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KiRKPATRICK, M.D. [Reprinted from the TreiiMciioiis of the Associatiop of American Physicians, 1895.] MON'TREAL, 1896. / % A CASE OF MADURA FOOT DISEASE. (Mycetoma Pkuis, Ociiroid Varikty.) By J. GEORGE ADAMI. M.A., M.D., M.R.C.S. Eno., I-ATE FEI-LOW OF JESUS COIXKOE, CAMDHIDOE ; PBOKESSOU OF PATICOI.OUV IN THE MCOII.I, UNIVERSITY, MONTREAL ; ANIJ R. C. KIRKPATRICK. MJ)., ASSISTANT SUliOEON TO THE MONTREAL HENEllAL HOSPITAL. Intehest in this curious disease, mycetoma pedis, lias been stimu- lated during the hist three or four years by the researches of Kan- thack.'\ " and of Boyce and Surveyor','',", ' in Enghmd, of Vincent in Algeria,'", '" and of Rassini ', "' in Italy ; and, whereas, it used to be considered as essentially a local disease, occurring only in India, and mainly in the Madura district, and in the Province of Scinde, we now know that it has been met with elsewhere: in Italy, in Algeria, and (I have this on the authority of Dr. Kunthack) in Germany. The case we record is, we believe, the first on this continent in which we are dealing with the true disease in a patient who has passed all his life in America. We say this with some hesitancy, for there are not a few features in connection with Kemper's case," published nineteen years ago, which render it probable that the appearances described were those of true Madura foot. Nevertheless, in more than one particular the short account given introduces variations from the typical symptomatology. To these we shall recur later. Another case occurring in the practice of Dr. Charles T. Parkes, of Chicago (quoted by Shoemaker), is invalidated in this connection by the fact that the patient had resided for many years in India. The following notes of this case were obtained by one of us (R. C. K.) with some little difficulty from the dense and taciturn patient. Xavier Lacompte, a French-Canadian, aged twenty-one years, was born in Montreal, and has always lived there, save for five years, between the ages of \ 2 A DA Ml, twelve un.l seventeen, i-asse.l in the I'rovince of Ontario. He lui.l never been out of C'annda. Hi-* imrents, three brotlierM, and four HJHterH are livinR and in perfeet health. There U no history of tul.enulosis in the hunily. When he was eleven years old a bluish spot appeared on the inner side of the riLrht foot; this grmlually inereased in si/.e until it was as large as a five-cent piece. One day. while walking, he struck the foot, breaking open the spot, and from it a little bloo.l escaped ; after this the spot disappeared. A few months later there appeared on the sole of the foot below the first and secon.] meta- tarsals what the imtient .lescribed as " un bouton de chair "-a button o» tlesh. After a time this was removed, leaving a little hole which ultimately healed. Three years later a similar growth api-eared on the dorsum of the foot, directly opposite to where the other nodule had been on the sole. Two yt-ars later he struck the f<.ot with an axe, bruising it severely; from that time it remained swollen and lender. Kio. 1. Apparently the patient never suffered severe pain, and he was able to use the foot and to walk until the autumn of 181)8, when the condition became aggravaied, button-like nodules developing all over the foot. As shown by the illustrations (Figs. 1 and 2), these buttons were in the main discrete but situ- ated in groups, of which the most extensive was upon the dorsum of the toot, from below the external malleolus downward along the outer side; another A CASK OF MAM'UA FOOT DIHKA.SK. 8 group extended over tlie nu'tiitiirsulu of the hiillux and stecoiid toe, imnHini; into 11 group iiboiit tlie lm«e« of the first tliree toen. There were otlier f^roiip.s Bitimted over the internal maileoluM, in the insteji and over wliat, fortheHuive of brevity, may 1)6 termed tlie pud of the seond and third toe;*. The iso- lated buttoiiH, and they were the majority, were of the size of a pea or some- what larger. Here and there tw(t or three such buttons were confluent. Where they were isolateil they were sub-pedunculated, the circumference Km. 1'. beyond the base being greater than at the base. Each of them represented a cutaneous overgrowth of low vitality, ])inicish or bluish-pink in color, formed around the opening of a sinus. Over the middle portion of the doraum of the foot, and to a less extent elsewhere, small cicatrices were observable, due evidently to breaking down and healing with closure of the sinuses. The.se sinuses were very extensive ; in fact the foot was riddled with them, and, as I found upon examination after removal of the foot, they passed deeply in all directions, and here and there led to carious bone, The muscles of the leg were atrophied, so that the leg contrasted strongly with the foot, which, from the ankle to the offset of the toes, was greatly swollen. Between these two regions there had been extensive destruction of the ligaments, for the bones could be felt freely moving on one another. How extensively the bones themselves had become afT'ected was demonstrated 4 A I) A M I , by a mounted preparation of tlie bones of the foot. Tliere was an extreme condition ofrarefyingosteitiiof all the tarsal and metatarsal bones, with caries. The bone destruction was "greatest in connection with the cuboid and external cuneiform. The articular surfaces of the tarso-metatarsal serle.s of joints had entirely disappeared save those of the spposed internal cuneiform and first met.itarsal. Towanl the periphery of the aU'ected ref^ion — on either side of the OS calcis below, on the cutaneous surfaces of the «caphoiil and internal cuneiform, atid on the i)ro.\imal halves of the four outer metatarsals — were delicate, fairly long radiating osteophytes. Tliere was a second smiiU area of advanced rarefying osteitis art'ecting the proximal ends of the three middle phalanges. The disturbance in this area tallied with the history given of the first external appearances of the typical mycetoma buttons, while the con- dition of the rareliod bone with its small cysts or ]iits hollowed out in it tal- lied wholly with the description and figures of the Indian disease given by Vandyke Carter. The foot being absolutely useless. Dr. Ivirk|)atrick: removed it in June, 1S94, performing a Syine's operation at the junction of the middle and lower thirds of the leg. Recovery was complicated by an attack of pleurisy, but eventu- ally the patient left the hospital with a well-healed stumj). The specimen, we greatly regret to state, arrived at the I'athologlcal Labor- atory at McGill University at a time when one of us (.V.) was preparing to de- pan for the summer, and in the stress of work was [)laced in preservation fluid before he had time to study it. When, two days later, he lieard that it was to be brought before the Montreal ^ledico-Chirurgical Society, he examined it hastily, and was immediately struck by its resemblance to s|)Ocimens of Ma- dura foot disease. There were the same fleshy buttons with sinuses, and upon pressure upon them n thin pus exuded in which were granules of a yellowish-gray color, of various sizes up to that of a pin-head, and yet larger. None as large as a pea, was obtained, but some as large as two or three hempseeds moulded together. The larger masses were formed of conglomerations of smn'ler particles. Upon studying the fresh discharge under the microscope, and again upon examination of .sections, the structure of these bodies could be easily seen. They were identical i:; general appearance with actinomyces, forming lobate- reniform masses, with central dense mycelium and a radiate arrangement of filaments or clubs around the periphery. These clubs, however, were larger than those of actinomyces ; in the fresh exudate they were easily distinguish- able by the low power of the micro.scope (Zeiss A), while under the high power there was observable a marked tendency for the clubs to bifurcate. The masses were surrounded by leucocytes, and in many cases the collections were so loose that the fungi tended to fall out. In the preserved specimens the clubs underwent considerable shrinking, so that they appeared not nearly so characteristic when seen en masse. Never- theless in these, also, upon breaking up and teasing the masses, the branching could frequently be seen. Here and therein some of the masses long hyphal ■1 Tliere was an extreme .rsiil bones, witli caries, lie ciil)()iil iind external itarsal series of joints nternal cuneiform and rpf^ioii — on either side scaphoid and internal Iter metatarsals — were s a second small area (Is of the three middle h the history given of juttons, while the con- hollowed out in it tal- idian disease given by noved it in June, lSi(4, iddle and lower thirds ' pleurisy, but eventu- imp. le Pathological Labor- .) was |)reparing to de- >d in preservation fluid he heard that it was to ociety, he examined it B to si)Ocimens of Ma- tons with sinuses, and ich were granules of a a pin-head, and yet le as large as two or asses were formed of cope, and again upon could be easily seen, lyces, forming lobate- uliate arrangement of , however, were larger rere easily distinguish- while under the high he clubs to bifurcate. ly cases the collections insiderable shrinking, seen en masse. Never- masses, the branching de masses long hyphal A CASK OF MADURA FOOT 1>1SKASK. 5 processes tending to bo swollen at their extremities i)rojected far beyond the main clump of rays, in these, while there was evidence of granulation and uneven .staining, I could detect no sign of transverse cleavage. In one specimen, however, I came across ample evidence of such, and it is neces- sary that the fact should mentioned. This was in a specimen of a large mass obtained from a sinus when first ! examined the foot. This was ])laeed in glycerin and surrounded with Canada balsam, and has formed one ol the best and most typical examples of the actinomyces-like appearances tliiit I have mounted. (Fig. 3.) Within tiie Last few days I have teased and faintly stained a portion of this mass, and to my great surprise have found that some of the separated liyphie show the clearest evidence of being formed of irregular joints of varying length and very varying breadth, some of the .segments being oval, and, in fact, almost spherical, although in general they are elongated. Of such segmentatiop, as i have said, not a trace is ob.serv- V\v.. :!. ^ able in the peripheral /one of the intact m.ass, and F am led to conclude either that these segmented hypli;e are an intrusion, that the sinus contained more than one form of fungus, or that in the deeper layers of the rayed masses true .segmentation does exi.st, as Carter originally noted anii. Kiiiillmck, as Ids iniiicr I'liMirly sliows, iii'ViT al,tcin|iU'cl 111 pnivc Uiat Uie iiiadiin' liiiinM-i is iiK'iilical witli actiuomycos liDininis or bovis only Ibat il is an actiiiorayces, a ray luugiis, belonging lo Ihe sami; groui> as llie Europeun ray fungus. A CASE OF MADl'RA FOOT DISKA.SK. »1^/ in the tissues. Tliis does not correspond -iatisfactorily witli the diagrams given by Kantliack and Hewlett, or with the apijeurances seen in our case. Insteail of a peripheral zone of chihlied rays, the iliagrain shows fitie broken fdaments, some of whicli radiate outward for a long distance between the cells of the mycetoma tubercle. Vet Vincent gives the usual clinical history and symptoms, anil his de- scription of the appearance of the grains obtained from the discharge out of the sinuses is fairly well in accord with what we have noted in our specimens. It is possible, therefore, that N'initent has figured an atypical and far advanceil mass rather than one that is typical. To sum up, the descriptions given by various oljservers are so con- traid other fungi have a tendency to assume a rayed <:rowtli within the or'^anism.'' The probaliilitv is that we have to deal with a series of actinomycoses, and it is a (picstion which the future must solve, as to how many fungi, of the more usual type — not only streptothrices. but also hyphomycetes — are capable of producing the clinical appeaninces (if Madura foot or mycetoma pedi.s. Ibissini's case, for example, does not wholly c(^nform with the classical descriptions of the black variety, nor again does Hewlett's third case.'- We have pointed out that in snme respects Vincent's case presents unusual features, and if further research in our own case brings to light addi- tional examples of true segmentation of the hyplne then it will have to be divided off from the rest. In its slowly progressive clmracter, its long divation. the absence of constitutional disturbance, and little local pain, in extern:'' appearance and the development of innumerable sinuses with affections of the bones of tlie foot, in the presence of characteristic large granules, recalling the appearance of enlarged actinomycotic masses — in all these particulars it will be seen that our case conforms remarkably ■with the disease as found in India. Kemper's case, on the otlier hand, presents many departures from the usual type. In it the patient, a native of Ohio, aged twenty-five years, suffered from a swollen foot for three montlis before any external lesion was manifest. Then several blebs the size of a split pea made their appearance. When first seen by Kem[)er there were five or six of these, about half an inch in diameter. In their centres a round opening existed with well-(lefined borders, and out of the sinuses exuded a glairy white fluid, resembling white of an egg and extremely offensive. These eventually coalesced into one large ulcer. Later other ulcers appeared. There was intense pain, so that the patient gained very little sleep. The foot was removed six montlis after the first swelling showed itself. Examination after removal revealed a large ulcer over the inner i ■fan A CASE OK MADUHA FOOT DISKASK. 9 side of tlio toot, wliose surf'iiec was (lepressoil (inarter of an iru-li Iicl')\v the jfoneral surface of tlie oriraii. Tliis was covereil hv a white, friahle, (lufty substance, appearing to the naked eye like mould or fungus. A probe could be passed into several sinuses opening into tlie ulcer. These sinuses containeil luiisses of tlie same iiurty material as wiis found upon the surfaces of the ulcers, and, in addition, yel- lowish, highly refractile bodies, forming muU»erry-liko groups. The sinuses opened into cavernous spaces within the substance of the foot. These were filled with white tubercles. It will be seen that this case of Kemper and Jameson varies from the usual clinical history and symptomatology in its short duration — six months — in the absence of any description of the very charac- teristic buttons, in the fact that redness, swelling, and tenderness of the foot preceded any recorded external inflammatory manifestation: and again in the fact that ulceration appears to have been the main pro- cess, and that the ulcers were covered with white, fluffy, mould-like sub- stance. This last point together with the description of blebs rather than buttons are points which I thiidv are unlike what would be de- scribed in connection with the true disease. litHMOOKAI'HY 01'' THR MORE RKCENT AN'Il IMl'ORIAXT 1>A1'KRS ON MYCKTOMA. I. Il't.ixiiii : An'li. [ic.T lo Soieiizo >tLMliclu', xii.. No. I'l. p. ;',(i',l nilistiaul in lllil. f. liiikt., |ss«, iv. p. ('m2). 2. Bocano: Imliiiii Mod. Kuconl, I'iilciuiii, IMi::. p. 1. ;'. TlU' l.aiicfl, lMi:l, ii. p. T'.IT. ■1. ISoi/re and SuiTtiinr : I'rocucdiiigs Kuyiil Soc. I.iiihIdii, IMi:',, vol. liii, [i. 111). .'i. Tmiis. I'lUh. Soc. London, IsiM, v. : iilso l,i\ncut, .\iiril JJ. 1^'.«. p. 'J:32. (i. Hnijec : Joiirniil of I'lUliolnuy. IWl, i. p. Ui;l. 7. HyKionisclie Kniidscliau, IMJI, iv. No. IJ. S, Caiier, WiikIi/I;':: (in MvicinMiii, London dliurchilli, 1-^71. !). Uonibiiy Mod. iind I'liys. Soo., ISSH, ix. (New .Series) p. SO. 10. Genii/ (iiid \'iiirnil: Anniil.s de LM'nniitoL et de Sypliilonraphio, Paris, 1^;):!, iii. p. .'i77. n. Ilewktl : The Lancot, Jnly J, \x'.n. VI Trans. Patli. .Soo. London, isu;!, .xliv. p. 171!. l:{. Kaiillinck : Tlie Lancet, .lunnary 'J:!, l^'.iJ, i. \k 1'.i-'>. II. Journal of I'ailioloKy, I>*ii:i, i. p. li'i. I'l. KeiniiT itiid Jiniimiii : Anierioaii Practitioner. Se|iteniljer. 1>'7(; dpiotod by Imliring. Skin iJiseasos). l(i. K'lhiicr: Arcli. f. Uerniat. iind Sypli., IS'.d. p. 81;!. 17. .S7(.(/|, T. .v.: .Meil. Reporter, (.'alcntta, IHIW, ii. p. 'Jij. IS. Vinci III : Annals do I'lnst. I'astoiir, ls'.ll. viii. p. l'.".t.