April 1976
TheJournalofPEDIATRICS
709
An hypothesis on the pathogenesis of Hemophilus influenzae buccal cellulitis
has a strange and hitherto unexplained propensity to localize in the cheek when it infects soft tissue. Two-thirds of reported soft tissue infections have been at this site. Excluding orbital cellulitis, there are 24 cases mentioned in the literature. 1-6 Sixteen involved the cheek, three the neck, three the arm, one the leg, and one the hand. Because bacteremia is a common, and probably constant, accompaniment of infections of the cheek, it has been postulated that bacteremia may be the primary event with localization in buccal soft tissue a secondary one, but no explanation for affinity to this area has been proffered. It has also been speculated that bacteria in the mouth may directly invade the buccal tissues, but characteristically in infants with buccal cellulitis due to Hemophilus there is neither obvious disruption of the oral buccal mucosa nor history Of trauma. In 1972 one of our resident physicians, Dr. Jorge B. Howard, called our attention to the relation of otitis media to buccal cellulitiS in a patient from whom H. influenzae type b was culture d by needle aspiration of the inflamed buccal tissue, from tympanocentesis fluid, and f r o m blood. The patient had unilateral ear infection, ipsilateral to the buccal cellulitis. Finding typable Hemophitus in the middle ear fluid was unusual because over 90% of uncomplicated cases of otitis media due to Hemophilus are due to untypable strains. 7 Since then we have paid particular attention to examination of the ears in infants with buccal cellulitis and in all patients have found an associated otitis media. Otitis has not been mentioned in previous reports of H. influenzae buccal cellulitis nor has accompanying meningitis, which we have seen in two Patients. (Alexander's'-' patient with cellulitis of the neck progressed to meningitis.) We have reviewed the records of 27 patients with the diagnosis of buccal ceilulitis due to H. influenzae, hospitalized between 1969 and 1)75, to characterize the clinical findings and to speculate upon the pathogenetic relation of otitiS media to infection of soft tissues of the cheek. Documentation of H. influenzae etiology was based upon HEMOPHILUS
INFLUENZAE
TYPE B
recovery of the organism from blood culture, from aspiration of the lesion, or both. The patients were fi'om three months to 37 months of age with an average of 11 months. There were 14 girls and 13 boys. Fever preceded the recognition of cellulitis in all patients. The infants were examined soon after onset of symptoms with an average of 14 hours (range two to 38 hours) and the mean temperature at the time of admission was 39~ (range 37.6~176 Cellulitis was always unilateral; it involved the right cheek in 15 patients and the left in 12. In most instances the area of cellulitis was described as being poorly demarcated, slightly indurated, and warm with a dusky or bluish-purple color. This characteristic coloration, however, was not always initially apparent. Some infants when first seen were described as having a red, shiny, tense appearance to the cheek but within a few hours it changed to the more typical color. Otitis media was recorded in 20 of the 27 patients (74%). It was unilateral in 13 and bilateral in seven. Otitis was ipsilateral to the buccal cellulitis in all those with unilateral middle ear disease. Diagnostic tympanocentesis was performed in ten patients, and H. influenzae, type b was recovered in all cases. Aspiration of tissue fluid from the border of cellulitis was attempted in 15, and H. influenzae type b was cultured from eight (53%) of the specimens. Blood cultures were obtained from all patients and were positive in 22 (82%). Lumbar puncture was done in 24 patients. Two had positive cultures and cerebrospinal fluid changes consistent with bacterial meningitis. Total white blood counts ranged from 9,500 to 31,000/ mm ~ with a mean of 20,680/mm ~ and in 54% there were more than 10% band forms in the differential count. In general, with the exceptions of otitis media and meningitis, the clinical features of our patients were similar to the 16 previously reported cases in regard to abrupt onset, characteristic coloration with ill-defined borders, and the virtually constant bacteremia. Ever since we were alerted to the association of otitis media with
Vol. 88, No. 4, part 1, pp. 709-710
7 10
Editor's' column
buccal cellulitis, it has been found whenever looked for, We cannot explain the absence of otitis media in previously reported cases but can only speculate that the more dramatic findings in the cheeks may have distracted physicians from careful examination of the ears. This appears to be the situation in our few patients without recorded otitis media. Their medical records do not have detailed descriptions of the appearance of the tympanic membranes. The fact that all Hemophilus influenzae cultured from middle ear aspirations were type b is distinctly unusual. Most otitis media of infancy is due to nontypable Hemophilus ~trains. Further evidence of a possible pathogenetic relationship between otitis media and buccal cellulitis is the fact that whenever ear infection was unilateral it was ipsilateral to the cellulitis. We do not k n o w the reason for association of otitis media and buccal cellulitis but propose that primary infection of the middle ear extends to the buccal soft tissues by way of lymphatic vessels. The lymphatics of the middle ear have not been studied but it is known that there i s a n abundant plexus of lymphatics in the tympanic membrane, and interconnections o f the lymphatic system in the face and neck are extensive and complex. In the absence of any other explanation for the unique predilection of H. influenzae cellulitis for the cheek and our observation that it is almost uniformly associated with
The Journal of Pediatrics April 1976
middle ear infection with type b organisms, the most plausible explanation is that infection of the middle ear extends through lymphatic channels to the buccinator nodes.
John D. Nelson, M.D. Charles M. Ginsburg, M.D. Department of Pediatrics University of Texas Southwestern Medical School 5323 Harry Hines Blvd. Dallas, Texas 75235 REFERENCES 1. Tomic-Karovic K: Der Haemophilus influenzae als Erreger akuter und chronischer inflammatorischer Prozesse, Wien Klin Wochenschr 65:940, 1953. 2. Alexander HE: Infuenza bacillus infections, in Barnett HL, editor: Pediatrics, ed 14, New York, 1968, AppletonCentury-Crofts, Inc, pp 598-600. 3. Green M, Fousek MD: Hemophilus influenzae type b cellulitis, Pediatrics 19:80, 1957. 4. Thilenius OG, and Carter RE: Cellulitis of the leg due to type B Hemophilus influenzae, J PEDIATR54:372, 1959. 5. Feingold M, and Gellis SS: Cellulitis due to Haemophilus influenzae type B, N Engl J Med 272:788, 1965. 6. Rapkin RH; and Bautista G: Hemophilus influenzae cellulifts, Am J Dis Child 124:540,1972. 7. Sell, SHW, and Karzon, DT, editors: Hemophilus influenzae, Nashville, Tennessee, 1973, Vanderbilt University Press.
Erratum. In the December, 1975, issue of THE JOURNALOF PEDIATRICS(87:958, 1975), in the article "Congenital hypothyroidism--signs and symptoms in the newborn period" by David W. Smith, Alan M. Klein, James R. Henderson, and Ntinos C. Myrianthopoulos, the infant in Fig. 1 was eight days old. Erratum. In January, 1976, issue of THE JOURNALOF PEDIATRICS(88:147, 19~6), the article "Leukemia! Is it so awful?" was submitted by Dr. Sorrell L. Wolfson. Erratum. In the February, 1976, issue of THE JOURNALOF PEDIATRICS(88:315, 1976), in the article "Commentary: An appraisal of tobramycin usage in pediatrics" by George H. McCracken, Jr., and John D. Nelson, in the second paragraph under the heading "Toxicity," the dosages for kanamycin and gentamicin have been transposed. The corrected sentence should read: We have just completed four-year follow-up examinations of children who received 15 mg/kg/day kanamycin or 5-7.5 mg/kg/day gentamicin during the neonatal period. Erratum. In the February, 1976, issue of THE JOURNALOF PEDIATRICS(88:363, 1976), in the letter "Chronic versus intermittent phenobarbital therapy" by Richard I. Sakai, the last sentence should read: In order to monitor compliance, the biggest problem encountered in chronic therapy, 9 periodic blood levels should be drawn and should be maintained at a level of at least 15 i~g/ml.
TheJournalofPEDIATRICS
709
An hypothesis on the pathogenesis of Hemophilus influenzae buccal cellulitis
has a strange and hitherto unexplained propensity to localize in the cheek when it infects soft tissue. Two-thirds of reported soft tissue infections have been at this site. Excluding orbital cellulitis, there are 24 cases mentioned in the literature. 1-6 Sixteen involved the cheek, three the neck, three the arm, one the leg, and one the hand. Because bacteremia is a common, and probably constant, accompaniment of infections of the cheek, it has been postulated that bacteremia may be the primary event with localization in buccal soft tissue a secondary one, but no explanation for affinity to this area has been proffered. It has also been speculated that bacteria in the mouth may directly invade the buccal tissues, but characteristically in infants with buccal cellulitis due to Hemophilus there is neither obvious disruption of the oral buccal mucosa nor history Of trauma. In 1972 one of our resident physicians, Dr. Jorge B. Howard, called our attention to the relation of otitis media to buccal cellulitiS in a patient from whom H. influenzae type b was culture d by needle aspiration of the inflamed buccal tissue, from tympanocentesis fluid, and f r o m blood. The patient had unilateral ear infection, ipsilateral to the buccal cellulitis. Finding typable Hemophitus in the middle ear fluid was unusual because over 90% of uncomplicated cases of otitis media due to Hemophilus are due to untypable strains. 7 Since then we have paid particular attention to examination of the ears in infants with buccal cellulitis and in all patients have found an associated otitis media. Otitis has not been mentioned in previous reports of H. influenzae buccal cellulitis nor has accompanying meningitis, which we have seen in two Patients. (Alexander's'-' patient with cellulitis of the neck progressed to meningitis.) We have reviewed the records of 27 patients with the diagnosis of buccal ceilulitis due to H. influenzae, hospitalized between 1969 and 1)75, to characterize the clinical findings and to speculate upon the pathogenetic relation of otitiS media to infection of soft tissues of the cheek. Documentation of H. influenzae etiology was based upon HEMOPHILUS
INFLUENZAE
TYPE B
recovery of the organism from blood culture, from aspiration of the lesion, or both. The patients were fi'om three months to 37 months of age with an average of 11 months. There were 14 girls and 13 boys. Fever preceded the recognition of cellulitis in all patients. The infants were examined soon after onset of symptoms with an average of 14 hours (range two to 38 hours) and the mean temperature at the time of admission was 39~ (range 37.6~176 Cellulitis was always unilateral; it involved the right cheek in 15 patients and the left in 12. In most instances the area of cellulitis was described as being poorly demarcated, slightly indurated, and warm with a dusky or bluish-purple color. This characteristic coloration, however, was not always initially apparent. Some infants when first seen were described as having a red, shiny, tense appearance to the cheek but within a few hours it changed to the more typical color. Otitis media was recorded in 20 of the 27 patients (74%). It was unilateral in 13 and bilateral in seven. Otitis was ipsilateral to the buccal cellulitis in all those with unilateral middle ear disease. Diagnostic tympanocentesis was performed in ten patients, and H. influenzae, type b was recovered in all cases. Aspiration of tissue fluid from the border of cellulitis was attempted in 15, and H. influenzae type b was cultured from eight (53%) of the specimens. Blood cultures were obtained from all patients and were positive in 22 (82%). Lumbar puncture was done in 24 patients. Two had positive cultures and cerebrospinal fluid changes consistent with bacterial meningitis. Total white blood counts ranged from 9,500 to 31,000/ mm ~ with a mean of 20,680/mm ~ and in 54% there were more than 10% band forms in the differential count. In general, with the exceptions of otitis media and meningitis, the clinical features of our patients were similar to the 16 previously reported cases in regard to abrupt onset, characteristic coloration with ill-defined borders, and the virtually constant bacteremia. Ever since we were alerted to the association of otitis media with
Vol. 88, No. 4, part 1, pp. 709-710
7 10
Editor's' column
buccal cellulitis, it has been found whenever looked for, We cannot explain the absence of otitis media in previously reported cases but can only speculate that the more dramatic findings in the cheeks may have distracted physicians from careful examination of the ears. This appears to be the situation in our few patients without recorded otitis media. Their medical records do not have detailed descriptions of the appearance of the tympanic membranes. The fact that all Hemophilus influenzae cultured from middle ear aspirations were type b is distinctly unusual. Most otitis media of infancy is due to nontypable Hemophilus ~trains. Further evidence of a possible pathogenetic relationship between otitis media and buccal cellulitis is the fact that whenever ear infection was unilateral it was ipsilateral to the cellulitis. We do not k n o w the reason for association of otitis media and buccal cellulitis but propose that primary infection of the middle ear extends to the buccal soft tissues by way of lymphatic vessels. The lymphatics of the middle ear have not been studied but it is known that there i s a n abundant plexus of lymphatics in the tympanic membrane, and interconnections o f the lymphatic system in the face and neck are extensive and complex. In the absence of any other explanation for the unique predilection of H. influenzae cellulitis for the cheek and our observation that it is almost uniformly associated with
The Journal of Pediatrics April 1976
middle ear infection with type b organisms, the most plausible explanation is that infection of the middle ear extends through lymphatic channels to the buccinator nodes.
John D. Nelson, M.D. Charles M. Ginsburg, M.D. Department of Pediatrics University of Texas Southwestern Medical School 5323 Harry Hines Blvd. Dallas, Texas 75235 REFERENCES 1. Tomic-Karovic K: Der Haemophilus influenzae als Erreger akuter und chronischer inflammatorischer Prozesse, Wien Klin Wochenschr 65:940, 1953. 2. Alexander HE: Infuenza bacillus infections, in Barnett HL, editor: Pediatrics, ed 14, New York, 1968, AppletonCentury-Crofts, Inc, pp 598-600. 3. Green M, Fousek MD: Hemophilus influenzae type b cellulitis, Pediatrics 19:80, 1957. 4. Thilenius OG, and Carter RE: Cellulitis of the leg due to type B Hemophilus influenzae, J PEDIATR54:372, 1959. 5. Feingold M, and Gellis SS: Cellulitis due to Haemophilus influenzae type B, N Engl J Med 272:788, 1965. 6. Rapkin RH; and Bautista G: Hemophilus influenzae cellulifts, Am J Dis Child 124:540,1972. 7. Sell, SHW, and Karzon, DT, editors: Hemophilus influenzae, Nashville, Tennessee, 1973, Vanderbilt University Press.
Erratum. In the December, 1975, issue of THE JOURNALOF PEDIATRICS(87:958, 1975), in the article "Congenital hypothyroidism--signs and symptoms in the newborn period" by David W. Smith, Alan M. Klein, James R. Henderson, and Ntinos C. Myrianthopoulos, the infant in Fig. 1 was eight days old. Erratum. In January, 1976, issue of THE JOURNALOF PEDIATRICS(88:147, 19~6), the article "Leukemia! Is it so awful?" was submitted by Dr. Sorrell L. Wolfson. Erratum. In the February, 1976, issue of THE JOURNALOF PEDIATRICS(88:315, 1976), in the article "Commentary: An appraisal of tobramycin usage in pediatrics" by George H. McCracken, Jr., and John D. Nelson, in the second paragraph under the heading "Toxicity," the dosages for kanamycin and gentamicin have been transposed. The corrected sentence should read: We have just completed four-year follow-up examinations of children who received 15 mg/kg/day kanamycin or 5-7.5 mg/kg/day gentamicin during the neonatal period. Erratum. In the February, 1976, issue of THE JOURNALOF PEDIATRICS(88:363, 1976), in the letter "Chronic versus intermittent phenobarbital therapy" by Richard I. Sakai, the last sentence should read: In order to monitor compliance, the biggest problem encountered in chronic therapy, 9 periodic blood levels should be drawn and should be maintained at a level of at least 15 i~g/ml.
