Preputial balanitis xerotica obliterans shortly after hand, foot and mouth disease in a 5-year-old boy: Coincidence or consequence?

摘要

Balanitis xerotica obliterans (BXO), a genital form of lichen scle- rosus (LS) in males, is a progressive, sclerosing, inflammatory dermatosis affecting the foreskin, frenulum, glans penis, meatus, and urethra. BXO was considered a disease of adulthood until 1962 when Catterall and Oates reported the first case of isolated BXO of the prepuce in a 7-year-old child. 1 The exact cause and eti- ology of BXO remain unknown. However, autoimmune dysregula- tion, various infections, genetic predisposition, chronic irritation through urine exposure, and hormonal influences have been asso- ciated with the development of BXO. 2 Recent studies indicate that BXO is more common than previously thought to cause acquired phimosis and meatal stenosis. 3 However, despite an increasing rate of diagnosis of BXO in children, little is known regarding the potential for associated comorbidities or an association with various human viruses. To the best of our knowledge, no cases of hand, foot, and mouth disease (HFMD) associated with BXO have been reported so far. A 5-year-old boy was referred to our depart- ment with an 8-month history of a painless progressive inability to retract the foreskin of the glans penis. The condition worsened to almost complete occlusion over the last 15 days. His father re- ported that the inability to retract the foreskin over the glans penis occurred ten days after the appearance of the HFMD. HFMD diag- nosis was based on clinical features in the form of initial fever and trouble swallowing, followed by oral erosions and vesicles affecting the palms and soles. The illness resolved spontaneously after a week. The family history was negative for any metabolic and immune-related disorders. On physical examination, whitish, non-retractable foreskin with acquired scarring phimosis was observed (Fig. 1). After preoperative preparation, the boy under- went circumcision. The meatus itself was normal with no stenosis. The circumcised foreskin was submitted for histopathological ex- amination. Histopathological examination of the biopsy sample was consistent with BXO (Fig. 2A and B). HPV testing was not done.