A PROBABLE CASE OF PRIMARY TUBERCULOSIS O F THE LACRYMAL SAC

By A. H u g g e T t , Stockholm.*)

Acute and chronic inflammations of the lacrymal sac can, in individual cases, be caused by tuberculosis. I t has, how- ever, been difficult to get any decided idea from the literature dealing with the subject as to how great the frequency is on account of the highly varying values attributed to it in dif- ferent collations. Elliot, for instance, found no cases of it at all in 235 lacrymal sacs, Bribak found 2 in 16, Rollet 8 in 100, Hertel3 in 43 and Axenfeld 13 in 400. These collations are all from older times, and it was only in a few of the cases that tubercle bacilli could be manifested. Instead the diagnosis has been made on the clinical or pathoanatomical picture which, of course, can 'be misleading in certain cases.

As Fage pointed out it is necessary to differentiate a se- condary tuberculosis in connection with other tuberculous diseases of the body such as for example visceral tuberculosis or local tuberculosis in the face or nose from a primary tuberculosis of the lacrymal sac. Inflammations of the lacry- ma1 sac in visceral tuberculosis and local tuberculosis in the face, especially lupus, are without comparison the most usual whereas a primary tuberculosis of the lacrymal sac seems to be very unusual. Many of the cases in the extensive collations mentioned above are probably secondary, but there are nu- merous separate cases of this nature that have been reported as well (see amongst others, Haab, Poulard, Grobe, Chappi,

') Received April 1%h, 1951. 22*

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Tavernier, and Baranger). Axenfeld and Bribak have both made an important indication in respect of the secondary forms i.e. that inflammations of the lacrymal sac in facial lupus are often non tuberculous. Instead they are due to a secondary chronic inflammation on account of the stoppage of ductus nasolacrymalis caused by the specific changes in the nasal mucous membrane. In those cases where it has really been possible to show that the inflammation of the lacrymal sac in connection with lupus was tuberculous, the opinions as to where the primary lesion was placed are also divided. Rollet is of the opinion that the dacryocystitis is the primary and that changes in the nasal mucous membrane are secondary to it whilst many other authors such as Morax and Cabouche think that nasal changes are primary.

A primary tuberculosis of the lacrymal sac is, as men- tioned, more unusual than the secondary forms, and yet all of Axenfeld’s 13 cases were primary. Other suchlike cases have been reported by Rollet, 1 case, Bribak, 2 cases and Wittich, 1 case. Poulard has 3 cases of dacryocystitis which he con- siders as being of the primary tuberculous type. In all of these, however, even the nasal mucous membrane was affected and the same thing applies to several other cases reported from French sources. In regard to the French cases, Valikre- Vialeix also emphasizes that the French term ))fuberculose pr imit i fe~ should for the most part include cases of endo- genous tuberculosis of the lacrymal sac, and only in very rare cases a true exogenous primary complex. If one disregards certain of the French cases, there are even others dating back to a time when one had not the same possibilities as now to preclude other tuberculous lesions in the body (as a matter of fact many of the cases were reported before the time of Roentgen), for which reason it is extremely uncertain how many cases have in reality been primary.

Are there, then, any possibilities of differentiating just from the clinical picture a tuberculous, and more especially a primary tuberculous inflammation of the lacrymal sac from other inflammatory changes in the lacrymal sac? The majority of investigators seem to have answered this question in the affirmative, and have formed a relatively typical picture of

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tuberculous inflammation of the lacrymal sac. The character- istic quality seems to be an ectasia of the lacrymal sac or a tumor like bulging out of it, but with a free passage in the lacrymal ducts for flushing. Another typical sign seems to be lacrymal fistulae, and a third one a swelling of the regional lymphatic glands. If a true primary complex exists, the re- gional glands ought always to be swollen, which is especially emphasized by Validre-Vialeix and which is far from being the case in the repdrts of primary tuberculosis of the lacrymal sac that have been published. Poulard, however, is of the opinion that glandular swelling ought not to be so constant an occurrence as it is in conjunctival tuberculosis, but, as stated above, the cases of this author are rather uncertain primary complex ones.

The course of tuberculous dacrocystitis is a long one with the formation of fistulae and scars, and the only radical the- rapy is generally considered as being extirpation of the lacry- ma1 sac followed, if necessary, by a cauterization of the base of the wound. Sallman, however, says that radium treatment will give good results. If the process heals spontaneously it will do so very slowly and with repeated acute outbursts. This means that a dacryocystorhinostomy can very rarely be per- formed and that tuberculous dacryocystitis is generally con- sidered as being a contra indication for this operation (Diaz- Caneja, Hallum, Lyle, Cross, Simpson and Fraser, Aalde and others). For that matter it would seem that no such operation has ever been performed during the acute stage of dacryo- cystitis, and even in regard to a tuberculous dacryocystitis that has healed, I have only been able to find the most in- complete information. It would appear then that de Jaeger has operated on two cases that had lupus with good results, but I have not been able to obtain any detailed descriptions of these two cases. Lupus in the face or mucous membrane of the nose is, for that matter, considered by the majority as being a contra indication (see Dupuy-Dutemps amongst others), and only a very few operate despite acute changes of this nature. (Diaz-Caneja, Averbach and Zuanova) .

In the author’s opinion a bacteriologically and clinically well healed tuberculous dacryocystitis should not be contra

indicative to dacryocystorhinostomy but should rather hold the same position as other forms of stenosis in the lacrymal duct.

This opinion is supported by the termination of t he fol- lowing case that has been under treatment since the autumn of 1949 at the eye clinic of Sabbatsbergs Hospital, Stockholm.

Report of the case: This is the case of a girl who in the autumn of 1949 was 51/2 years

of age and who had no hereditary traits of tuberculosis. On the 19th August, 1949, an elderly man who was an old and trusted friend of the family came on a visit. Shortly afterwards it transpired that his expectorations contained tubercle bacilli although, at the time of his visit, he was unaware of his being ill. The man was only a few hours in the girl's company but he played and talked with her rather much. From this time and until the girl fell ill he did not visit the family. The girl had previously been in good health and was even healthy the following month. A s a matter of fact the family were in good health the whole time. On the 19th October the mother took the girl to see a general practitioner because she had had a temperature of between 38" and 38.5" for five days on end. At this time the girl's throat was slightly reddened and there was a solid and painful gland as big as a hasel nut over the parotic region. No glandular swellings were observed on the neck. The appearance of the region of the lacrymal sac did not, give rise to any special attention. A wet-warming fomentation was applied to the parotic region, and she was sent home. On 1st November she returned, this time with very marked swelling and redness in the region of the lacrymal sac a s well as fistular formation. On this occasion the glands in the mandibular angle were also swollen. As her condition became much worse she was remitted to the ophtnal- mic polyclinic of Sabbatsbergs Hospital on the 5th November. On her arrival we found a swelling as large as a hasel nut correspondiiig to the lower part of the lacrymal sac together'with a non secreting fistulous formation. There was no tenderness 017er the upper part of the lacrymal sac. No resistance was felt when flushing the lower lacrymal point, most of the fluid was pressed downwards to the nose and the flushed liquid was clear. None of the fluid passed nut to the fistula. Relatively solid glands as big as hasel nuts existecl preauricu!arly and in the mandibular angle. The eye, and especially the conjunctival sac, presented normal conditions. The whole wa4 regarded as being a prelacrymal inflammatory process (tumor prae- lacrymalis Rollet, Terson and others) with fistular formation and regional glandular swelling. A further superficial incision was made

whereby a profusion of thin pus ran out. Two days later pus could be pressed out through the lower lacrymal point by exercising pres- sure upon the swollen region, and we began to regard the disease as being a dacryocystitis with glandular swelling. The fluid used for flushing now began to pass out through the fistula wherefore tlie lacrymal ducts were flushed daily with either a preparation of sulfa or penicillin. On account of the lengthy course of the disease, its poor tendency to heal and the increasing glandular swelling, a sus- picion gradually grew that a tuberculous disease was manifest. On the 15th November a Mantoux test was taken which was highly positive for 0.001 mgr. As the mother was still unwilling to allow LhP girl to be taken into the Eye Department of the hospital for treat- ment, the polyclinic flushing with penicillin was continued to all intents and purposes every day. On the 23rd November, however, thr glands showed a tendency to fuse and the girl was admitted to hospital. At that time the ESR was 23 mm in one hour. Two (lags later tests were taken from the fistula but there was no trace of tubercle bacilli to be found in the direct test. A test was made from a fused gland which showed the presence of bacilli. When concen- trating the following day a profusion of tubercle bacilli were ob- tained even from the fistula. Guinea pigs were simultaneously inoculated and later on they showed a positive reaction. Unfor- tunately, however, no determination a s to whether it was a question of human or bovine tuberculosis was ever carried out. The glands were subjected to repeated puncturing during the next few days, and the lacrymal ducts were treated by local flushing as before. On the 28th November the girl was examined a t the Ear Department where a large, sticky adenoid was discovered in the epipharynx and otherwise normal conditions, and especially no signs of tuberculosis of the mucous membrane. A Roentgen picture showed a suspected changed gland in the diffusely congested lower part of the left hilus region. The pulmonal parenchyma showed no changes.

As the inflammatory condition, despite the treatment, showed no improvement, dihydrostreptomycin was administered in doses of 50 mgr 4 times a day, and already one week later the inflammation in the lacrymal sac had healed and the fistula had practically dis- appeared. The girl was then transferred to the tuberculosis wards for treatment of her glands which showed no signs of improved healing during the short streptomycin treatment.

When controlled after discharge from the eye clinic the lacrymal duct has been flushable at times, but usually the flushing solution passed out through the fistula. At odd times in the beginning there was a phlegm floccule in the lacrymal sac but usually the flushing solution was quite clear. A new inoculation experiment was made on guinea pigs with the flushing solution from the fistula in April 1950, but this was negative. A fresh examination of the nose and throat which was made in January 1950 showed that both tonsils

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were papular and slightly reddened similar to the adenoid in the epipharynx. No definite tuberculous changes, however, could be ob- served, and it was not thought necessary to make a test excision in order to preclude tuberculosis. Later examinations of the nose and throat manifested completely normal conditions. Roentgen pictures of the lungs were controlled in February 1950, when one found an unchanged condition with a rounded congestion on the left side which might possibly be caused by a gland.

As the glandular swellings on the neck gradually almost dis- appeared altogether, and the lacrymal flow from the eye was trouble- some, it was decided a t the beginning of November to make a trial with a dacrocystorhinostomy. The operation was performed in ac- cordance with Toti-Kuhnt. A small shrunken lacrymal sac was revealed with a central lumen in connection with both canaliculi lacrymalis. The lower one of these was, however, very congested a t the inlet to the lacrymal sac. The periosteum and bone in the neighbourhood of the lacrymal sac manifestated no morbid changes, and the appearance of the nasal mucous membrane was normal. A part of the shrunken lacrymal sac was cut away and sent for patho- anatomical examination. This showed only an insignificant, chronic, non-specific inflammation, and no signs of tuberculosis. The inner orifice of the inferior canaliculus became more and more congested after the operation and had to be dilated four weeks after it. After three dilations a free passage was obtained, since which a good passage without inflammatory irritation has ensued (the last exa- mination took place six months after the operation). In connection with the operation, an endeavour was made to cultivate tuberculous bacilli from a remaining gland in the neck in order, to find out whether it was a matter of a human or bovine strain. The exudate from the gland, however, proved to be sterile.

We have regarded this case as being a tuberculous primary complex with the primary lesion in the lacrymal sac and with swollen regional lymphatic glands. One might even suppose that the primary lesion had its seat in the tonsils or the pharynx, but the fact that no definite tuberculous changes were manifested in these parts together with the fact that the practitioner who sent the case to us reported that the pre- auricular lymphatic gland was the first to be attacked would not justify this supposition. The absence of changes in the conjunctival sac precludes a conjunctival tuberculosis. As the nasal mucous membrane had been normal the whole time and because no osteitic processes could be discovered, there is nothing which would indicate that it was a tuberculous pro- cess that had been affected from the surroundings. Therefore

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in all probability it is a matter of a tuberculous primary com- plex in the lacrymal sac where one also knows about the oc- casion of exposure which is well within the time limit as well.

A striking feature in the morbid picture is the rapid healing of the dacryocystitis following the administration of dihydro- streptomycin. One cannot entirely preclude that a spontaneous healing could not have taken place in the same manner, but for those who were able to follow the development the whole time there was a striking change from the time of the strepto- mycin treatment. The fact that the glands were scarcely af- fected by this short treatment coincides well with previous experiences in this sphere. The rapid action on the tuberculous inflammation of the lacrymal sac also shows a striking agree- ment with the frequent rapid and good effect of streptomycin in tuberculosis of the mucous membrane of the larynx and bronchi (with regard to the action of streptomycin in these cases see, amongst others, Hinshaw, Bunn, Pfuetze and Ashe, Lincoln and Kirmse). The fact that the specific inflammation in the lacrymal sac healed completely from this treatment is manifested by both the uncomplicated course of the dacryo- cystorhinostomy and the absence of tuberculous changes in the parts of the lacrymal sac that were taken care of.

Summary. In a girl of 5% years of age who had previously been in

good health, a tuberculous dacryocystitis that was in all pro- bability a primary one, manifested itself after she had been exposed for a brief period to tuberculous infection. A rapid healing takes place from streptomycin treatment and the girl was operated on. one year later in accordance with Toti-Kuhnt with good results.

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Bunn: Dis. of Chest 1948 - 14 - 670. Caboche: An. de l'oreille 190.7 - 33 - 321. Chappd: An. d'Oc. 1905 - 133 - 177. Diaz-Caneja: An. d'Oc. 1933 - 170 - 384. Dupuy-Dutemps: An. d'Oc. 1933 - 170 - 361. Elliot: 0. Rev. 1938 - 27 - 33. Fage: A. d'O. 1910 - 30 - 352. Grobe: Diss. Jena 1898. Haab: A. f. 0. 1879 - 25 - 163. Hallum: Am. J. 0. 1949 - 32 - 1197. Hertel: A. f. 0. 1899 - 48 - 21. Hinshaw: J. A. M. A. 1947 - 135 - 6.41. de Jaeger: An. doc. 1935 - 172 - 56. Lincoln a n d K i m s e : Pediatr ics 1950 - 5 - 280. Lyle, Cross, Simpson a n d Fraser: B. J. 0. 1946 - 30 - 102. Morax: A. d'O. 1898 - 18 - 466. Pfuetze a n d Ashe: Dis. of Chest 1948 - 14 - 446. Poulard: A. d'O. 1903 - 23 - 773. Rollet: An. d'Oc. 1900 - 123 - 331. Snllmann: K. M. Aug. 1924 - 73 - 781. Tauernier: These Lille 1897. Validre-Vialeix: Trait6 d'0pht. P a r i s 1939. Witt ich: K. M. Aug. 1913 - 51 - 577.