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Case Report

Balanoposthitis and Penile Edema

Atypical Manifestations of Primary Syphilis

Rovira-López, Roger MD; García-Martínez, Pablo MD; Martín-Ezquerra, Gemma MD, PhD; Pujol, Ramon M. MD; Aventín, Daniel López MD

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doi: 10.1097/OLQ.0000000000000322
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CASE REPORT

A previously healthy, 46-year-old heterosexual man presented to the emergency department with a 10-day history of painless penile shaft edema, along with diffuse swelling and crusted erosions over the entire glans penis and prepuce (Figs. 1 and 2). Examination also revealed an indurated ulceration on the glans, underneath the crusts. Bilateral voluminous tender inguinal lymphadenopathy was palpable. The rest of the physical examination showed no abnormalities. He referred last unprotected sexual intercourse with a woman 2 months earlier. Bacteriological and mycological cultures from the glans resulted positive for Staphylococcus aureus and Haemophilus parainfluenzae, but negative for Candida albicans. Polymerase chain reaction (PCR) for herpes simplex virus DNA was negative. A multiplex PCR assay for the simultaneous detection of Chlamydia trachomatis, Neisseria gonorrhoeae, Trichomonas vaginalis, and Mycoplasma genitalium in urethral swab specimen resulted negative. Serological testing for syphilis was performed: a rapid plasma reagin test gave a result of 1:8 and specific immunoglobulin M enzyme immunoassay against Treponema pallidum yielded values of 4.56 U/mL (reference range, 0.1–1.5 U/mL). HIV serology was negative. Histopathological examination from a nonulcerated zone of the glans penis revealed a psoriasiform epithelial hyperplasia covered with a parakeratotic and multilocular serous exudative horny layer, vascular dilatation, and a dense and diffuse infiltrate of numerous plasma cells, lymphocytes, and histiocytes in the upper subepithelial connective tissue (Fig. 3). A real-time PCR for detection of T. pallidum was performed in formalin-fixed and paraffin-embedded skin biopsy and resulted positive. Primary syphilis presenting as balanoposthitis with associated underlying chancre, penile edema, and bilateral inguinal lymph nodes enlargement was diagnosed. The presence of S. aureus was considered a secondary infection and H. parainfluenzae as a mucosal commensal. Treatment with a single intramuscular injection of 2.4 million units of benzathine-penicillin and topical 2% fusidic acid ointment led to the resolution of all clinical manifestations within 3 weeks.

FIGURE 1
FIGURE 1:
Prominent penile edema and erosive and crusted balanoposthitis.
FIGURE 2
FIGURE 2:
Detail of the glans penis and foreskin. Note the erosive erythema, whitish macules of the glans and prepuce, adherent crusts on the glans, and an indurated ulceration compatible with a chancre on the left side of the glans.
FIGURE 3
FIGURE 3:
Biopsy from the glans penis showing elongation of rete ridges, vascular dilatation, and a dense and diffuse infiltrate of numerous plasma cells, lymphocytes, and histiocytes in the upper subepithelial connective tissue (hematoxylin-eosin, original magnification ×100).

Balanitis is defined as inflammation of the glans penis, which often involves the prepuce (balanoposthitis). There are a wide range of causes, but infection is the most commonly reported etiology.1 Syphilitic balanoposthitis is a rare manifestation of syphilis. Follmann was the first to describe 3 cases of primary syphilis presenting as erosive balanitis without a chancre.2–4 Since then, around 10 cases have been published in the medical literature.3–8 Clinical features of the syphilitic balanitis or balanoposthitis are variable, including an edematous and dark erythematous balanitis and the presence of erosions, crusts, or coalescent smooth white/pink papules and plaques on the surface of the glans.3,4,6,7 Diffuse cardboard-like induration of the glans penis may be present.3,4 In syphilitic balanitis of Follmann, a chancre may be present before, concomitant with (as was the case in our patient), or after the balanitis. However, it may also be absent in some cases.3,4,7 In only one case, balanitis and penile edema occurred simultaneously with a cutaneous eruption on his buttocks, lower abdomen, scrotum, thighs, and soles representing an unusual manifestation of secondary syphilis.8 The pathogenesis of syphilitic balanitis of Follmann was discussed by Lejman and Starzycki.7 A massive intraepidermal accumulation of T. pallidum secondary to an active penetration of the spirochetes through the walls of capillaries and the epidermal basal membrane would cause a diffuse inflammation of the glans penis. Inguinal lymphadenopathy is usually present and must lead to a suspicion of primary syphilis.3,4 Diagnosis of syphilitic balanoposthitis requires the exclusion of other infectious (C. albicans, groups B and D streptococci, anaerobes, herpes simplex virus) and noninfectious (lichen sclerosus et atrophicus, lichen planus, Zoon balanitis, psoriasis, Reiter syndrome, eczema, fixed drug eruption, erythroplasia of Queyrat) causes of balanoposthitis.1,3,4 In addition, acute penile edema can be caused by infection, filariasis, vigorous sexual intercourse, irritant or allergic contact dermatitis, injury, paraphimosis, adverse drug reaction, or insect bite.8,9 The presence of S. aureus in a previously reported patient was considered by the authors as a secondary infection,3 as it was in the present case.

Syphilitic balanitis and balanoposthitis may be the unique clinical expression of primary syphilis. Serological tests for syphilis should be performed in any case of balanitis, especially when the glans penis is indurated and inguinal lymphadenopathy is present.3 Demonstration of T. pallidum by direct detection methods (dark-field examination, PCR, immunohistochemistry, or Warthin-Starry argentic staining depending on local expertise and availability of each laboratory) provides definitive diagnosis of syphilitic infection.10 Treatment of syphilitic balanitis of Follmann is identical to that of primary syphilis consisting of a single dose of 2.4 million units of benzathine-penicillin administered intramuscularly.4 Finally, although a very uncommon condition, syphilitic balanitis should be considered in the differential diagnosis of balanitis attending to the recrudescence of syphilis during the last years.

REFERENCES

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