Saxophone penis after unilateral inguinal bubo of lymphogranuloma venereum

Sir,

Secondary lymphedema is predominantly caused in India by filariasis and sexually transmitted infections (STIs) like lymphogranuloma venereum (LGV).[1] Other important cause is surgical removal of lymph nodes.[2] ‘saxophone penis’ is a typical deformity of penis arising secondary to diseases causing chronic lymphatic obstruction in bilateral inguinal region. Here we report a rare case of ‘saxophone penis’ resulting from surgical intervention in right inguinal region for inguinal hernia and left inguinal bubo due to LGV.

A 45-year-old male patient presented with a nontender, enlarged (6” in length and 5” in diameter), solid, twisted penis along with a tender swelling in left inguinal region [Figure 1]. He had a history of operation for right inguinal hernia; evident by a scar mark and swelling in right inguinal region. He had an unprotected sexual contact with a commercial sex worker 7 months back. Consequently, he felt pain in left inguinal region along with fever, chill, myalgia, and malaise. There was gradually progressive swelling and tenderness of right inguinal lymph nodes which initially were firm, tender, and movable. Later, they became matted and fixed to underlying structures and overlying skin. After more 2 months, he noted gradual thickening of skin of penis and scrotum along with twisting of the penis. The surface of scrotum became verrucous due to lymphangiectatic papules. He denied any history of previous genital ulcer or urethritis. In last 4 months, the penis got considerably swollen, woody, indurated, and deformed giving rise to the so called ‘ramrod’, ‘ram's horn’ or ‘saxophone penis’.

Figure 1.

Saxophone penis with lymphangiectatic papules on scrotum, left inguinal bubo, and scar mark of surgery in right inguinal region

All routine tests including Mantoux test, chest X-ray, ultrasound of abdomen, serum Venereal Disease Research Laboratory (VDRL) test were within normal limits. The patient was seronegative for HIV-1 and 2. Repeated midnight peripheral blood smears for microfilaria, after provocation by 100 mg tablet of diethylcarbamazine were negative. Serological tests for LGV like complement fixation tests and microimmunofluorescence tests were not done due to nonavailability. On the basis of history and clinical findings, a diagnosis of LGV induced left inguinal bubo was made. Surgical intervention for right inguinal hernia and left inguinal bubo due to LGV resulted in bilateral inguinal lymphatic obstruction; resulting in secondary lymphedema of penis and scrotum with a ‘saxophone’ deformity. The patient was prescribed tablet doxycycline 100 mg twice daily for 1 month resulting in marked decrease in left inguinal bubo, regression of sizes of lymphangiectatic papules, and mild decrease in bend of shaft of penis [Figure 2].

Figure 2.

Marked decrease in left inguinal bubo, regression of sizes of lymphangiectatic papules on scrotum

The causative agent of LGV is Chlamydia trachomatis serovars L1-L3. It is a lymphotropic organism which initiates the disease process primarily in the lymphatic channels, leading to thrombolymphangitis and perilymphangitis. LGV causes bilateral inguinal bubo in one-third of cases[3] and ‘saxophone penis’ after LGV is well known. In most endemic areas like India, the diagnosis of LGV is often differential; after other causes of inguinal lymphadenopathy have been ruled out.[3]

In India there are few reports of penoscrotal elephantiasis[4] and ‘saxophone penis’ due to LGV.[5] Paucity of reports of ‘saxophone penis’, especially in the Indian literature, prompted us to report this case here. LGV generally causes ‘saxophone penis’ after formation of bilateral inguinal ‘bubo’. Our case was unique as it was sequel to left inguinal bubo due to LGV and surgical intervention in right inguinal region.