British Journal of Dermatology (1980) 103, 375.

Streptococcus milleri causing treatable infectionin perineal hidradenitis suppurativa

A.S.HIGHET, R.E.WARREN*, R.C.D.STAUGHTONf AND S.O.B.ROBERTSDepartments of Dermatology and * Bacteriology, Addenbrooke's Hospital, Cambridge

Accepted for publication 6 March 1980

SUMMARY

Streptococcus milleri was isolated from the active lesions of three patients with perineal hidradenitissuppurativa. In each patient, elimination of this organism by appropriate antibiotic therapy wasaccompanied by marked clinical improvement.

Hidradenitis suppurativa is a chronic relapsing suppurative condition of apocrine gland follicles.Apocrinc glands usually open into pilosebaccous follicles, and occur normally in the skin of the axilla,buttocks, perineum, external genitalia, and breasts, and occasionally in other sites (Montagna, 1962).The pathogencsis of hidradenitis suppurativa is not well understood (Gordon, 1978, Roberts & Rook,1979). The initial event is believed to be occlusion of the mouth of the apocrine gland follicle. Thefrequent presence of grouped comedones in affected areas suggests an underlying structural abnor-mality of the pilosebaccous unit. Inflammatory changes occur deep to the obstruction. In severe casesthe gland is destroyed, and marked induration and scarring ensue with chronic sinus formation. Theimportance of bacterial infection is not clear. Staphylococcus aureus is sometimes isolated, and anae-robes have recently been implicated in axillary hidradenitis suppurativa (Leach et al., 1979).

We report our preliminary findings implicating Streptococcus milleri in three patients with chronicsevere hidradenitis suppurativa of the anogenital region, with an encouraging response to treatment.

CASE REPORTS

Case IClinical details are shown in Table i.

This 32-year-old man was referred for a final dermatological opinion before major surgery (Fig. i).He had suffered from hidradenitis suppurativa of the buttocks and natal cleft for 16 years. For 18months painful abscesses had made sitting virtually impossible, and he had been unable to work for ayear. Profuse purulent discharge rendered him housebound, and he had expressed suicidal intentions.Extensive immunological investigation had failed to reveal any defect of polymorph function or ofhumoral or cellular immunity.

t Present Address: Department of Dermatology, Westminster Hospital, London S.W.r.O007-0963/8o/iooo-0375$02.oo C) 1980 British Association of Dermatologists

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FIGURE I. Buttocks of Case i.

TABLE 2. Previous streptococcal isolates from active lesions of Case r

Date Streptococcal isolate

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FIGURE 2. Vulva and perineum of Case 3,

The unusual severity of the disease prompted careful bacteriological investigation leading to theisolation of Strep, milleri. A review of previous bacterial culture results suggested that this strepto-coceus had previously been repeatedly isolated, but its importance not recognized or its identity notspecifically confirmed (Table 2).

Case 2Details shown in Table 3.

This 32-year-oId man had a 12-year history of hidradenitis suppurativa of the peHanal skin and themedial aspects of both buttocks. There was much malodorous discharge and considerable pain.

Case 3Details shown in Table 4.

The initial manifestations of hidradenitis suppurativa in this 31-year-old woman were recurrentabscesses in and around the breast areolae beginning at the age of 18. Five years later the vulva,groins, perineum, perianal region, and axillae became involved. During the period of our investigation,significant disease activity afFected the vulva and perineum only (Fig. 2).

380 A.S.Highet et al.

None of these three patients gave a family history of hidradenitis suppurativa, and none reported apersonal or a family history of acne of unusual type or severity.

DISCUSSION

Leach ei al. (1979) reviewed their bacteriological findings in fifty-two patients with axillary abscessIn twelve cases, all with axillary hidradenitis suppurativa, anaerobic bacteria, especially Gram-positivecocci, were isolated, but Strep, milleri was found in only one of these. Our experience in three consecu-tive cases of anogenital hidradenitis suppurativa is quite different in that Strep, milleri was isolatedfrom aaive lesions in each patient. If this finding is confirmed, it would have important implicationsfor treatment.

Strep, milleri is a homogeneous biochemically defined taxon of streptococci (Lutticken et ai, 1978).The organism may be /?-, y-, or non-haemoljaic, although use of these terms may disguise the factthat an organism has not been identified to species level. Its growth on primary inoculation is oftendependent on or enhanced by incubation in 5" „ carbon dioxide. It is not an obligate anaerobe and ismetronidazole resistant. It has been termed Peptostreptococcus intermedius in many American papers,which is unfortunate, as the genus Peptostreptococcus is usually considered to be strictly anaerobic andmetronidazole sensitive. When grown on blood agar Strep, milleri often has a characteristic caramelodour. The organism commonly carries Lancefield Group F antigen, although 'minute* strains ofstreptococci carrying the Lancefield Group A, C or G antigens, or no detectable Lancefield Groupantigen, are also included in the taxon. Rapid Lancefield Grouping techniques for Group F strepto-cocci have only recently become commercially available. Only Strep, milleri bearing Group F antigenwere sought in the three patients we describe.

Poole & Wilson (1979) detected Strep, milleri as a commensal in healthy subjects in the throat(3i%)> on extracted teeth (28%), in the vagina (18/;), and in the faeces (15/,,). It is now one of thecommonest isolates from abscesses of brain (de Louvois, 1978), lung (Bartlett & Fincgold, 1972),pleura (Bartlett et al., 1974), liver (Sabbaj, Sutter & Finegold, 1972; Bateman, Eykyn & Phillips,1975) and other intra-abdominal sites (Poole & Wilson, 1976). In addition, it has been isolated inMeleney's synergistic bacterial gangrene (Meleney, 1931) and in our laboratory from patients withotitis media, paronychia, or pilonidal sinuses. There are no recent reports on its resistance to anti-biotics. Our records based on susceptibility testing with discs of tetracycline (10 /jg), erythromycin(5 /jg), clindamycin (2 /(g) and penicillin (i unit) show the incidences of resistance in isolates fromindividual patients to be:

tetracyclines 87 of 240= 36% resistanterythromycin 4 of 160 = 2-5% resistantclindamycin i of 92 = 1% resistantpenicillin o o f 2 4 0 = 0% resistant.

However, as Case i illustrates, benzylpenicillin in high dosage, even when combined with anaminoglycoside, gentamidn, does not seem to be effective in eliminating the organism from thesedeep-seated dermal sinuses. The reason for this is not clear, but it is possible that the drug does notpenetrate lesions well, either because of scarring or because of poor lipid solubility. In streptococcicross-resistance between erythromycin and clindamycin is the rule (Dixon & Lipinski, 1979), althoughresistance to these antibiotics is not common. Susceptibility testing of Gram-positive organisms totrimethoprim is often not reproducible between laboratories and may therefore not be a reliableguide to therapy.

Streptococcus milleri in hidradenitis suppurativa 381

In each of these three cases of perineal hidradenitis suppurativa, at a time of active disease withmarked tenderness and purulent discharge, Strep, milleri was isolated as the predominant or onlybacterium from lesional pus. Case 2 was on no antibiotic when investigated. In each of Cases i and 3the Strep, milleri initially found was resistant to the antibiotics then being given, namely clindamycin(and, of course, metronidazole) in Case i, and doxycycline in Case 3. Administration of an antibioticto which the patient's organism was sensitive was followed by marked improvement in each case; inCase I doxycycline appears to have been the most effective drug, and Case 3 improved on erythro-mycin. It was noted, however, that lesions in Case i subsequently yielded Strep, milleri, a strain nowresistant to tetracyclines, as well as to erythromycin and clindamycin, without clinical relapse.

It is interesting that the best results were in Cases i and 2 in whom Strep, milleri was eliminatedfrom the stool following oral vancomycin (with neomycin to obtain bactericidal synergy and nystatinto prevent candidiasis). In Case 3, the organism was not found in the faeces initially and vancomycinwas not given. This patient relapsed soon after stopping erythromycin and her stool was then foundto contain Strep, milleri. It may be that treatment aimed at eliminating Strep, milleri from the gutshould be given even if the initial stool culture is negative. A simple regimen seems desirable and theagent used should not select for a resistant flora. If subsequent work suggests that elimination ofStrep, milleri from the gut is indeed necessary for effective therapy of hidradenitis suppurativa infectedwith that organism, vancomycin alone may be useful. Our addition of neomycin and nystatin mayprove to be unnecessary. Vaginal carriage may require separate treatment.

FURTHER FINDINGS

Our investigations have now included a further ten patients with active anogenital hidradenitissuppurativa. In five of these ten cases. Strep, milleri was isolated from lesions as the predominantorganism.

CONCLUSION

These preliminary observations suggest that infection with Strep, milleri, a frequent commensal inthe gastrointestinal and female genital tracts, may cause readily treatable acute exacerbations inchronic hidradenitis suppurativa of the anogenital region. The simplest explanation is that the organismgains access to pre-existing sinuses in areas of scarring. We have not studied the possible role ofStrep, milleri in the early stages of hidradenitis suppurativa.

Routine laboratory methods may fail to provide accurate identification of Strep, milleri, andcollaboration between clinician and bacteriologist is advised. Sensitivity testing (to erythromycin andtetracyclines in particular) should permit effective treatment of these acute exacerbations.

Strep, milleri appears to be less important in axillary hidradenitis suppurativa (Leach et al., 1979)and it seems likely that perineal lesions are more readily colonzied from the gut or the vagina. Treat-ment aimed at eliminating the organism from the flora appears logical, but we have insufficientevidence at present to confirm its value.

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