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European Journal of Pediatrics

, Volume 164, Issue 11, pp 673–677 | Cite as

Pilomatricoma in childhood: a retrospective study from three European paediatric centres

  • Bruno Cigliano
  • Nikolaos Baltogiannis
  • Marianna De Marco
  • Elsa Faviou
  • Alesandro Settimi
  • Stefanos Tilemis
  • Michail Soutis
  • Evangellos Papandreou
  • Sergio D’Agostino
  • Maria Angelica Fabbro
Original Paper

Abstract

Pilomatricoma is characterised as a common, slowly growing benign cutaneous tumour that appears generally within the first decades of life. The clinical diagnosis is frequently missed, especially by the paediatrician unfamiliar with these tumours. We present the experience gained in three European tertiary care paediatric centres with the treatment of pilomatricoma and also current data on the aetiology, clinical presentation and management. A retrospective study was carried out in 83 patients suspected for pilomatricoma during a 7-year period (1996–2002) at the departments of Paediatric Surgery of the Children’s University Hospital “Federico II”, Naples, Hospital “San Bortolo”, Vicenza and “Aghia Sophia” Children’s Hospital, Athens. The age range was from 10 months to 17 years, median age 8 years. All patients were treated by surgical excision and all specimens were examined by histopathological assessment. The follow-up varied from 5 months to 6 years. The correct diagnosis was made preoperatively in 68 patients (82%). The female/male ratio was 2:1. The sites of occurrence were the head (47.5%), especially in the periorbital region, the neck (9%), the upper limbs (35.5%), the inferior limbs (4%) and the thorax (4%). Each patient exhibited a single pilomatricoma except for two patients who had multiple lesions (2.4%). One of them had Steinert disease (myotonic dystrophy). No recurrences were observed during the follow-up period. Conclusion:Pilomatricoma is one of the most common cutaneous adnexal neoplasms in children. Surgical excision including clear margins and its overlying skin in most cases is the treatment of choice. The recurrence as well as malignant evolution is rare.

Keywords

Calcifying epithelioma of Malherbe Childhood Cutaneous tumour Pilomatricoma 

References

  1. 1.
    Agarwal RP, Handler SD, Matthews MR, Carpentieri D (2001) Pilomatrixoma of the head and neck in children. Otolaryngol Head Neck Surg 125: 510–515CrossRefPubMedGoogle Scholar
  2. 2.
    Arnold HL (1977) Pilomatricoma (letter). Arch Dermatol 13: 1303Google Scholar
  3. 3.
    Barberio E, Nino M, Dente V, Delfino M (2002) Multiple pilomatricomas and Steinert’s disease. Eur J Dermatol 12: 293–294CrossRefPubMedGoogle Scholar
  4. 4.
    Chan EF, Gat U, McNiff JM, Fuchs E (1999) A common human skin tumor is caused by activating mutations in beta-catenin. Nat Genet 21: 410–413CrossRefPubMedGoogle Scholar
  5. 5.
    Cooper PH, Fechner RE (1983) Pilomatricoma-like changes in the epidermal cysts of Gardner’s syndrome. JAAD 8: 639–644Google Scholar
  6. 6.
    Danielson-Cohen A, Lin SJ, Hughes CA, An YH, Maddalozzo J (2001) Head and neck pilomatrixoma in children. Arch Otolaryngol Head Neck Surg 127: 1481–1483PubMedGoogle Scholar
  7. 7.
    Delfino M, Monfrecola G, Ayala F, Suppa F, Piccirillo A (1985) Multiple familial pilomatricomas: a cutaneous marker for myotonic dystrophy. Dermatologica 170: 128–132PubMedGoogle Scholar
  8. 8.
    Demircan M, Balik E (1997) Pilomatricoma in children: a prospective study. Pediatr Dermatol 14: 430–432PubMedGoogle Scholar
  9. 9.
    Duflo S, Nicollas R, Roman S, Magalon G, Triglia JM (1998) Pilomatrixoma of the head and neck in children: a study of 38 cases and review of the literature. Arch Otolaryngol Head Neck Surg 124: 1239–1242PubMedGoogle Scholar
  10. 10.
    Forbis R, Helwing EB (1961) Pilomatrixoma (calcifying epithelioma). Arch Dermatol 83: 606–608PubMedGoogle Scholar
  11. 11.
    Geh JLC, Moss ALH (1999) Multiple pilomatrixomata and myotonic dystrophy: a familial association. Br J Plast Surg 52: 143–145CrossRefPubMedGoogle Scholar
  12. 12.
    Hardsson D, Linares D, Cuevas-Santos J, Contreras F (2001) Pilomatrix carcinoma. A clinicopathologic study of six cases and review of the literature. Am J Dermatopathol 23: 394–401CrossRefPubMedGoogle Scholar
  13. 13.
    Hassnein AM, Glanz SM (2004) Beta-catenin expression in benign and malignant pilomatrix neoplasms. Br J Dermatol 150: 511–516CrossRefPubMedGoogle Scholar
  14. 14.
    Hughes J, Lam A, Rogers M (1999) Use of ultrasonography in the diagnosis of childhood pilomatrixoma. Pediatr Dermatol 16: 341–344CrossRefPubMedGoogle Scholar
  15. 15.
    Jacobsen AS, Bowen J, Bruce J (1995) The calcifying epithelioma of Malherbe in children: a 15 year experience. Pediatr Surg Int 10: 44–45CrossRefGoogle Scholar
  16. 16.
    Julian CG, Bowers PW (1998) A clinical review of 209 pilomatricomas. J Am Acad Dermatol 39: 191–195PubMedGoogle Scholar
  17. 17.
    Kajino Y, Yamaguchi A, Hashimoto N, Matsuura A, Sato N, Kikuchi K (2001) Beta-catenin gene mutation in human hair follicle-related tumors. Pathol Int 51: 543–548CrossRefPubMedGoogle Scholar
  18. 18.
    Kethley JL, Gamble JW, Grafton WD (1978) Pilomatrixoma (calcifying epithelioma of Malherbe). J Oral Surg 36: 618–622PubMedGoogle Scholar
  19. 19.
    Knight PJ, Reiner CB (1983) Superficial lumps in children: what, when and why? Pediatrics 72: 147–153PubMedGoogle Scholar
  20. 20.
    Lan M-Y, Lan M-C, Ho C-Y, Li W-Y, Lin C-Z (2003) Pilomatricoma of the head and neck. A retrospective review of 179 cases. Arch Otolaryngol Head Neck Surg 129: 1327–1330CrossRefPubMedGoogle Scholar
  21. 21.
    Lemos MM, Kindblom LG, Meis-Kindblom JM, Ryd W, Willén H (2001) Fine-needle aspiration features of pilomatrixoma. Cancer Cytopathol 93: 252–256Google Scholar
  22. 22.
    Malherbe A, Chenantais J (1880) Note sur l’epitheliome calcifie des glandes sebaces. Prog Med 826–837Google Scholar
  23. 23.
    McCulloch TA, Singh S, Cotton DWK (1996) Pilomatrix carcinoma and multiple pilomatrixomas. Br J Dermatol 134: 368–371CrossRefPubMedGoogle Scholar
  24. 24.
    Mikhaeel NG, Spittle MF (2001) Malignant pilomatrixoma with multiple local recurrences and distant metastases: a case report and review of the literature. Clin Oncol 13: 386–389Google Scholar
  25. 25.
    Migirov E, Fridman E, Talmi YP (2002) Pilomatrixoma of the retroauricular area and arm. J Pediatr Surg 37: E20CrossRefPubMedGoogle Scholar
  26. 26.
    Moehlenbeck WF (1973) Pilomatrixoma (calcifying epithelioma): a statistical study. Arch Dermatol 108: 532–534CrossRefPubMedGoogle Scholar
  27. 27.
    Moreno-Bueno G, Gamallo C, Perez-Gallego L, Contreras F, Palacios J (2001) Beta-catenin expression in pilomatrixomas. Relationship with beta-catenin gene mutations and comparison with beta-catenin expression in normal hair follicles. Br J Dermatol 145: 576–581CrossRefPubMedGoogle Scholar
  28. 28.
    Noguchi H, Kayashima K, Nishiyama S, Ono T (1999) Two cases of pilomatrixoma in Turner’s syndrome. Dermatology 199: 338CrossRefPubMedGoogle Scholar
  29. 29.
    Pirouzmanesh A, Reinish JF, Gonzalez-Gomez I, Smith EM, Meara JG (2003) Pilomatrixoma: a review of 346 cases. Plast Reconstr Surg 112: 1784–1789CrossRefPubMedGoogle Scholar
  30. 30.
    Rotenberg M, Laccourreye L, Putterman M, Brasnu D (1996) Head and neck pilomatrixoma. Am J Otolaryngol 17: 133–135CrossRefPubMedGoogle Scholar
  31. 31.
    Sanchez CS, Bascunana AG, Quirante FAP, Romero MSM, Fernandez JC, Perez JS (1996) Mimics of pilomatrixoma in fine-needle aspirates. Diagn Cytopathol 14: 75–83CrossRefPubMedGoogle Scholar
  32. 32.
    Sassmannshausen J, Chaffins M (2001) Pilomatrix carcinoma: a report of a case arising from a previously excised pilomatrixoma and a review of the literature. J Am Acad Dermatol 44: 358–361CrossRefPubMedGoogle Scholar
  33. 33.
    Sevin K, Can Z, Yilmaz S, Saray A, Yormuk E (1995) Pilomatrixoma of the earlobe. Dermatol Surg 21: 245–246CrossRefPubMedGoogle Scholar
  34. 34.
    Shoji A (1988) Pilomatricoma. Skin Res 30: 305–311Google Scholar
  35. 35.
    Street ML, Rogers RS (1991) Multiple pilomatricomas and myotonic dystrophy. J Dermatol Surg Oncol 17: 728–730PubMedGoogle Scholar
  36. 36.
    Solanki P, Ramzy I, Durr N (1987) Pilomatrixoma. Cytological features with differential diagnostic considerations. Arch Pathol Lab Med 11: 294–297Google Scholar
  37. 37.
    Thomas RW, Perkins JA, Ruegemer JL, Munaretto JA (1999) Surgical excision of pilomatrixoma of head and neck: a retrospective review of 26 cases. Ear Nose Throat J 78: 541–548PubMedGoogle Scholar
  38. 38.
    Viero RM, Tani E, Skoog L (1999) Fine needle aspiration (FNA) cytology of pilomatrixoma: report of 14 cases and review of the literature. Cytopathology 10: 263–269CrossRefPubMedGoogle Scholar
  39. 39.
    Yencha MW (2001) Head and neck pilomatricoma in the pediatric age group: a retrospective study and literature review. Int J Pediatr Otorhinolaryngol 57: 123–128CrossRefPubMedGoogle Scholar

Copyright information

© Springer-Verlag 2005

Authors and Affiliations

  • Bruno Cigliano
    • 1
  • Nikolaos Baltogiannis
    • 2
    • 4
  • Marianna De Marco
    • 1
  • Elsa Faviou
    • 2
  • Alesandro Settimi
    • 1
  • Stefanos Tilemis
    • 1
  • Michail Soutis
    • 2
  • Evangellos Papandreou
    • 2
  • Sergio D’Agostino
    • 3
  • Maria Angelica Fabbro
    • 3
  1. 1.Department of PaediatricsUniversity Hospital “Federico II”Naples Italy
  2. 2.2nd Surgical Department“Aghia Sophia” Children’s HospitalAthens Greece
  3. 3.Department of Paediatric Surgery“San Bortolo” HospitalVicenza Italy
  4. 4.P. Faliro, Athens Greece

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