Abstract
Lichen sclerosus (LS) is a severe, chronic, dermatosis characterised by inflammatory, sclerotic, pruritic lesions that causes significant morbidity in patients of all genders and ages. In boys, the lesions typically affect the foreskin and glans (termed balanitis xerotica obliterans (BXO)), leading to phimosis and potentially meatal stenosis. The incidence of the disease is not well reported but the average age of affected boys is 8 years (range 1–16). Diagnosis can often be made clinically, although histological study remains important to rule out important differential diagnoses. Complications include genital scarring, urinary and sexual dysfunction as well as the development of carcinomas in adult life. Circumcision has been regarded as definitive management of BXO in boys, but this may be supplemented with medical therapies such as topical steroids, immune modulators, intralesional triamcinolone and ozonated olive oil. Supportive measures including emollients, avoidance of irritants, surveillance of complications and recurrence as well as education and counselling remain important.
Conclusion: BXO remains an important cause of phimosis in boys. The frequency of this condition appears unclear but seems likely to be less than 1% of males. Treatment generally involves circumcision, with some evidence that topical steroids or immunomodulators may decrease the incidence of recurrent meatal stenosis.
What is Known: • Surgical circumcision is considered the definitive management of BXO • Many aspects of BXO are still in contention or require further study including the epidemiology and aetiology. | |
What is New: • There is increasing awareness of non-surgical modalities that may be used in adjunct to surgery including topical corticosteroids, immune modulators, intralesional triamcinolone and ozonated olive oil • Awareness of meatal stenosis-related BXO has led to the development of surgical techniques such as preputioplasty as well as buccal mucosal inlay grafts |
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Abbreviations
- BXO:
-
Balanitis xerotica obliterans
- CI:
-
Confidence interval
- LS:
-
Lichen sclerosus
- OR:
-
Odds ratio
- OZOILE® cream:
-
Ozonated olive oil with vitamin E acetate
- RANTES:
-
Regulated on activation, normal T cell expressed and secreted
References
Ashraf J, Turner A, Subramaniam R (2018) Single-stage urethroplasty with buccal mucosal inlay graft for stricture caused by balanitis xerotica obliterans in boys: Outcomes in the medium term. J Pediatr Urol 14:66.e61–66.e65
Barbagli G, Palminteri E, Mirri F, Guazzoni G, Turini D, Lazzeri M (2006) Penile Carcinoma in Patients With Genital Lichen Sclerosus: A Multicenter Survey. J Urol 175:1359–1363
Becker K (2011) Lichen sclerosus in boys. Dtsch Arztebl Int 108:53–58
Becker K, Meissner V, Farwick W, Bauer R, Gaiser MR (2013) Lichen sclerosus and atopy in boys: coincidence or correlation? Br J Dermatol 168:362–366
Bercaw-Pratt JL, Boardman LA, Simms-Cendan JS (2014) Clinical recommendation: pediatric lichen sclerosus. J Pediatr Adolesc Gynecol 27:111–116
Beuke M, Riechardt S, Fisch M (2007) Complications after Surgery of the External Genitalia in Children with Lichen Sclerosus (BXO). J Pediatr Urol 3:S36
Bochove-Overgaauw DM, Gelders W, De Vylder AM (2009) Routine biopsies in pediatric circumcision: (non) sense? J Pediatr Urol 5:178–180
Celis S, Reed F, Murphy F, Adams S, Gillick J, Abdelhafeez AH, Lopez P-J (2014) Balanitis xerotica obliterans in children and adolescents: a literature review and clinical series. J Pediatr Urol 10:34–39
Charlton OA, Smith SD (2018) Balanitis xerotica obliterans: a review of diagnosis and management. Int J Dermatol 58:777–781
Chernosky ME, Derbes VJ, Burks JW (1957) Lichen sclerosus et atrophicus in children. Arch Dermatol 75:647–652
Chi C-C, Kirtschig G, Baldo M, Lewis F, Wang S-H, Wojnarowska F (2012) Systematic review and meta-analysis of randomized controlled trials on topical interventions for genital lichen sclerosus. J Am Acad Dermatol 67:305–312
Christman MS, Chen J, Holmes N (2009) Obstructive complications of lichen sclerosus. J Pediatr Urol 5:165–169
Clouston D, Hall A, Lawrentschuk N (2011) Penile lichen sclerosus (balanitis xerotica obliterans). BJU Int 108:14–19
Curro M, Russo T, Ferlazzo N, Caccamo D, Antonuccio P, Arena S, Parisi S, Perrone P, Ientile R, Romeo C, Impellizzeri P (2018) Anti-Inflammatory and Tissue Regenerative Effects of Topical Treatment with Ozonated Olive Oil/Vitamin E Acetate in Balanitis Xerotica Obliterans. Molecules 23:645
Depasquale I, Park AJ, Bracka A (2000) The treatment of balanitis xerotica obliterans. BJU Int 86:459–465
Drake T, Rustom J, Davies M (2013) Phimosis in childhood. BMJ 346:f3678
Ebert AK, Rosch WH, Vogt T (2008) Safety and tolerability of adjuvant topical tacrolimus treatment in boys with lichen sclerosus: a prospective phase 2 study. Eur Urol 54:932–937
Fischer G (1995) Lichen sclerosus in childhood. Australas J Dermatol 36:166–167
Fistarol S, Itin P (2013) Diagnosis and Treatment of Lichen Sclerosus. Am J Clin Dermatol 14:27–47
Folaranmi SE, Corbett HJ, Losty PD (2017) Does application of topical steorids for lichen sclerosus (balanitis xerotica obliterans) affect the rate of circumcision? A systematic review. J Pediatr Surg 53:2225–2227
Fuchs ME, Beecroft N, Dajusta DG, McLeod DJ (2017) The Association Between BXO and Obesity in Boys Undergoing Circumcision. Global Pediatr Health 4:2333794x17742749
Gargollo PC, Kozakewich HP, Bauer SB, Borer JG, Peters CA, Retik AB, Diamond DA (2005) Balanitis xerotica obliterans in boys. J Urol 174:1409–1412
Ghysel C, Vander Eeckt K, Bogaert GA (2009) Long-Term Efficiency of Skin Stretching and a Topical Corticoid Cream Application for Unretractable Foreskin and Phimosis in Prepubertal Boys. Urol Int 82:81–88
Green PA, Bethell GS, Wilkinson DJ, Kenny SE, Corbett HJ (2019) Surgical management of genitourinary lichen sclerosus et atrophicus in boys in England: A 10-year review of practices and outcomes. J Pediatr Urol 15:45.e41–45.e45
Hartley A, Ramanathan C, Siddiqui H (2011) The surgical treatment of Balanitis Xerotica Obliterans. Indian J Plast Surg 44:91–97
Hengge UR, Ruzicka T, Schwartz RA, Cork MJ (2006) Adverse effects of topical glucocorticosteroids. J Am Acad Dermatol 54:1–15
Holbrook C, Tsang T (2011) Management of boys with abnormal appearance of meatus at circumcision for balanitis xerotica obliterans. Ann R Coll Surg Engl 93:482–484
Homer L, Buchanan KJ, Nasr B, Losty PD, Corbett HJ (2014) Meatal stenosis in boys following circumcision for lichen sclerosus (balanitis xerotica obliterans). J Urol 192:1784–1788
Howard M, Hall A (2018) Treatment of penile lichen sclerosus with topical corticosteroids for over 25 years' duration: A case report. SAGE Open Med Case Rep 6:2050313X18795047-12050313X18795047
Jayakumar S, Antao B, Bevington O, Furness P, Ninan GK (2012) Balanitis xerotica obliterans in children and its incidence under the age of 5 years. J Pediatr Urol 8:272–275
Kirtschig G (2016) Lichen sclerosus - Presentation, diagnosis and management. Dtsch Arztebl Int 113:337–343
Kiss A, Csontai A, Pirót L, Nyirády P, Merksz M, Király L (2001) The response of balanitis xerotica obliterans to local steroid application compared with placebo in children. J Urol 165:219–220
Kiss A, Király L, Kutasy B, Merksz M (2005) High Incidence of Balanitis Xerotica Obliterans in Boys with Phimosis: Prospective 10-Year Study. Pediatr Dermatol 22:305–308
Kizer SW, Prarie FT, Morey FA (2003) Balanitis Xerotica Obliterans: Epidemiologic Distribution in an Equal Access Health Care System. South Med J 96:9–11
Kuehhas FE, Miernik A, Weibl P, Schoenthaler M, Sevcenco S, Schauer I, Tosev G, Oezsoy M, Lassmann J (2013) Incidence of Balanitis Xerotica Obliterans in Boys Younger than 10 Years Presenting with Phimosis. Urol Int 90:439–442
Legendre L, Barnetche T, Mazereeuw-Hautier J, Meyer N, Murrell D, Paul C (2015) Risk of lymphoma in patients with atopic dermatitis and the role of topical treatment: A systematic review and meta-analysis. J Am Acad Dermatol 72:992–1002
Margolis DJ, Abuabara K, Hoffstad OJ, Wan J, Raimondo D, Bilker WB (2015) Association Between Malignancy and Topical Use of Pimecrolimus. JAMA Dermatol 151:594–599
Mattioli G, Repetto P, Carlini C, Granata C, Gambini C, Jasonni V (2002) Lichen sclerosus et atrophicus in children with phimosis and hypospadias. Pediatr Surg Int 18:273–275
McLelland J (2004) Lichen sclerosus in children. J Obstet Gynaecol 24:733–735
Meuli M, Briner J, Hanimann B, Sacher P (1994) Lichen sclerosus et atrophicus causing phimosis in boys: a prospective study with 5-year followup after complete circumcision. J Urol 152:987–989
Mohammed A, Shegil IS, Christou D, Khan A, Barua JM (2012) Paediatric balanitis xerotica obliterans: an 8-year experience. Arch Ital Urol Androl 84:12–16
Moreno G, Corbalán J, Peñaloza B, Pantoja T (2014) Topical corticosteroids for treating phimosis in boys. Cochrane Database Syst Rev 2:CD008973
Nasca MR, Innocenzi D, Micali G (1999) Penile cancer among patients with genital lichen sclerosus. J Am Acad Derm 41(6):911–914
Pilatz A, Altinkilic B, Schormann E, Maegel L, Izykowski N, Becker J, Weidner W, Kreipe H, Jonigk D (2013) Congenital phimosis in patients with and without lichen sclerosus: distinct expression patterns of tissue remodeling associated genes. J Urol 189:268–274
Pradhan A, Patel R, Said AJ, Upadhyaya M (2019) 10 Years’ Experience in Balanitis Xerotica Obliterans: A Single-Institution Study. Eur J Pediatr Surg 29:302–306
Russo T, Curro M, Barbera A, Caccamo D, Antonuccio P, Arena S, Montalto AS, Parisi S, Marseglia L, Gitto E, Ientile R, Impellizzeri P, Romeo C (2016) Expression of Transglutaminase in Foreskin of Children with Balanitis Xerotica Obliterans. Int J Mol Sci 17:1551
Sandler G, Patrick E, Cass D (2008) Long standing balanitis xerotica obliterans resulting in renal impairment in a child. Pediatr Surg Int 24:961–964
Shankar R (1999) The incidence of phimosis in boys. BJU Int 84:101–102
Singh J, Priyadarshi V, Goel H, Vijay M, Pal D, Chakraborty S, Kundu A (2015) Penile lichen sclerosus: An urologist's nightmare! - A single center experience. Urol Ann 7:303–308
Singh L, Sengar M, Goyal S, Mansi M, Khurana N, Mohta A (2018) Childhood Phimosis Secondary to Lichen Sclerosus: Is There a Spatial Pattern of Histopathological Changes? Am J Dermatopathol 40:824–828
Sneppen I, Thorup J (2016) Foreskin Morbidity in Uncircumcised Males. Pediatrics 137:e20154340
Somov P, Chan B, Wilde C, Corbett H (2017) Bleeding after circumcision is more likely in children with lichen sclerosus (balanitis xerotica obliterans). J Pediatr Urol 13:208.e1–208.e4
The Royal Children’s Hospital Melbourne (2018) The penis and foreskin. https://www.rch.org.au/clinicalguide/guideline_index/The_penis_and_foreskin/. Accessed 28/5/2019
Tong LX, Sun GS, Teng JMC (2015) Pediatric Lichen Sclerosus: A Review of the Epidemiology and Treatment Options. Pediatr Dermatol 32:593–599
Villa M, Dragonetti E, Grande M, Bove P, Sansalone S, Rulli F, Tambucci R, Tucci G, Baldi A (2012) Skin phototype and local trauma in the onset of balanitis xerotica obliterans (BXO) in circumcised patients. In Vivo 26:143–146
Vincent MV, MacKinnon E (2005) The response of clinical balanitis xerotica obliterans to the application of topical steroid-based creams. J Pediatr Surg 40:709–712
Virgili A, Borghi A, Cazzaniga S, Di Landro A, Naldi L, Minghetti S, Verrone A, Stroppiana E, Caproni M, Nasca MR, D’Antuono A, Papini M, Di Lernia V, Corazza M (2017) New insights into potential risk factors and associations in genital lichen sclerosus: Data from a multicentre Italian study on 729 consecutive cases. J Eur Acad Dermatol Venereol 31:699–704
Wilkinson DJ, Lansdale N, Everitt LH, Marven SS, Walker J, Shawis RN, Roberts JP, Mackinnon AE, Godbole PP (2012) Foreskin preputioplasty and intralesional triamcinolone: a valid alternative to circumcision for balanitis xerotica obliterans. J Pediatr Surg 47:756–759
Acknowledgements
The authors would like to thank Dr Jack Garland from the Department of Anatomical Pathology at Liverpool Hospital, NSW, Australia, for his assistance with the histological slide.
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Alexander T M Nguyen - Literature search, Drafting of manuscript, Final approval of manuscript.
Andrew J A Holland - Concept for article, Literature search, Critical revision of article, Final approval of manuscript.
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Nguyen, A.T.M., Holland, A.J.A. Balanitis xerotica obliterans: an update for clinicians. Eur J Pediatr 179, 9–16 (2020). https://doi.org/10.1007/s00431-019-03516-3
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DOI: https://doi.org/10.1007/s00431-019-03516-3