pneumocystis carinii pneumonia associated with hypogammaglobulinÆmia responding to pentamidine
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On Feb. 28, 1961, three months after subtotal gastrectomy,the patient was readmitted to hospital with haematemesis andmelxna. This time the attacks of abdominal pain were evenmore severe than before, and on account of a supposed jejunalulcer or perforation a second operation had to be performed onMarch 11. In the periduodenal region, enlarged lymph-nodes,as well as a large inflammatory mass, were found in the regionof the retrocolic gastrojejunal anastomosis. A cavity in themass communicated with a perforated jejunal ulcer. A secondgastric resection was performed, but 16 hours after the opera-tion, despite vigorous treatment with blood, electrolytes, andantibiotics, the patient died.Necropsy revealed an active ulcer in the sutured end of the
duodenum and in the resected jejunum. In the mesentery,around the pancreas there were several enlarged lymph-nodes. The body of the pancreas contained a well-circum-scribed tumour. Microscopic sections of this pancreatictumour confirmed its islet-cell origin. There was evidence oftumour metastases-just as at the first operation-in the
peripancreatic lymph-nodes.D. LEHOCZKY.Postgraduate School of Medicine,
Budapest, Hungary.
PNEUMOCYSTIS CARINII PNEUMONIA
ASSOCIATED WITH HYPOGAMMAGLOBULIN-
ÆMIA RESPONDING TO PENTAMIDINE
T. S. RODGERS.
M. H. K. HAGGIE.
Kent and Canterbury Hospital,Canterbury.
Royal Victoria Hospital,Folkestone.
SIR,-The first recovery from Pneumocystis carinii
pneumonia associated with hypogammaglobulinsemia as aresult of treatment with pentamidine has just been
reported by Marshall et al. We describe here a secondcase. Unfortunately, the condition cannot be diagnosedwith certainty in life without lung biopsy. In our patientthe diagnosis was based on clinical and radiological find-ings that seemed to us to accord closely with thosedescribed in patients in whom the diagnosis had beenconfirmed post mortem.2 3 We were fortunate to hear ofDr. Marshall’s experience and to have his advice.The patient, a boy, was born on June 19, 1963, of healthy
parents. He has two sisters and a brother, whose y-globulin isnormal, and there have been no early deaths in male children.He had developed normally until six weeks before he wasadmitted to hospital on Dec. 13, 1963, weighing 14 lb. 12 oz.(6-7 kg.) with a persistent " cold "-i.e., he was slightlycatarrhal and had a dry cough, but his condition gave nocause for alarm. A week before admission his respiratory ratehad increased; his rectal temperature was between 99° and
100°F, feeding distressed him, he had an infrequent cough, andthere was a tinge of cyanosis. The child was admitted to
hospital as a case of early pneumonia that had failed to respondto oxytetracycline at home. Over the next five days thecyanosis increased, and the child had a tachypnoea of 100 andmore to the minute, with slight intercostal recession, a cough, atemperature seldom above 100°F, and an increasing need foroxygen. The only abnormal signs in the lungs were finecrepitations at both bases and extending into the axilloc. Thesevaried from day to day. The chest X-ray showed widespread,confluent, fluffy opacities in striking contrast to the paucity ofphysical signs in the lungs. The heart was normal in size, andthere was no clinical or electrocardiographic evidence of acongenital cardiac lesion.
Investigations.-The white-blood-cell count was 22,400 perc.mm. with neutrophils 5%, eosinophils 2%, and lymphocytes93%. Later counts varied from 13,000 to 23,000 with adifferential averaging 25% neutrophils and 75% lymphocytes.Trypsin was present in the stool to a dilution of 1 /320, and thefingerprint sweat test was normal. A throat swab grew entero-cocci and Streptococcus viridans; repeated gastric aspirationrevealed no tubercle bacilli and subsequent cultures weresterile. The Mantoux was negative at 111000.1. Marshall, W. C., Weston, H. J., Bodian, M. Arch. Dis. Childh. 1964,
39, 18.2. Bird, T., Thomson, J. Lancet, 1957, i, 59.3. McKay, E., Richardson, J. ibid. 1959, ii, 713.
Treatment and subsequent course.-There was no response topenicillin, streptomycin, isoniazid, erythromycin, cloxacillin, orfusidic acid, which were tried at various times. The cyanosisbecame continuous, and respiratory distress so severe as to
necessitate tube feeding. At this stage, one of us (T. S. R.)suggested the diagnosis of hypogammaglobulinxmia and
pneumocystis infection. The zinc-sulphate turbidity was lowat 1’5 units, and electrophoresis showed absence of y-globulin,Even after the administration of 1750 mg. of y-globulin overthree days the serum level was only 100 mg. per 100 ml.-alevel acceptable for inclusion in the Medical Research Counciltrial series.4 The patient received 170 mg. y-globulin weekly(0-025 g. per kg. body-weight), and antibiotics were con.
tinued; but for the next month he remained deeply cyanosedexcept when asleep in the oxygen tent, the slightest move-ment precipitating cyanosis. The temperature was seldomabove 100°F, the respiratory rate reached 80 to 110 to theminute, and the boy’s fingers and toes gradually becameclubbed. The anoxic capillary dilatation was so gross that thepulse could be recorded by simply feeling the tip of the child’sfinger or toe. -
At this stage, twelve weeks after the onset of the prodromalsymptoms, and six weeks after admission to hospital, treatmentwith pentamidine isethionate, as recommended by Marshall etal./ was begun. The boy was given 28 mg. a day by intra-muscular injection for ten days (4 mg. per kg.). At first therewas no change, but a week after the end of treatment his condi-tion began to improve, and after two weeks the crepitations haddisappeared, and he was able to come out of the tent all daywithout cyanosis. Three weeks after treatment had been
stopped, the chest X-ray showed improvement, and by thefifth week the child was clinically cured and ready for home.
NEONATAL STAPHYLOCOCCAL INFECTION
SiRjŃThompson et al. 5 and Williams have reportedthat neonatal staphylococcal disease is more frequent inmales than in females. This increased frequency is morepronounced in skin disease. Thompson et al. andGezon et al. have hinted that this may be related to
circumcision, which was undertaken in 95% of the maleinfants in their series.
Surveys in Geelong of staphylococcal infection in the new-born have already been reported. 9 10 Now a review of staphy-lococcal disease in the 14,717 infants born between July, 1957,and June, 1963, has shown the following:
The sex difference is of particular interest as a total prohi-bition of infant circumcision was instituted in this hospital inOctober, 1956, owing to the then alarmingly high incidence ofstaphylococcal disease in the newborn. This ban was notlifted until August, 1963. Consequently, between July, 1957,and June, 1963, no circumcision was performed in this hos-pital on males less than 2 weeks of age, and less than 1% ofmales were circumcised before 6 weeks of age.
Further analysis of staphylococcal disease in these 14,717infants showed that the incidence of staphylococcal diseasewas consistently higher in males, irrespective of their age:
Unlike Thompson et al. we have found no sex difference in4. Soothill, J. F. Proc. R. Soc. Med. 1962, 55, 395.5. Thompson, D. J., Gezon, H. M., Hatch, T. F., Rycheck, R. R., Rogers,
K. D. New Engl. J. Med. 1963, 269, 337.6. Thompson, D. J., Gezon, H. M., Hatch, T. F., Rycheck, R. R., Rogers;
K. D. J. Pediat. 1963, 63, 869.7. Williams, R. E. O. Lancet, Feb 1, 1964, p. 274.8. Gezon, H. M., Thompson, D. J., Rogers, K. D., Hatch, T. F., Taylor,
P. M. New Engl. J. Med. 1964, 270, 379.9. Plueckhahn, V. D. Brit. med. J. 1961, ii, 779.
10. Plueckhahn, V. D., Banks, J. Med. J. Aust. 1963, ii, 519.