Massive exophytic abscesses and fibrotic masses of thechin: A variant of the follicular occlusion triad

Scott W. Meyers, MD,a Lionel Bercovitch, MD,c Kimberly Polley, PA-C,a James Taira, MD,e Nicole DeCamp, MD,f

Meera Mahalingam, MD, PhD,a Greg Grillone, MD,b and Donald Grande, MDd

Stoughton, Boston, and Medford, Massachusetts, and Oklahoma City, Oklahoma

We present a patient with an extensive cluster of exophytic nodules that developed on his chin. These nodules consisted of abscessesand fibrotic areas. Lesion morphology, histology, and microbiology support a follicular occlusion triad entity. However, thedistribution is striking and does not fit the entities described in the triad. We present the case to show that follicular occlusion wasthe inciting factor in our patient’s eruption and to broaden our concept of clinical manifestations that can arise from this pathologicprocess. (J Am Acad Dermatol 2003;48:S47-50.)

W e report a unique case of massive, exophytic ab-scesses and fibrotic masses occurring on the chinand tracheostomy site of a 46-year-old white man

who is ventilator dependent.Although the morphologic features of the lesions on the chin

do not fit precisely into one of the entities that constitute thefollicular occlusion triad, we believe that they share similarpathogenic and histologic features and should be regarded as avariant.

CASE REPORTThe patient is a 46-year-old white man who is ventilator

dependent and has been in a vegetative state since he sustainedhead injuries in a motor vehicle accident in 1979. Shortly afterthe accident, his mother noticed persistent “white heads” locatedprimarily on his chin and nose. Inflammatory pustules and pap-ules then developed on the chin in 1993, when a change wasmade from using a blade to an electric razor to shave the patient.The electric razor was used aggressively and often caused bleed-ing and redness over the chin. No treatment was sought for thiseruption.

He was first examined by a dermatologist (L. B.) in 1994when filiform warts developed on his left cheek. No inflamma-tory lesions were noted on the chin during that visit. A year laterhe was noted to have draining cysts and nodules of the groin,pubic area, scrotum, and base of the penis. Cultures were neg-ative and a diagnosis of hidradenitis suppurativa was made.These lesions responded to tetracycline by a percutaneous en-doscopic gastrostomy (PEG) tube.

In the summer of 1997, inflammatory papules and pustulesrecurred on the chin. A trial of tetracycline by a PEG tube was

given for presumed acne conglobata. Short-term improvementoccurred. However, repeated courses of tetracycline could notreproduce the initial response. Subsequently, doxycycline andminocycline by a PEG tube were tried on separate occasions butneither produced any significant response. Intralesional triam-cinolone also was not effective in reducing the size of thelesions. The lesions became progressively larger and were inter-mittently drained of pus.

The deep-seated pustules and inflammatory nodules progres-sively increased in size on the chin and behind the ears for thenext year. The lesions eventually became a massive cluster ofexophytic, abscessed, and fibrotic masses on the chin (Fig 1).Cystic, fluctuant masses were also present at the retroauricularsulci, tracheotomy site, and inguinal and crural folds. Foul-smelling drainage was noted over the pedunculated masses onthe chin.

The patient was seen by us in consultation in October 1998for surgical evaluation because the lesions were becoming suf-ficiently large to impinge on the tracheostomy site. The clinicalphotographs demonstrate the extent of the process on the chinat the time of operation in December 1998.

The patient has taken phenytoin and phenobarbital for 18years to treat seizures. Other medications include lorazepam,potassium chloride, docusate sodium, and psyllium mucilloid.By history, no medication has been temporally associated withthe exacerbation of this cutaneous process. His medical historyis otherwise unremarkable.

The patient was treated with continuous-wave carbon diox-ide laser ablation to remove the masses from the chin, retroau-ricular areas, and tracheotomy site (Fig 2). Photographs taken 8weeks postoperatively demonstrate the beneficial result (Fig 3).Current treatment includes the use of a Silastic sheet molded tothe chin for daily use. If an exacerbation occurs, we plan toadminister isotretinoin by the PEG tube.

Work-up included Gram staining of purulent material, cul-tures (AFB, fungal, bacterial), routine histology, and specialstains (Brown-Brenn, GMS, and acid-fast). Histology showedfollicular hyperkeratosis (occlusion) and an infundibulofollicu-litis superficially (Fig 4). Deeper sections were characterized byan interstitial lymphocytic inflammatory process with many neu-trophils concentrated around follicles, suggesting a deep follic-ular inflammatory process with follicular rupture (Fig 5). Fibrosisand naked hair shafts were also noted in several sections. All

This supplement is made possible through an unrestrictededucational grant from Stiefel Laboratories to the AmericanAcademy of Dermatology.

From the Departments of Dermatologya and Otorhinolaryngology,b BostonUniversity; private practices in Stoughtonc and Medford,d Mass, andOklahoma Citye; and Oklahoma City Clinic.f

Reprint requests: Donald Grande, MD, 92 High St T-21, Medford, MA 02155-3840. E-mail: djgrande319@aol.com.

Copyright © 2003 by the American Academy of Dermatology, Inc.0190-9622/2003/$30.00 � 0doi:10.1067/mjd.2003.145

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special stains were negative for organisms. Cultures were repeat-edly negative; however, a gram-positive, anaerobic bacterialisolate, Peptostreptococcus micros, was cultured from one of thesurgical specimens. This organism was thought to be a second-ary bacterial invader and not pathogenic in this case. On thebasis of the pathology, we suggest that follicular occlusion wasthe primary pathogenic factor in the development of these le-sions.

DISCUSSIONDiseases in the follicular occlusion triad (hidradenitis suppu-

rativa, acne conglobata, and dissecting cellulitis of the scalp) arethought to have similar pathogenic, histologic, and clinical fea-tures. All show follicular hyperkeratosis and subsequent poralocclusion as initiating events.1-3 Plugging of the pilosebaceousunit is followed by distension and eventual rupture of the folli-cle. A deep follicular inflammatory process ensues with abscessand sinus tract formation. Chronically, fibrosis becomes a signif-icant feature in all of these entities. Any bacterial involvement issecondary.4,5

Clinically, each follicular occlusion triad process is character-ized by erythematous fluctuant nodules, draining sinuses, andchronic deep-seated scars. Distribution is the primary clinicalfactor that differentiates each disorder: hidradenitis suppurativa(axillae and groin), acne conglobata (back, chest, and buttocks),and dissecting cellulitis of the scalp. All 3 of these entities havebeen documented in a single patient.6 The simultaneous occur-rence of the triad in 1 patient provides evidence that each ofthese 3 processes is similar and shares a common pathogenicmechanism.

Early in the course of disease, our patient had a facial erup-tion that was typical of acne vulgaris according to his mother’sdescription. Later, recurrent bouts of spontaneously drainingfluctuant nodules developed in the groin and deep-seated pus-tules on the chin. He carried concurrent diagnoses of acneconglobata and hidradenitis suppurativa. The association ofacne conglobata and hidradenitis suppurativa is well estab-lished. However, several reports also support an associationbetween acne vulgaris and hidradenitis suppurativa.7-10 Im-

Fig 1. Large cluster of exophytic abscesses.

Fig 2. Immediate postoperative result of carbon dioxide ablation.

Fig 3. Results at 8-week follow-up.

Fig 4. Superficial infundibular folliculitis. (Original magnification�10.)

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provement with tetracycline occurred on the face and in thegroin, which is consistent with each of these diagnoses. There-fore, the early course can be explained by acne vulgaris alone.The later course is more consistent with acne conglobata andhidradenitis suppurativa.

The onset of deeper inflammatory nodules on the chin oc-curred without any change in medication or medical treatment.We are not sure what role the aggressive use of an electric razormay have played in inducing these deeper lesions. Temporally,it is the only change in his care that was associated with the onsetof the nodules and abscesses on the chin. He was diagnosedwith acne conglobata, although this process rarely affects theface alone.

By December 1998, our patient had a massive cluster ofpedunculated abscesses and fibrosed nodules on his chin. Ourdifferential diagnosis included acne conglobata, acne keloidaliswith an unusual distribution, pseudofolliculitis barbae with sec-ondary abscess formation, and infectious pyoderma. The mor-phology and distribution was unusual for any of these entities. Inaddition to the chin nodules, he had smaller but similar erythem-atous nodules around his tracheostomy site and behind his ears.Given the typical course of hidradenitis suppurativa in the groin,we considered the facial eruption to be a variant of one of thefollicular occlusion triad entities. Further support of this obser-vation is that one previous report has demonstrated hidradenitissuppurativa around an ostomy site, which was also present inthis case.11

Histology supports a follicular occlusion–type process, andspecial stains and cultures were negative for an infectious cause.Several histologic sections showed follicular occlusion, which

we consider to be the inciting event. An infundibulofolliculitiswas also noted and is a frequent finding in hidradenitis suppu-rativa.2 Other findings consisted of follicular distention, rupture,and extensive fibrosis. These findings are consistent with follic-ular occlusion triad disorders. Recent reports demonstrate thatthe pilosebaceous unit is the primary site occluded in hidrade-nitis suppurativa and that any apocrine ductal occlusion is sec-ondary.1,2,12,13 Therefore, it is conceivable that any site contain-ing follicles could be affected. In our patient, the chin,postauricular areas, and peristomal areas happened to be thesites affected. Microbiologic cultures excluded an infectious pro-cess. However, a culture was positive for a gram-positive anaer-obic bacterium, P micros. We considered this organism to be asecondary pathogen on the basis of the routine histologic find-ings and the low pathogenic potential of this organism. Previousstudies have shown that the presence of bacteria in hidradenitissuppurativa occurs secondarily and that Peptostreptococcus is themost common genus of anaerobe that is isolated in cases ofhidradinitis suppurativa.5

Although the histology, microbiology, and early morphologicfeatures suggest a follicular occlusion–type process, the distri-bution and morphology of the lesions on the chin are quiteunusual. Hidradenitis suppurativa and dissecting cellulitis of thescalp have not been reported on the chin. In addition, acneconglobata rarely affects the face. Therefore, the face is typicallyspared in these disorders. However, a case has been reported ofhidradenitis suppurativa of the eyelid.14 In this case, the glandsof Moll were presumed to be the site occluded. Our patient’seruption was primarily on the chin and occurrence of theselesions at this site has not previously been reported.

The markedly exophytic and pedunculated masses on thechin were a late morphologic feature. These late features can beexplained by the protracted course without successful medicaltreatment or surgical intervention during the past 12 months. Theend result is an exaggerated, dramatically exophytic process thatdoes not fit the clinical description of any acneiform process orpyoderma previously described. The early morphology, patho-genesis, and histology are all consistent with a follicular occlu-sion triad disorder. Therefore, we present this case as an exten-sive example of hidradenitis suppurativa with an unusualdistribution on the chin. Both the distribution and extent of theeruption were unique, making our patient an intriguing case.

We thank Dr John S. Strauss for his help and recommenda-tions regarding this manuscript.

REFERENCES1. Attanous RL, Appleton MA, Douglas-Jones AG. The pathogenesis of

hidradenitis suppurativa: a closer look at apocrine and apoeccrineglands. Br J Dermatol 1995;133:254-8.

2. Jemec GB, Hansen U. Histology of hidradenitis suppurativa. J Am AcadDermatol 1996;34:994-9.

3. Lucas S. Bacterial disease. In: Lever’s histopathology of the skin. 8th ed.New York: Lippincott-Raven; 1997. p. 461-3.

4. Djawari D, Homstein OP. Recurrent chronic pyoderma with cellular im-munodeficiency. Dermatologica 1980;116:116.

5. Brook I, Frazier EH. Aerobic and anaerobic microbiology of axillaryhidradenitis suppurativa. J Med Microbiol 1999;48:103-5.

6. Chicarilli ZN. Follicular occlusion triad: hidradenitis suppurativa, acneconglobata, and dissecting cellulitis of the scalp. Ann Plast Surg 1987;18:230-7.

7. Chow ET, Mortimer PS. Successful treatment of hidradenitis suppura-tiva and retroauricular acne with etretinate. Br J Dermatol 1992;126:415.

8. Shenefelt PD. Pyoderma gangrenosum associated with cystic acne and

Fig 5. Deep follicular inflammation and follicular rupture. (Originalmagnification �10.)

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hidradenitis suppurativa controlled by adding minocycline and sul-fasalazine to the treatment regimen. Cutis 1996;57:315-9.

9. Chalmers RJ, Ead RD, Beck MH, Dewis P, Anderson DC. Acne vulgaris andhidradenitis suppurativa as presenting features of acromegaly. Br Med J1983;287:1346-7.

10. Hurley HJ. Disease of the apocrine sweat glands. In: Moschella andHurley’s dermatology. 3rd ed. Philadelphia: Saunders; 1992. p. 1506-11.

11. Fleisher I, Bryant D, Spiegel J, Christian R, Farraye FA. Peristomal hidra-denitis suppurativa. J Wound Ostomy Continence Nurs 1996;23:171-3.

12. Boer J, Weltevreden EF. Hidradenitis suppurativa or acne inversa: a clin-icopathological study of early lesions. Br J Dermatol 1996;135:721-5.

13. Yu CC, Cook MG. Hidradenitis suppurativa: a disease of follicular epithe-lium, rather than apocrine glands. Br J Dermatol 1990;122:763-9.

14. Sachs DD, Gordon AT. Hidradenitis suppurativa of glands of Moll. ArchOphthalmol 1967;77:635-6.

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