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Fox-Fordyce Disease with Hidradenitis Suppurativa* Rachel Ford Spiller, M.D

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Fox-Fordyce Disease with Hidradenitis Suppurativa* Rachel Ford Spiller, M.D View metadata, citation and similar papers at core.ac.uk brought to you by CORE provided by Elsevier - Publisher Connector FOX-FORDYCE DISEASE WITH HIDRADENITIS SUPPURATIVA* RACHEL FORD SPILLER, M.D. AND JOHN M. KNOX, M.D. Poral closure is a feature of both Fox-Fordyceacneform or furuncular inflammation of the disease and hidradenitis suppurativa. Shelley (1,apocrine glands described in 1864 by Verneuil. 2) has demonstrated histologically the poral clos-Sutton (22) believes that two diseases manifest ure phenomenon in both diseases. Sulzbergerf hasabscesses of the apocrine sweat glands; apocrine stated that there is a relationship between Fox-acne and apocrine furunculosis. He says the apo- Fordyce disease and hidradenitis suppurativa,crine acne is not primarily parasitic, but bears with both having obstruction of the follicle. Itmarked resemblance to acne conglobata, some might be logical to suspect that the two diseasescases of acne keloidalis, and perifolliculitis abs- would be found together with considerable fre-cedens et suffodiens. The apocrine furunculosis quency; however, the English literature has beenmay be a primary or secondary bacterial adenitis. surveyed and hidradenitis suppurativa as a com-Ormsby and Montgomery (23) state that hidrad- plication of Fox-Fordyce was mentioned in onlyenitis is fundamentally an infection of the one report (3). We are reporting three instancesapocrine glands, but genetic and hormonal in which Fox-Fordyce disease and hidradenitisfactors may be implicated in its etiology. Hidrad- suppurativa occurred together. enitis was reviewed in 1933 by Lane (24), and Fox-Fordyce disease is a rare, itchy disorder ofin 1939, Brunsting (25) stressed the relation to unknown etiology, affecting primarily women (4)acne conglobata. Most subsequent writings have and characterized by a persistent eruption ofbeen about therapy, relation to acne, and patho- small, discrete, dry papules located symmetricallygenesis. in the axillary, areolar, pubic, and sternal regions. The two original eases of this rare, papular dis- SUMMARIES OF CASES ease involving the axillary region were described Case1. (FiguresNo. 1 and No. 2) E. M., a 33 by Fox and Fordyce (5) in 1902, with one caseyear old colored patient, came to the clinic for occurring in a female and one in a male. Fordyce'streatment of a few, deep, painful, nodular lesions (6) early impression was that this process was aof hidradenitis suppurativa. This developed in November of 1954, shortly after beginning the use variety of neurodermatitis, probably toxic in of a new deodorant. An incidental finding was the origin, and the clinical manifestations were sec- presence of numerous, small, flat-topped, papular ondary to scratching. After Schiefferdecker'slesions in the axillae, about the nipples, and in the (1, 7) monumental treatise on sweat glands invulvar region. She stated that these lesions had 1922, many accepted that Fox-Fordyce diseasebeen present for 10 to 12 years and that they pro- is a functional derangement of the large apocrineduced considerable itching. Hair was scanty in sweat glands based on some endocrine disorder.the axillae. Most writings on this disease have been case The hidradenitissuppurativaresponded reports (8—16), hypothesis as to etiology (17),promptly to therapy. It is of interest that the interesting associated conditions (18, 19) andpruritis associated with the Fox-Fordyce disease was relieved by oral Terramycin; however, various histochemical and histopathologic studies (20,forms of treatment had no objective effect on the 21). Attention at the present time seems to bepapular lesions. These included 150,000 units of focused on pathogenesis, with speculation as toVitamin A per day, antihistamines, Gantrisin, and whether the primary pathology is an obstructiontopical hydrocortisone. of the apocrine ducts or a change in the apocrine Laboratory work-up, including examination of glandular activity with secondary factors con-the blood and urine, was normal. The chest x-ray tributing to the total pathological picture. revealed no pathology. Hidradenitis suppurativa is a deeply seated Pathological examination (Figures No. 3 and No. 4) showed the stratum corneum to be irregu- *Fromthe Department of Dermatology, Baylorlar, with alternate areas of normal thickness and University College of Medicine, Houston, Texas;hyperkeratosis. In an occasional area there was Everett R. Seale, Chairman. Received for publication February 26, 1958. poral closure with keratinous material plugging f Sulzberger, Marion B. in a discussion (3). the duct. Undet l'Ing the keratinous plug there 127 128 THEJOURNAL OF INVESTIGATIVE DERMATOLOGY FOX-FORDYCE WITH HIDRADENITIS SUPPURATIVA 129 • •--r ! It rY. •41 S.-:'I'. CD L4•1 130 THEJOURNAL OF INVESTIGATIVE DERMATOLOGY FOX-FORDYCE WITH HIDRADENITIS SUPPURATIVA 131 (. FIG.7 was dilatation of the duct and surrounding intra-of those found in hidradenitis suppurativa. The cellular and intercellular edema. The rete ridgescentral area of necrosis and inflammation was were both elongated and broadened. The super-quite large and extensive. ficial dermis was normal. From the mid-dermis to Cese 2.(FigureNo. 5) I. F. J., a 19 year old the base of the specimen submitted for examina-colored female, manifested the typical papular tion, there were numerous foci of inflammatorylesions of Fox-Fordyce disease with the eruption cells. These were primarily located about eccrinelocated in the axillary, areolar, periumbilical, and sweat glands and apocrine sweat glands. In gen-perineal regions. The disease began at the age of eral, there was more inflammation about the16 and since onset, had been constant and un- apocrine sweat glands. In some sections there waschanged. The lesions itched only when she was a large abscess in the deep dermis. Necrotic debristense, nervous, or excited, and the pruritus would filled the center of the abscess. Connective tissue and appendages were destroyed throughout aeventually subside spontaneously. In addition, large area. Polymorphonuclear leukocytes pre-the patient had periodically developed tender, dominated in the inflammatory infiltrate. Aroundswollen, subcutaneous nodules in the axillae, the periphery of the abscess, lymphocytes andbreasts, and on the labia. The latter lesions fibrocytes were numerous. brought the patient to the physician. The superficial histopathologic changes were On physical examination, the patient had those of Fox-Fordyce disease and manifestedtender, swollen, subcutaneous nodules in the clearly the classic sweat retention vesicle in theaxillae and on the labia underlying areas of Fox- proximity of the poral outlet. The large abscessFordyce disease. in the dermis was non-specific, but characteristic On routine laboratory examination, the urine 132 THE JOURNAL OF INVESTIGATIVE DERMATOLOGY Fio. 8 was normal. The blood was normal except for anosis of hidradenitis suppurativa. This abscess leukocytosis of 11,800. was confined to the level of the sweat gland coil. Pathological examination (Figures No. 6 and CGSC 3.(FigureNo. 8) This 22 year old colored No. 7) showed that, except for a slight hyperkera-woman was diagnosed as having Fox-Fordyce tosis, the stratum corneum was normal. Thedisease in February 1956. For the previous two granular cell layer manifested no significantmonths she had noted pruritus of the axillac, histologic changes. The malpighian layer wassternal areas, and perineal area. This pruritus was acanthotic with elongation and broadening of themost pronounced in hot weather. Since October rete ridges. In one large, pilosebaceous structure1955, the patient had also complained of recur- there was a massive keratin plug that producedrent episodes of tender nodules in the axillae follicular and poral closure. There was intracellu-with suppuration and drainage. She also gave a lar and intercellular edema about the apocrinehistory of having tender nodules on the external sweat ducts. Developing sweat retention vesiclesgenitalia on one occasion. were noted. Inflammatory cells, primarily lympho- The past medical history revealed two items of cytes, were also present. In the mid-dermis therepossible significance. In September of 1955, the were foci of inflammatory cells, located primarilypatient was diagnosed as having carbuncles in about small blood vessels and appendages. Inthe auditory canals and an associated external the deeper dermis there was a large abscess filledotitis media. This was accompanied by pruritus with necrotic debris and polymorphonuclearand excoriation of the external auditory canal. leukocytes. This abscess was sharply localizedIn December 1955, the patient was seen in the at the level of the apocrine and cccrine sweatGynecology Clinic for the sudden onset of dys- glands. In the vicinity of the abscess, appendagespareunia. There was pain during, but no pain and connective tissue were destroyed and poly-after intercourse. On pelvic examination there morphonuclear lcukocytes were numerous. Inwere definite, freely movable nodules palpable in adjacent areas, some of the apocrine and eccrinethe vaginal wall. This pelvic disease was not sweat glands were completely normal. Others werefollowed as the patient did not return to the surrounded by a few inflammatory cells whichGynecology Clinic. were mainly mononuclear with lymphocytes On physical examination, the patient presented predominating. A few plasma cells were alsopinhead-sized, papular lesions of Fox-Fordyce in noted. The deepest portions of the dermis werethe axillae, surrounding the areola, and
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