TUBERCULOSIS ACQUIRED THROUGH RITUAL CIRCUMCISION

L. EMMETT HOLT, M.D.
Professor of Diseases of Children, Columbia University

NEW YORK

Tuberculosis by direct wound inoculation while not a frequent method of contracting the disease has yet occurred often enough to demonstrate the fact that this is a real danger. In 1887 Willy Meyer collected reports of a number of such instances and added a report of a case of his own of tuberculosis acquired through ritual circumcision. Since that time many additional observations on this subject have been published but only two, so far as I am aware—one by Ware and one by Sara Welk-Kakels—in this country. From inquiries, however, I am led to the opinion that many cases occur which do not find their way into print.

REPORT OF CASE

The report herewith presented is of more than ordinary interest by reason of the virulence of the infection as shown by the wide-spread lesions and the fact that the death occurred at an earlier age than in any case of which I have found a record.

History.-On February 24 of this year there was admitted to the Babies’ Hospital an infant 3 mos of age with an extensive ulceration of the penis. The child was one of seven, four of whom were dead from various acute diseases. The parents were healthy as were the other living children. There was no evidence of syphilis or tuberculosis in the family. The infant at birth was a large child, reported to have weighed 11 pounds, and had been breast-fed up to the date of admission. When eight days old the operation of ritual circumcision had been performed, the blood being sucked up in the usual manner. The wound did not heal properly and at the end of a week suppuration was present and ulceration followed which began in the prepuce and gradually extended, up to the time the child was brought into the hospital. About two weeks after operation swellings of lymph-nodes in the groin were observed and these also had steadily increased in size although showing no tendency to suppuration. The digestion had been good and the nutrition well preserved until the last three weeks, during which there had apparently been a steady loss in weight, refusal of food, and slight fever and cough. There was also a discharge from one ear and from the nose. Four or five days previously a scanty maculovesicular eruption had been observed over the trunk and extremities.

Examination.-A well nourished, normally developed infant was found, weighing 11 � pounds, who did not appear acutely ill. Scattered over the body there were fifteen or twenty lesions which closely resembled those of varicella. Two or three were found on the scalp and several over the neck, shoulders, trunk and thighs. None were seen on the hands or feet and none on the mucous membranes. They were all very similar in character, from 3 to 4 mm. in diameter, vesicular at the periphery and crusted at the center, giving a somewhat umbilicated appearance. A very narrow zone of redness surrounded the lesions. Apparently there was no itching as they had not been injured by scratching. Elsewhere the skin, including that of the palms and soles was quite normal. Many r�les were present over both lungs, but there was no signs of consolidation present. The heart was normal. The abdomen was moderately distended. The spleen was much enlarged, reaching fully two inches below the costal margin. The liver was also markedly enlarged, its lower border being three inches below the costal margin. In the inguinal region, both sides, was a chain of large nodular swelling about the size of the last phalanx of the little finger. No signs of suppuration were present. The penis showed complete destruction of the skin by ulceration quite to the abdominal wall and to the base from which pus was freely discharging. At the meatus was quite a deep ulceration producing an appearance resembling a slight hypospadias. The tuberculin skin test gave a strongly positive reaction. Tubercle bacilli were found in the discharge from the penis, in the sputum and in scrapings from the cutaneous lesions described. The bacilli from the ulcers of the penis were carefully differentiated from smegma bacilli.

Course of Disease.-The child lived sixteen days after admission. During this period there was a constant temperature usually varying between 99 and 101 F. but occasionally going to 102 or 103. There was progressive weakness, steadily increasing cough and r�les throughout the chest. The glandular swellings in the groin became larger but did not soften. The other external lymph-nodes were not enlarged. The blood examination at date of admission showed a leukocytosis of 16,000.; polymorphonuclears 85 per cent.; lymphocytes 14 per cent.; hemoglobin 65 percent. Just before death the leukocytes increased to 24,000 and the polymorphonuclears to 92 per cent. No cerebral symptoms were present, but there was considerable restlessness and lumbar puncture was made two days before death. A clear fluid under normal pressure was obtained which gave a faint globulin reaction and showed an excess of cells-twenty to the cubic millimeter. The skin lesions present on admission dried up slightly during the period of observation but no new ones appeared. Death occurred from exhaustion.

Necropsy.-Body well nourished, the lesions on the skin still showing.

Brain: Three or four military tubercles were seen over the base and a few over the convexity, but the ventricles were not distended and no evidences of meningitis were present.

Lungs: The pleura was not adherent; both lungs were everywhere studded with military tubercles and small tuberculous nodules. Considerable bronchitis but very little pneumonia was present. The lungs looked much as if they had been infected by a syringeful of tubercle bacilli injected into the trachea. No part of the lungs had escaped. The bronchial and mediastinal lymph-nodes were caseous but not very large, none being over 1� cm. in diameter, and none showed softening.

Heart: A small yellow tubercle was seen on the coronary artery about its center. A tuberculous nodule, 2 mm in diameter, was found in the wall of the right ventricle, and another smaller one in the wall of the right auricle near the entrance of the superior vena cava. This involved the pericardium, endocardium and cardiac muscle and was cheesy.

Abdomen: The parietal peritoneum was studded with military tubercles and tuberculous nodules, especially over the diaphragm. The spleen measured 8 by 4.5 cm., and on the surface showed many tuberculous nodules in moderate numbers, chiefly in the cortex. Both adrenals containeld tuberculous nodules, one of which was caseous. Two similar ones were found in the pancreas. The stomach and duodenum were normal, but throughout the rest of the small intestine were great numbers of tuberculous ulcers some of which extended quite to the peritoneal coat. There were large tuberculous ulcers in the cecum nad throughout the colon, even in the rectum.

The mesenteric lymph-nodes were 3cm. in length and 1.5 cm in breadth. They were caseous but not softened. Extending upward from these was a chain of large caseous retroperitoneal lymph-nodes which followed the iliac arteries and then the aorta. One of the iliac arteries contained a cheesy lymph-node in its adventitia. The tuberculous character of this lesion was confirmed by microscopic examination. Three tubercles were seen on the mucous membrane of the bladder near its base. Both middle ears contained pus.

An opportunity was given to examine the man who had performed the operation. He was a pale, thin, almost emaciated individual, who looked decidedly tuberculous although no physical signs of the disease in his lungs could be detected. In his sputum two acid-fast bacilli were found which looked like tubercle bacilli.

This case seems complete and the evidence of infection through the circumcision wound, conclusive. A healthy child, born of healthy parents, breast-fed, developed local symptoms of infection within a few days after the operation and these persisted, being followed after a few weeks by signs of general infection which continued until death. The lesions found at necropsy point strongly to a spreading of the infection through the lymphatic system beginning from the wound, and afterward to a general blood infection. At death, which occurred when the child was 3 � months old, practically every organ in the body was involved. Specially worthy of note in the lesions are the myocardium; the latter lesion I have seen but once before; also the tuberculides of the skin, a lesion which I believe is often overlooked; it has been observed at the hospital in two other cases during the present season, both patients being young infants.

A search through the medical literature brings to light a considerable number of examples of tuberculosis spread by means of ritual circumcision. In all I have collected with the assistance of Doctors Alan Brown and Stafford McLean, references or reports more or less detailed of forty other cases. These are of sufficient interest to make brief summary desirable.

SUMMARY OF LITERATURE

LINDMANN’S¹ CASES: Two cases. The operation was done in the usual manner, the operator sucking the wound; one child died and the other recovered after a long illness. In both cases tuberculous ulceration occurred followed by caseous degeneration of the inguinal lymph-nodes. Ages of children are not given; the operator died two months after performing the operation, presumably of tuberculosis.

LEHMANN’S² CASES: Ten children with almost identical symptoms infected by the same operator. The operator subsequently died of tuberculosis. The wound was sucked in the usual manner. The early signs were similar-irregular, grayish preputial ulcers which gradually extended. After two or three weeks there was swelling of the inguinal lymph-nodes which is most cases suppurated. No microscopic examinations were made, the diagnosis resting on the clinical symptoms.

ELSENBERG’S³ CASES: One case in full. The first diagnosis made was syphilis. Subsequently the patient developed suppuration of the inguinal lymph-nodes and afterwards erysipelas. Tubercle bacilli were found in the sputum of the operator. The author reports that he has seen three similar cases but gives no details.

EVE’S⁴ CASES: One case in full. Infant 5 months old, admitted with suppurative inguinal adentitis (double) and preputial ulcers. Ritual circumcision had been done on the eighth day. A few weeks afterward inguinal glandular swellings were noticed, which steadily increased. Abscesses weer opened and curetted. Sinuses followed which still were present at 8 months. At 10 months the child was well. Pus from inguinal glandular lymph-nodes injected into guinea pigs produced tuberculosis. Operator died from tuberculosis seven and a half months after the circumcision. He did not suck the wound in this case but merely ejected some wine from his mouth over it. Author refers to another similar case but gives no details.

DEBROVITZ’⁵ CASES: Infant 7 months old had multiple ulcers of the prepuce with extensive ulceration of the meatus. Inguinal lymph-nodes were much swollen. Subsequently they broke down and were incised. At 12 months of age wounds healed; patient was regarded as well. Operator was examined and found to be tuberculous; he rinsed his mouth with red wine before sucking the wound. The writer refers to three other cases but gives no details.

GESCHEIT’S⁶ CASES: Five cases with similar clinical symptoms. Details of one case given. Patient’s age 8 months. Circumcision on the eighth day. Wound healed in ten days except a dorsal preputial ulcer. There was edema of the parts. Inguinal lymph nodes swelled to size of pigeon’s egg. Syphilis was suspected. Antisyphilitic treatment was used without success. All five cases occurred in healthy families. Three children died within six months of meningitis and enteritis; two at 7 and 8 months were living at the time of the report. All showed evidence of a primary local lesion. The operator was “thoroughly tuberculous” and had long been ill. Although he had done many previous operations these were the first infants known to have been infected. The author does not state how the diagnosis was made in his cases.

WILLY MEYER’S⁷ CASE: Infant 12 months old. Family healthy. Only child. Circumcised on eighth day by an old man. At 4 weeks inguinal swellings were noticed and syphilis was suspected. Mecurial treatment was begun. At 4 months ulcer was noted on the frenum. Moderate inguinal adenitis was present. Syphilis was still regarded as the diagnosis and treatment continued. There was no improvement under syphilitic treatment. Inguinal lymph-nodes subsequently broke down and were curetted. Tubercle bacilli in great numbers were found in the pus. Subsequent history not given.

HOFMOKL’S⁸ CASE: Infant, 8 months old. Family healthy. The early symptoms identical with those in the other cases reported. Syphilis suspected and for a long time antisyphilic treatment continued without success. Inguinal lymph-nodes subsequently extirpated; found caseous. Diagnosis established by microscopic examination.

KOLIZEW’S⁹ CASES: Seven cases of tuberculosis of the penis are reported following circumcision by a rabbi suffering from tuberculosis. Two patients died; two made partial recovery; two were lost sight of, and one recovered. In only one of the families was there tuberculosis. Arluck and Winocouroff state that this report led to a movement of far-reaching social effect for the reformation of the performance of ritual circumcision.

WARE’S¹⁰ CASE: Infant, 3 months old. Parents healthy. Ritual circumcision done at one week. Two weeks later inflammation of the prepuce. Swelling of inguinal lymph-nodes, both sides, first seen at 4 weeks. Preputial ulceration with moderate secretion also present. Syphilis suspected. Antisyphilitic instituted but without benefit. Lymph-nodes broke down and were curetted. Tubercle bacilli not found, but microscopic examination of the lymph-nodes showed giant cells and tuberculosis was diagnosticated. Case passed from observation.

WELT-KAKELS’¹¹ CASE: Infant, 6 months old. Healthy parents. Circumcision the ninth day by man presumably tuberculous. Wound sucked. Inguinal adenitis observed at the end of four weeks followed by suppuration. Tubercle bacilli found in the pus. Von Pirquet test positive. Lymph-nodes removed.

At four years of age the child was reported by the writer to be living, but, from physical appearance, was regarded as still suffering from tuberculosis.

J. M. T. FINNEY’S¹² CASE: Infant came under observation “some months after the circumcision” with local ulceration and enlargement of inginal lymph-nodes. Tubercle bacilli in large numbers were obtained from the scrapings. Ulcerated areas were curetted. Inguinal lymph-nodes were excised, both sides. Bacilli were found in the lymph-nodes. Subsequently tuberculosis of the one knee developed. The patient was under observation many years and is reported never to have any other evidence of tuberculosis. Investigation by the family revealed the facto that “several other children,” all of whom had been circumcised by the same man, had been similarly infected. He is stated to have been definitely tuberculous. The parents reported that he spat on a clothe which was used as a dressing for the wound after operation.

ARLUCK AND WINOCOUROFF’S¹³ CASES: Infant of 5� months. Parents healthy. Circumcision on eighth day. Wound was sucked by local ulceration, swelling and breaking down of inguinal lymph-nodes. The child wasted with signs of generalized tuberculosis. The entire penis ulcerated quite to the scrotum. There was great enlargement of inguinal lymph-nodes. Wassemann was negative; also the von Pirquet test, the latter thought to be due to the wide-spread infection. One tubercle bacillus was found in scrapings from the preputial ulcer. Death occurred four days later. Necropsy showed general tuberculosis involving lungs, intestines, spleen, mesenteric and inguinal lymph-nodes. The infection was believed throughout the lymphatic system.

Of the forty-one patients, including my own, sixteen are known to have died; seven are reported as having partially recovered or being scrofulous; in twelve the final results are not given; and only six are stated in the histories to have recovered. The youngest fatal case is that of the patient whose history I have reported. In several cases death has occurred as late as 11 months from tuberculous meningitis. The usual cause of death has been general tuberculosis. In many of the reports several children have been infected by a single operator. Thus, in Lehmann’s cases, all ten of the children were thus infected; Gescheit reports four infants infected by the same operator; Lindmann, two patients, Debrovitz, four patients. In nearly all the reports the fact is stated that the families were free from tuberculosis. As a rule the earliest symptoms of infection have been observed in about a week after the operation. The wound does not heal, but suppuration occurs and ulceration soon follows. The early ulcer may be anywhere on the prepuce but is often on the frenum. It may remain as a localized process or be general. At the end of a second or third week inguinal adenitis develops. In a considerable number of cases it is reported that the lymph-nodes broke down and abscess formed, usually in two or three months after the initial infection. The cases in which early suppuration of the inguinal lymph-nodes took place and which were operated on, either by removal or curetting, were among those in which the results were the best. The symptoms of a wide-spread general infection rarely occurred earlier than the third or fourth month.

The diagnosis has been made in many of the cases by the clinical symptoms and history alone. Some of these were reported before systematic search for the tubercle bacillus in wounds was practiced or modern tuberculin tests were employed. In nearly all of the later cases reported, the diagnosis has been established by the discovery of the bacilli in the inguinal abscesses, sometimes from the preputial ulcers. In the latter situation they must be carefully differentiated from smegma bacilli. That the infection spreads through the lymphatic system seems certain and early removal of the inguinal lymph-nodes would therefore appear to be the most important measure to be employed in checking the extension of the infection. To be successful this must of course be done early.

In a very large proportion of the cases reported the first diagnosis made was syphilis, and the patients were treated for weeks and months by antisyphilitic measures without benefit and with loss of valuable time. It is my own belief that syphilis is less frequently acquired in this manner than is tuberculosis and the latter disease should be first suspected. With the modern means of diagnosis in tuberculosis the early recognition of these cases ought not to be difficult. While the number of reported instances of tuberculosis acquired through circumcision is considerable there must be a much larger number that have never found their way into literature. It is certain also that syphilis has been spread in this manner. These facts lead me to emphasize the statement made by the late Professor Maas, the German surgeon, that “it is the duty of physician to raise his protest against the performance of ritualistic circumcision in every case.”

14 West Fifty-Fifth Street.

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��1. Lindmann : Deutsch. med. Wchnschr., 1883, No. 30, p. 442.
��2. Lehman : Deutsch. med. Wchsthr., 1886, Nos 9 and 13, pp. 144 and 218.
��3. Elserberg : Berl. Klin. Wchnschr., 1886, No. 35, p. 581.
��4. Eve : Lancet, London, Jan 28, 1888.
��5. Debrovitz : Pest. Med.-chir. Presse, 1899, no. 23, p. 529.
��6. Gescheit : Internat. Klin. Rundschau, 1889, No. 23, p. 964.
��7. Meyer : New York med. Presse, June, 1887.
��8. Hofmokl : Wien. Med. Presse, 1886, Nos. 22 and 23, pp. 714 and 750.
��9. Kolizew : Ztschrp d. russ. Gesellsch. D. Volksgsndhtsf�rsorge, 1891; abstr. in Monatsh. F. prakt. Dermat., 1893; xvi; 491.
10. Ware, M. W. : New York Med. Jour., Feb. 26, 1898.
11. Welt-Kakels, Sara : Arch. Pediat., 1909, p. 460.
12. Unpublished report.
13. Arluck and Winocouroff : Beitr. F. Tuberk., 1912, xxii, 341.