This report describes the histologic and immunohistochemical features of a peculiar type of digital fibroma that shares some clinical and microscopic features with the more common inclusion-body type infantile digital fibromatosis. However, this type does not exhibit inclusion bodies and its cells are reactive for vimentin but not for actin. Significantly, it presents in combination with a constellation of other clinical findings, i.e., mainly positional and bone abnormalities of the fingers and toes, and skin pigmentary defects. Thus, noninclusion-body digital fibromatosis may represent the first clue for the diagnosis of the so-called terminal osseous dysplasia and pigmentary defects syndrome.
Reye RDK. Recurrent digital fibrous tumors of childhood. Arch Pathol Lab Med80:228-2311965
2.
Kempson RL, Fletcher CDM, Evans HL, Hendrickson MR, Sibley RK. Tumors of the soft tissues, Atlas of tumor pathology, AFIP, Washington, pp. 61-63, 2001
3.
O’Connell J. Inclusion body fibromatosis. In Fletcher CDM, Unni K, Merten F (eds.): Pathology and genetics of tumours of soft tissue and bone, IARC Press, Lyon, pp. 64-65, 2002
4.
Purdy LJ, Colby TV. Infantile digital fibromatosis occurring outside the digit. Am J Surg Pathol10:787-7901984
5.
Canioni D, Richard S, Rambaoud C, Manach Y, Nezelof C. Lingual localization of an inclusion body fibromatosis (Reye’s tumor). Pathol Res Pract107:886-8891991
6.
Viale G, Doglioni C, Iussolino P, Bontempini L, Colombi R, Coggi G, Dell’Orto P. Infantile digital fibromatosis-like tumour (inclusion body fibromatosis) of adulthood: Report of two cases with ultrastructural and immunocytochemical findings. Histopathology12:415-4241988
7.
Hiroaka N, Mukai M, Hosoda Y, Hata J. Phyllodes tumor of the breast containing the intracytoplasmatic inclusion bodies identical to infantile digital fibromatosis. Am J Surg Pathol10:506-5111994
8.
Pettinato G, Manivel JC, Gould EW, Albores-Saavedra J. Inclusion body fibromatosis of the breast. Two cases with immunohistochemical and ultrastructural findings. Am J Clin Pathol101:714-7181994
9.
Horii E, Sugiura Y, Nakamura R. A syndrome of digital fibromas, facial pigmentary dysplasia, and metacarpal and metatarsal disorganization. Am J Med Genet80:1-51998
10.
Breuning MH, Oranje AP, Langemeijer RAThM, Hovius SER, Diepstraten AFM, den Hollander JC, Baumgartner N, Dwek JR, Toriello H. Recurrent digital fibroma, focal dermal hypoplasia, and limb malformations. Am J Med Genet94:91-1012000
11.
Bacino CA, Stockton DW, Sierra RA, Heilstedt HA, Lewandowski R, Van den Veyver IB. Terminal osseous dysplasia and pigmentary defects: Clinical characterization of a novel male lethal X-linked syndrome. Am J Med Genet94:102-1122000
