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16-Year Remission of Rheumatoid Arthritis After Unusually Vigorous Treat

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16-Year Remission of Rheumatoid Arthritis After Unusually Vigorous Treat Clinical and Experimental Rheumatology 2002; 20: 555-557. CASE REPORT 16-year remission of ABSTRACT vide a source of arthritogenic products. rheumatoid arthritis after This rep o rt describes a remission of Second, it is not necessary to hypothe- rheumatoid arthritis (RA) of 16 years si z e a path o genic bacterial strai n , whi c h unusually vigorous treat- d u rat i o n , ap p a re n t ly caused by the will mostly be transient, for one can ment of closed dental foci extraction of endodontically well-treat - i m agine that norm a l , n o n - p at h oge n i c ed, healthy looking teeth. The only clue and thus persisting commensals in A.C. Breebaart1, J.W.J. that the teeth were contributing to the these closed foci may deliver, by the Bijlsma2, W. van Eden3 disease pathogenesis in this case of RA constant pressure exerted on them, the was that the patient was able to repro - cross-reacting products we are looking 1Department of Ophthalmology, University ducibly induce severe attacks of arthri - for and which induce RA in susceptible of Amsterdam;2Department of Rheumatol- tis after prolonged, heavy pressure on individuals. The possibility of such a ogy and Clinical Immunology, University some of his teeth tre ated with ro o t mechanism is illustrated by the follow- Medical Center Utrecht; 3Department of canal fillings. After extraction, a small ing case report. The patient, a physician Veterinary Medicine, University of Utrecht, pus layer was found to cover the apices h i m s e l f, is also one of the authors The Netherlands. of the clinically healthy looking teeth. (ACB). Adrian C. Breebaart, MD, PhD, Professor The rheumatoid factor (RF) became Emeritus of Ophthalmology; Johannes n egat ive and the patient re m a i n e d Case report W.J. Bijlsma, MD, PhD, Professor of Rheumatology; Willem van Eden, MD, symptom free for the next 16 years. The The patient, a male Caucasian born in PhD, Professor of Immunology. p o s s i ble connections between micro - 1935, was diagnosed with RA at 34 Please address correspondence and organisms in closed dental foci under ye a rs of age. Po lya rt h ritis of both reprint requests to: Prof. Dr. A.C. constant pre s s u re and the ch ro n i c i t y hands (MCPs and PIPs) and wri s t s Breebaart, Offenbachlaan 76, and exacerbations of RA are discussed. were the leading signs, with a positive Heemstede 2102 ZP, The Netherlands. RF (Rose-test 1/128), that lasted from E-mail: [email protected] Introduction 1969 to 1971. Received on October 24, 2001; accepted Rheumatoid arthritis (RA) is a disease Though there was no evidence of RA in in revised form on March 1, 2002. of still unknown etiology. Current hy- his family, there was a familial history © Copyright CLINICAL AND potheses favor a possible autoimmune of joint pains with a striking relation EXPERIMENTAL RHEUMATOLOGY 2002. etiology by the action of bacteria or between teeth and joints. Both the pa- b a c t e rial products mimicking joint tient’s mother and her grandmother on Key words: Rheumatoid arthritis, components after bre a k d own of self- her mother’s side had a phenomenon of tooth, endodontics, focal infection, tolerance. Our ignorance of the loca- joint pains, which could be aggravated autoimmunity, microbiology. tion of such bacteria, if any, and their by pressing some teeth and wh i ch apparent persistence in these locations, could be ‘ c u re d ’ by ex t racting these as indicated by the chronicity of RA, is teeth. still a major drawback of this theory. Once the patient’s attention was drawn The fl o ra of oral cavity or dental to this curious relation, it appeared to plaques has been incri m i n at e d, bu t be a reproducible phenomenon also in without much success. Remembrances his case.Though his teeth were regular- of the ill-fated theory of focal infection ly checked by a dentist he could not still linger in our minds. boast a good set of teeth as 4 had re- To be sure, the case to be described quired treatment with root canal antibi- here also points to teeth as the location otics and jackets, partly because of an of the causative micro-organisms, but accident 10 years prior causing 2 bro- there are essential differences with the ken teeth and partly by loosening of focal infection theory. First, a form of some adjacent teeth. By exe rting a mechanical pressure exerted on bacte- large amount of pressure on these teeth ria lodged in a closed focus is seen as a (best generated by eating hard food like p re re q u i s i t e. Bacteria lining the ora l peanuts, raw carrots and the like) the cavity, within dental plaques or in the patient could in each instance induce an tonsils do not live in a closed niche and ex a c e r b ation of his art h ritis (pain, do not endure mechanical pre s s u re. swelling, warmth and decreased range H oweve r, m i c ro - o rganisms in ro o t of motion of the involved joints) which canals tre ated with fillings or at the started about 1 to 2 hours after pressing apices of teeth with extensive recon- the tooth and lasted for several hours. structions do live in a closed surround- This relation was so constantly repro- ing and endure continuous pre s s u re d u c i ble that a dentist was asked to from grinding teeth which could pro- extract the tooth that was once broken, 555 CASE REPORT 16-year remission of rheumatoid arthritis / A.C. Breebaart et al. had been rep a i red with a cap , a n d It is notewo rt hy that the patient had causative micro-organisms (2) and the which had been treated with antibiotics received 3 inoculations with BCG-vac- sites where they persist for some time in the root canal. The dentist objected cine at 17 years of age, as was then cus- are known. Here, too, it is assumed that on the grounds that the tooth was well tomary for beginning medical students either bacteria or bacterial pro d u c t s treated and did not show, clinically or who had negative Mantoux reactions. A re a ch the joints, mimic body tissue radiologically, any sign of disease. mildly positive Mantoux reaction ulti- components, and in susceptible hosts However, at the patient’s insistence the mately occurred. stimulate immune cells to react with tooth was extracted. It was found to a u t o a n t i gens. A l s o , a u t o i m mune ani- have a thin layer of pus covering the Discussion mal models, especially adjuvant arthri- apex. The affected joints suffered an The striking aspect of the disease in t i s , point to similar mechanisms of ex a c e r b ation of infl a m m ation in the this patient, and the reason for writing c ro s s - re a c t ivity of bacterial pro d u c t s following days – a phenomenon also this case report, was a three-fold rela- with disease critical self-antigens of the ex p e rienced by the pat i e n t ’s mother tion between teeth and joint symptoms. joints (3). Although the re l at i o n s h i p and great grandmother – after which Fi rs t , m e chanical pre s s u re caused an between immunity to heat shock pro- period the arthritis subsided for several exacerbation of the arthritis. This flare- teins (hsp) and arthritis is still under months. The disease once again be- up came about 2 to 3 hours after the study, the model of mycobacteria-in- came active, and again exacerbations of mechanical pressure, lasted for some duced adjuvant arthritis has shown that the baseline arthritis could be induced hours and then subsided.This was a a peculiar mimicry between the hsp60 by pressure on loose teeth. This led to c o n s t a n t ly rep ro d u c i ble phenomenon. molecule of mycobacteria and cartilage the successive extraction of another 3 To our knowledge, such a direct corre- proteoglycan-associated molecules can teeth, with the same result. During this l ation in time between mech a n i c a l lead to induction of arthritis. On the period 1969 - 1971 ‘traditional’ treat- stress on the teeth and exacerbations of other hand, T cell immunity to conserv- ment consisted of salicylates. RA symptoms has not been described. ed non-mimicry epitopes of hsp now From 1971 onward the patient remain- The second re l ationship consisted of has been shown to be related to the pro- ed completely free of symptoms, the similar exacerbations, but lasting a few duction of regulatory T cells having the RF had become negative, and for 16 days, after extraction of the involved capacity to control arthritic inflamma- years no signs or symptoms of joint teeth. The third relationship was remis- tion. While the hsp molecule in isola- problems were present. sion for some months after each extrac- tion tends to promote this form of regu- In 1988 a re c u rrence occurred; the tion, and the complete remission for 16 l at i o n , when it is presented to the hands and wrists were only mildly af- years after the last of the 4 treated and immune system in the context of other fected this time but the shoulders, an- loose teeth had been extracted.
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