Letter to Editor Preputial balanitis xerotica obliterans shortly after hand, foot and mouth disease in a 5-year-old boy: Coincidence or consequence? Keywords: Balanitis xerotica obliterans Hand Foot and mouth disease Children Dear Editor, 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. The etiology of BXO remains unknown. Various microorganisms (acid-fast bacilli, spirochetes, and viral agents such as human papillomavirus/HPV/and hepatitis C/HCV/) have been linked with BXO. 2,4 The presence of HPV by a polymerase chain reaction (PCR) in some pediatric patients with confirmed BXO 4 does not prove the association between HPV and BXO. However, it may indi- cate that HPV infection may be superimposed on BXO. A possible association of HCV with BXO has also been investigated but without clear evidence. 4 HFMD is an infectious disease caused by the Picor- naviridae family members, such as Enterovirus 71 (EV71) and Cox- sackievirus A16 (CVA16). These infections typically affect children with underlying immune or metabolic disorders. 5 Since the current literature is sparse, we can only speculate whether immune dysfunction or other mechanisms may underlie these two entities. Nevertheless, the treatment of preputial BXO with circumcision is well established as the preferred therapeutic modality, as confirmed in our case. In conclusion, the association between BXO and HFMD has not been previously reported. Further studies should reveal whether it is a real association or a coincidence. Fig. 1. Preputial appearance characterized by whitish cicatricial phimosis and scarred foreskin. Contents lists available at ScienceDirect Asian Journal of Surgery journal homepage: www.e-asianjournalsurgery.com Asian Journal of Surgery 44 (2021) 773e774 https://doi.org/10.1016/j.asjsur.2021.02.014 1015-9584/© 2021 Asian Surgical Association and Taiwan Robotic Surgery Association. Publishing services by Elsevier B.V. This is an open access article under the CC BY-NC- ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).