Periostitis ossificans versus GarrSs osteomyelitis. Part I. What did Garri: really say? R.E. Wood, DDS, MSc, C.J. Nortjt, BChD, PhD, F. Grotepass, BChD, MChD, S. Schmidt, and A.M.P. Harris, BChD, HonsBSc, Tygerberg, Republic of South Africa

DEPARTMENTS OF MAXILLOFACIAL RADIOLOGY AND ORAL SURGERY, FACULTY OF DENTISTRY, UNIVERSITY OF STELLENBOSCH, TYGERBERG, REPUBLIC OF SOUTH AFRICA

In 1893, C. Garre published an article dealing with the manifestations of acute osteomyelitis. Since then, his name has been associated with diseases such as Garre’s osteomyelitis, chronic sclerosing osteomyelitis, and periostitis ossificans, among others. Scrutiny of a translated version of the original article reveals that Garre was not responsible for the description of the disease that now bears his name. (ORAL SURC ORAL MED ORAL PATHOL 1988;65:773-7)

I n 1893, Professor Dr. C. Garrt wrote a 57-page treatise on the subject of acute osteomyelitis. The title of the article was “Ueber besondere Formen und Folgezustande der akuten infektiosen Osteomyeli- tis.“’ It was a summary of the author’s findings in a series of patients with osteomyelitis seen at the Tiibinger clinic of Prof. Dr. Bruns. The article was written in German and contained several hand- drawn illustrations but no English abstract. The title, translated, means “Special forms and complications of acute infective osteomyelitis.” The term Garri’s osteomyelitis has been discarded by most medical pathologists but still enjoys acceptance in the dental field where occurrence in the jaws has been termed unequivocal.2

Since 1893, there have been at least 18 references in the dental literature to the condition Garre alleg- edly described.2-‘9 The disease, proliferative periosti- tis of GarrB,2 has been attributed to him, as have the conditions known as chronic sclerosing inflammation of the jaw3; periostitis ossificans4; Garri’s osteomy- elitisss6; osteomyelitis with proliferative periostitis7; and GarrC’s proliferative periostitis.s If the reader has been paying close attention, he or she will realize that we spell GarrB’s name in a decidedly different manner than do other authors. It seems that in the 18 articles or major dental textbooks in which Garre has been specifically mentioned, his name has appeared as Garre, not Garre. It is a fine point but makes one

wonder what the statistical odds are of all 18 authors misspelling his name in exactly the same way. Further investigation of the aforementioned refer- ences revealed that 13 of the 18 authors failed to cite Garri’s article in their material even though they attribute certain specific features to the disease that bears his name. Even more discouraging is the fact that the five authors that cite Garre have the title spelled incorrectly in their citatio+ ‘* R, I33 I9 (Fig. 1). One author had listed the date of publication as 1883.14 This prompted us to attempt to clarify exactly what Car& did say.

MATERIALS AND METHODS

The problem of discerning the truth in this matter is very difficult since the journal in which Garre published his work (Beitriige zur klinischen Chirur- gie) stopped being listed by the Index Medicus in the late 1950s and halted publication completely in 1974. Fortunately, one of us had previously read the article in an original volume of the journal. Two photocopies of the article were obtained from the library of the Faculty of Dentistry, University of Toronto, in Toronto, Ontario, Canada. The original cumbersome German language in the article present- ed great difficulty in translation. The work was translated by one of us, and this translation was verified by a second person. Both translators had excellent working knowledge of German and also the

773

774 Wood et al.

AUS DER

TiIIBINGER CHIRURGISCHEN KLINIK.

DES PROF. DR. BRUNS.

IX.

Ueber bwondere Formen und FolgezwMde tier n.k den infcktiiisea Osteomyelitis.

Van rc

Prof. Dr. C. Oar:+, rrrtcm Ad~leosarrt dsr Klinik.

(Hierw ‘hf. V-VI).

Fig. 1. Photograph of first page of article written by C. GarrZ in name (Garrk-not Garrt, as indicated by arrow).

advantage of having medical training. The work was translated into understandable English with mini- mum number of changes in syntax and structure. The final version was compared to the original article by one of the translators. This version was then compared to articles in the modern dental litera- ture.

DlSCltSSlON

Garre’s article was a brilliant example of thorough clinical observation. Unlike some more recent authors, he strived to explain the many features of osteomyelitis as being the result of a single disease entity. He described varying clinical appearances of acute osteomyelitis rather than creating new nomen- clature.

Garre used 72 illustrative cases (98 sites) to discuss 10 specific manifestations and complications of acute osteomyelitis. The duration of disease in his patients ranged from a few days to 29 years. This is in direct contradiction to authors in the dental literature who suggest that he described a new form of chronic osteomyelitis.3.5,7-9 The ten forms of acute osteomyelitis he described are listed in Table I. It is evident that he did not restrict himself to the investigation of a single aspect of osteomyelitis but preferred to tackle it in its entirety.

Garrb attempted to describe the 10 separate sub- jects he discussed as forms or complications of a single disease-acute osteomyelitis (Table I). This is

illustrated in the first condition he discussed, perio-

1893. Note the spelling of the author’s

stitis albuminosa. He believed that this disease was an acute osteomyelitis with qualitative differences in the subperiosteal fluid. Sequestration and involu- crum formation occurred in association with it and the causative organism was thought to be Strepto- coccus pyogenes. He presented six patients who had this form of acute osteomyelitis.

Although Garre believed that the subacute form of acute osteomyelitis could behave as such right from the start, the term really referred to progression of an acute disease. He cited two illustrative patients, both of whom were acutely ill, requiring surgery and extended hospitalization.

The fourth form of acute osteomyelitis delineated by Garre was the bone abscess. It was said to be a rare accompaniment of acute osteomyelitis. From an examination of the material in Garre’s article, it may be surmised that this condition could have been a large accumulation of granulation tissue caused by gross localized osteomyelitis. He spent very little time in discussing this form.

The title “recidivistic form” is a descriptive term that, in this case, means a relapsing osteomyelitis. Garre believed that this form of recurrence consti- tuted a new infection rather than recurrence at a dormant nidus. He included as part of his series one patient whose second lesion occurred 18 years after healing of the primary lesion. He postulated that a localized change in the vascular architecture may

Volume 65 Periostitis ossijcans versus Garrt’s osteomyelitis 775 Number 6

Table I. The ten subjects discussed by Garre Table II. Sites and relative ages of patients with (1893) different acute osteomyelitis as defined by Garre

Subject

1. Periostitis albuminosa 2. Subacute form 3. Sclerosing nonpurulent form 4. Bone abscess 5. Recidivistic osteomyelitis 6. Primary adult osteomyelitis 7. Multiple osteomyelitis 8. Disease of the epiphysis 9. Epiphysiolysis

10. Spontaneous fracture

Page numbers

243-254 254-251 257-263 263-265 265-273 213-276 276-280 280-286 287-293 293-298

have predisposed patients to further infectious epi- sodes.

Total Chronic Sites number Adults Children sclerosing

Femur 40 6 34 5 Tibia 30 8 22 4 Humerus 14 3 I1 1 Radius 3 1 2 1 Metatarsals 2 1 1 Fibula 2 1 I Mandible 2 1 1 Carpals 1 1 Clavicle 1 I Maxilla 1 1 Metacarpals 1 I Phalange 1 I

Adult = 25 years of age (as defined by Garri).

Adult osteomyelitis accounted for 2% to 3% of the cases at the Ttibinger clinic, and Garre included 14 illustrative cases of acute osteomyelitis in this age group. He also discussed six patients who had blood- borne multiple osteomyelitis, all of whom had typical signs and features of acute disease. Involvement of the epiphysis with epiphysiolysis and spontaneous fractures secondary to osteomyelitis of long bones was also described in detail.

The remaining manifestation of acute osteomyeli- tis that Garre discussed was the sclerosing nonpuru- lent type. He described this as a form of acute osteomyelitis characterized by a thickening and “ris- ing” of the bone. He provided nine interesting examples and emphasized that this form of acute osteomyelitis began with a typical acute onset and features such as fever, swelling, pain, and thickening of the bones. He stressed that the thickening of the bones occurred without pus formation. Despite this claim, a review of his illustrative material reveals that pus drainage or fistula formation occurred in five of nine patients cited. Two patients died within days of onset of the disease. There was not a single case in which the periosteum was elevated nor was there an example of cortical reduplication.

With the article itself put aside for a moment, it can be said that Garrb as a physician and surgeon was even more remarkable when one analyzes the limitations he worked with. The most striking of these was the total absence of radiographic appara- tus. Riintgen was to discover x-rays 2 years after the publication of Garr&‘s article, which was several years after Garri had gathered the relevant clinical data.‘O Koch discovered the tubercle bacillus in 1882 by which time Garrb had already gathered much of his material.*O Penicillin would not come into clinical use until 50 years later. *’ Another limitation was the poor state of the art of bacterial culturing (anaerobic culturing methods were unavailable). Finally, the knowledge of bone pathology was poor as important discoveries were yet to be made: vitamin D deficiency as the cause of rickets in 19 18; the discovery of osteopetrosis in 1903.2’ Renowned bone pathologists such as H. L. Jaffe, L. Lichtenstein, and D. C. Dahlin were yet to be born.22

Garrt mentioned the jaws in three instances. None of these patients were included in the chronic scle- rosing group. A case involving the mandible demon- strated thickening of the bone adjacent to a right lower first molar, the crown of which had been lost (presumably as a result of caries). This led to what was probably a periosteal reaction with subsequent production of bone. It represented a typical so-called periostitis ossificans, which is simply a form of acute osteomyelitis.

Although Garrb’s article of 1893 contained a vast amount of material, there were many statements ascribed to him that he did not in fact make. Several authors state that Garre outlined a specific type of osteomyelitis that had not been described previous- ly.3,5,7-9 This could not be further from the truth. He made a great effort in describing the features of the subtypes as being representative of acute disease. He proposed no new nomenclature or suggested taxono- my. It has been said that he was the first to describe nonsuppurative osteomyelitis.2s ‘OS 13, I5 One author stated that pus never formed with this condition,14 yet in five of GarrB’s nine illustrative cases pus was evident clinically, and in those cases in which it did not occur, it was strongly suspected to have been

776 Wood et al.

present. Those who say that Garr6 described a chronic nonsuppurative infection must be reminded that chronicity is a relative term. Two of his patients with “chronic” sclerosing osteomyelitis died within days.

The concept that this affliction arises from mild irritation or infection is also false.6, lo, 12. I33 I6 Many of Garrb’s patients were bedridden for months or years. Several died, and complications including meta- physeal lysis and pathologic fractures were well documented. Such complications would not be expected from mild infection.

Another frequent statement is that the disease most often involved the anterior surface of the tibia.4.6.y.‘i This is not true; the femur was the most frequent site for both patients with the chronic sclerosing form and all patients in Garrb’s series (Table II). One author did correctly state that the femur was the most frequent locale.‘*

A favorite claim is that new periosteal bone is produced by chronic sclerosing osteomyeli- tis.2,h. ‘, 9, 12-14. I63 I9 While this is likely the case, Garrb did not say that it was the reason for the increase in bone girth, nor did he ever mention periostitis, periosteal duplication, or onion skinning. He did refer to periostitis associated with syphilis in the introduction to his article. Claims such as “charac- teristic histologic material”16 and “specific radiologic picture “6. I5 are ridiculous when one considers that he did not have pathologic specimens for microscopic examination, nor were any radiographs taken.

Some claims in the dental literature are sound, such as the claim that the lesion causes osseous expansion2 or that pus is associated with this lesion.18 Others have questioned the use of this eponym.”

With the term Garri’s osteomyelitis being unac- ceptable, criteria for its diagnosis2 must also be discarded. The criteria of Eversole are ill conceived in light of the material Garrt: actually presented in his thesis. They may be applicable to the condition known as periostitis ossificans or proliferative perio- stitis, but these disorders all represent simple osteo- myelitis that happens to have a predominant perio- steal reaction.

Periosteal reaction is a normal accompaniment in some cases of osteomyelitis. Its production is analo- gous to the process of involucrum formation in osteomyelitis of the long bones and should be afforded no special name or disease subtype. There likely is a true form of sclerosing osteomyelitis of the jaws, capably described by Jacobsson, but this bears no resemblance to so-called GarrZs osteomy- elitis in any of its forms.

CONCLUSIONS

1. Garr6 did not describe the typical radiologic, histologic, or bacteriologic features of acute oeteo- myelitis since these materials were not available to him.

2. Chronic sclerosing osteomyelitis was never out- lined by Cart+. He really described recalcitrant osteomyelitis, which was essentially untreatable at’ that time.

3. The term Garrk’s osteomyelitis should not be used.

The authors express their sincere thanks to Ms. S. Roberts and Mrs. F. Du Toit for preparation of the manuscript; to Mr. M. Jooste and Ms. D. Truter of the photography department; and to Mr. A. Louw of the graphic arts department of the Faculty of Dentistry, University of Stellenbosch.

REFERENCES

1.

2.

3.

4.

5.

6.

1

8.

9.

10.

Il.

12.

13.

14.

IS.

16.

17.

18.

Garrt C. Ueber besondere Formen und Folgezusttinde der akuten infektiiisen Osteomyelitis. Beitr z klin Chir 1893;10:241-98. Eversole LR, Leider AS, Corwin JD, Karian BK. Prolifera- tive periostitis of GarrC: its differentiation from other neoper- iostoses. J Oral Surg 1979;37:725-31. Mattison GD, Gould AR, George DI, Neb JL, Garr?s osteomyelitis of the mandible: the role of endodontic therapy in patient management. J Endod 1981;12:559-64. Perriman A, Uthman A. Periostitis ossificans. Br J Oral Surg 1972;10:21 l-6. Batcheldor GD, Ciansanti JS, Hibbard ED, Waldron CA. Garrk’s osteomyelitis of the jaws: a review and report of two cases. J Am Dent Assoc 1973;87:892-7. Ellis DJ, Winslow JR, Indovina AA. GarrB’s osteomyelitis of the mandible. ORAL SURG ORAL MED ORAL PATHOL 1977;44: 183-9. Smith SN, Farman AG: Osteomyelitis with proliferative periostitis (GarrZs osteomyelitis). ORAL SURG ORAL MED ORAL PATHOL 1977;43:315-8. Eisenbud L, Miller J, Roberts IL: Garrk’s proliferative periostitis occurring simultaneously in four quadrants of the jaws. ORAL SURG ORAL MED ORAL PATHOL 1981;51:172-8. Shafer WG, Hine MK, Levy BM. A textbook of oral pathology. 4th ed. Philadelphia: WB Saunders, 1982:506-8. Goaz PW, White SC. Oral radiology principles and interpre- tation. St. Louis: The CV Mosby Company, 1983:3,421-4. Wood NK, Goaz PW. Differential diagnosis of oral lesions. 2nd ed. St. Louis: The CV Mosby Company, 1980:573-5. Thoma KH: Garrt’s osteomyelitis of the mandible. ORAL SURG ORAL MED ORAL PATHOL 1956;9:444-9. Pell GJ, Shafer WC, Gregory T, Ping RS, Spear LB. Garrk’s osteomyelitis of the mandible. J Oral Surg 1955;13:248-52. Panders AK, Hadders HN. Chronic sclerosing inflammations of the jaw. ORAI SURG ORAL MED ORAL PATHOL 1970;30:396- 12. Monteleone L, Hagy DM, Hernandez A. GarrC’s osteomyeli- tis. J Oral Surg 1962;20:423-3 I. Lichty G, Langlais RP, Aufdemorte T. Garrt’s osteomyelitis. ORAL SURG ORAL MED ORAL PATHOL 1980;50:309-13. Langland 0, Langlais RP, Morris CR. Principles and practice of panoramic radiology. Philadelphia: WB Saunders, 1982:355-6. Worth HM. Principles and practice of oral radiologic inter-

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pretation. Chicago: Year Book Medical Publishers, 1963:231- 3.

19. Pindborg JJ, Hjorting-Hansen E. Atlas of diseases of the jaws. Copenhagen: Munksgaard, 1974: 156-7.

20. Henschen F. The history of diseases. London: Longmans, 1962:96-l.

21. Schmidt JE. Medical discoveries, who and when. Springfield, Illinois: Charles C Thomas, 1959.

22. Long ER. A history of american pathology. Springfield, Illinois: Charles C Thomas, 1962:360-l, 382.

Reprint requests to: Dr. R.E. Wood Department of Maxillofacial Radiology Faculty of Dentistry University of Stellenbosch Private bag Xl, Tygerberg 7505 Republic of South Africa

23. Jacobsson S. Diffuse sclerosing osteomyelitis of the mandible. Int J Oral Surg 1984;13:363-85.