop. Conclusions The Swiss physicians Kocher, Veraguth, and Brun made remarkable early contributions to the understanding of lumbar pathologies and their surgical treatment, encompassing IVD pathologies, diseases of the lumbar spine, and the spinal cord. Even though Veraguth s contribution has not been appreciated internationally for the reasons stated above, his article is among the first to report in great detail the clinical course of diagnosis and transdural surgical treatment of a patient with conus-cauda syndrome after herniation of a lumbar disc. 21 However, for Veraguth and his surgeon Brun, the origin of the surgical specimen remained unclear. Yet given the limited experience and literature on such a condition up to the 1920s, the modern management strategy used in their patient deserves to be acknowledged. It was our aim to highlight this previous work and honor the contributions of the physicians mentioned. Acknowledgments We thank Dr. Christian Öhlschlegel (Neuropathology, Kantonsspital St. Gallen, Switzerland) for reexamination and interpretation of Figure 2 and Irina Idrissova and Dr. Nicolas Roydon Smoll for their editorial support. Dedication We dedicate this article to the 70th birthday of Prof. Norfrid Klug, former head of the university clinic for neurosurgery in Cologne, Germany. Disclosure The authors report no conflict of interest concerning the materials or methods used in this study or the findings specified in this paper. Author contributions to the study and manuscript preparation include the following. Conception and design: Hildebrandt, Stienen. Acquisition of data: Hildebrandt, Stienen, Tröhler. Analysis and interpretation of data: Hildebrandt, Stienen, Tröhler. Drafting the article: Hildebrandt, Stienen. Critically revising the article: all authors. Reviewed submitted version of manuscript: all authors. Administrative/technical/material support: Stienen. Fig. 5. Timeline ( ) displaying the important steps toward a modern pathophysiological understanding and treatment of a herniated lumbar disc. References 1. Cotugno DFA: De Ischiade Nervosa Commentarius. Naples: Fratres Simonios, Cushing H: Haematomyelia from gunshot wounds of the spine. A report of two cases, with recovery following symptoms of hemilesion of the cord. Am J Med Sci 115: , Dandy W: Loose cartilage from intervertebral disc simulating tumor of the spinal cord. Arch Surg 19: , Elsberg CA: The extradural ventral chondromas (ecchondroses), their favorite sites, the spinal cord and root symptoms they produce, and their surgical treatment. Bull Neurol Inst NY 1: , Foerster O: The dermatomes in man. Brain 56:1 39, Goldthwait JE: The lumbosacral articulation: an explanation of many cases of lumbago, sciatica, and paraplegia. Boston Med Surg 164: ,

7 Lumbar disc surgery at the turn of the 20th century 7. Head H, Campbell AW: The pathology of herpes zoster and its bearing on sensory localization. Brain 23: , Hildebrandt G, Surbeck W, Stienen MN: Emil Theodor Kocher: the first Swiss neurosurgeon. Acta Neurochir (Wien) 154: , Kocher T: Die Läsionen des Rückenmarks bei Verletzungen der Wirbelsäule. Mitt Grenzgeb Med u Chir 1: , Kocher T: Die Verletzungen der Wirbelsäule zugleich als Beitrag zur Physiologie des menschlichen Rückenmarks. Mitt Grenzgeb Med u Chir 1: , Markham JW: Surgery of the spinal cord and vertebral column, in Walker AE (ed): A History of Neurological Surgery. Baltimore: Williams & Wilkins, 1951, pp Middleton GS, Teacher JH: Injury of the spinal cord due to rupture of an intervertebral disk during muscular effort. Glasgow Med J 76:1 6, Mixter WJ, Barr JS: Rupture of the intervertebral disc with involvement of the spinal canal. N Engl J Med 211: , Oppenheim H, Krause F: Ueber Einklemmung bzw. Strangulation der Cauda equina. Dtsch Med Wochenschr 35: , Parisien RC, Ball PA: William Jason Mixter ( ). Ushering in the dynasty of the disc. Spine (Phila Pa 1976) 23: , Schmorl G: Über Knorpelknötchen an der Hinterfläche der Wirbelbandscheiben. Fortschr Röntgenstr 40: , Sherrington CS: Experiments in examination of the peripheral distribution of the fibres of the posterior roots of some spinal nerves, part II. Phil Trans R Soc B 190:45 186, Sonntag VKH: History of degenerative and traumatic diseases of the spine, in Greenblatt SH, Dagi T, Epstein MH (eds): A History of Neurosurgery In Its Scientific and Professional Contexts. Park Ridge, IL: American Association of Neurological Surgeons, 1997, pp Surbeck W, Stienen MN, Hildebrandt G: Emil Theodor Kocher valve surgery for epilepsy. Epilepsia 53: , Süssli P: Otto Veraguth : Neurologe und Professor für physikalische Therapie. Zürich: Juris, Veraguth O: Neurologische Skizzen. Schweiz Med Wschr 59: , Virchow RLK: Untersuchungen über die Entwickelung des Schädelgrundes im Gesunden und krankhaften Zustande. Berlin: G Reimer, 1857 Manuscript submitted November 5, Accepted August 19, Please include this information when citing this paper: published online September 27, 2013; DOI: / SPINE Address correspondence to: Gerhard Hildebrandt, M.D., Department of Neurosurgery, Kantonsspital St. Gallen, Rorschacher Str. 95, CH-9007, St. Gallen, Switzerland. gerhard.hildebrandt@ kssg.ch. 773