Congenital onychodysplasia of the index fingers. Iso and Kikuchi syndrome

Baran, R.

doi:10.1001/archderm.120.2.243

Cited by 14 publications

(26 citation statements)

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“…Congenital onychodystrophy of the index fingers (COIF) identifies a clinical syndrome characterized by nail dysplasias of the index fingers associated with bone abnormalities. In our cases the terms congenital onychodysplasia or Iso and Kikuchi syndrome, rather than COIF, were preferred because the location could vary . Isolated congenital onychodystrophy of the toes with underlying bony abnormality has rarely been reported.…”

mentioning

confidence: 98%

Image Gallery: Congenital onychodysplasia of the toenails

Yang

Kim

et al. 2017

Br J Dermatol

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confidence: 98%

Image Gallery: Congenital onychodysplasia of the toenails

Yang

Kim

et al. 2017

Br J Dermatol

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“…5 Nail dysplasias of various forms and severity occur in combination with disturbances of hair growth, dentition, and/or sweating within the large group of ectodermal dysplasias, 6 in association with deafness, 7 and as a result of skeletal abnormalities, such as in nail-patella syndrome 8 and Iso and Kikuchi syndrome. 9 The onychopathy in this family clearly differs from these conditions by the type of nail deformity and the absence of extracutaneous findings.…”

Section: Commentmentioning

confidence: 75%

Isolated Congenital Nail Dysplasia

Hamm

Karl²,

Bröcker³

2000

Arch Dermatol

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Developmental nail abnormalities are extremely heterogeneous, with hereditary isolated conditions being a small and rare subgroup. An unusual congenital nail dysplasia observed in a large South German kindred was characterized clinically to review the question of uniqueness.Design: Case series of affected family members.Setting: University department of dermatology and houses of patients. Patients:The history and clinical features in 22 affected family members (13 females and 9 males, aged 5 to 74 years) were recorded and documented by photographs. Nail biopsy samples were taken from 2 patients. Interventions: None.Results: The pedigree spanning 5 consecutive generations was best compatible with autosomal dominant in-heritance with complete penetrance. Nail alterations were mostly present since birth and soon reached an individually variable degree of severity. Affected persons showed longitudinal streaks and thinning of nail plates, mostly of all fingernails and toenails, with some accentuation of the thumbnail and big toenails, poorly developed lunulae, longitudinal angular ridges of individual nail plates occasionally starting proximally from a reddish prominence, platonychia and koilonychia of individual nails often overgrowing the lateral folds, and notches and fissures of the free margins. Histological abnormalities included a prominent granular layer of the nail matrix and epithelial strands and buds extending from the nail bed. There were no associated anomalies. Other nail dystrophies were excluded by differences in clinical and histological features. Conclusion:The nail abnormality observed in our family represents a new autosomal dominant disorder for which we propose the term isolated congenital nail dysplasia.

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“…Splitting of the index ®nger nail with unequal growth of the two components seems to be the main characteristic of COIF [Iso, 1969a;Murakoa et al, 1977], but the full spectrum of the nail dysplasia include irregular lunula, malalignment, micronychia (hypoplastic and rudimental), polyonychia (split rudimental), and anonychia [Baran, 1980;Kikuchi et al, 1981;Baran and Stroud, 1984] (Table I). In polyonychia, there are two small incomplete nails on one index ®nger, with the radial one being smaller than its ulnar counterpart.…”

Section: Discussionmentioning

confidence: 99%

“…Iso [1969aIso [ , 1969b ®rst described a defect as``congenital nail defect of the index ®ngers '' and Kikuchi et al [1974] proposed its present name (COIF). Although COIF has been mainly reported in Japan, some cases have been also referred in USA [Millman and Strier, 1982;Baran and Stroud, 1984], in England [Harper and Weber, 1985], in France [Baran, 1980;Claudy, 1980], and in Korea [Youn et al, 1996].…”

Section: Introductionmentioning

confidence: 99%

Peculiar facial appearance and generalized brachydactyly in a patient with congenital onychodysplasia of the index fingers (Iso-Kikuchi syndrome)

et al. 2001

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We report on a patient with clinical manifestations consistent with a diagnosis of congenital onychodysplasia of the index fingers (COIF). This syndrome has been found mainly in Japan, and as far as we know, this is the first case reported in Italy. In addition to the typical bilateral split nail of the second finger, the patient showed bilateral inguinal hernia, a peculiar face, and short hands. The metacarpophalangeal profile showed a generalized brachydactyly with all the hand long bones below x3 SD. The patient's father showed a peculiar kind of micronychia on both the fifth toes, suggesting a possible autosomal dominant transmission of the syndrome. In utero ischemia of the palmar digital artery and a dysplastic change in the crescent-shaped cap of the distal phalanx are the two main candidate pathogenetic mechanisms that have been proposed. In our opinion, the gradual broadening of the spectrum of this syndrome brings support to the hypothesis of a basal dysplastic pathogenetic mechanism involving not only the index fingers but also perhaps other tissues outside. We think that for the moment the definition of COIF for this syndrome should be maintained, the alternative proposed term "congenital onychodysplasia" being too indefinite.

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Congenital onychodysplasia of the index fingers. Iso and Kikuchi syndrome

Cited by 14 publications

References 1 publication

Image Gallery: Congenital onychodysplasia of the toenails

Image Gallery: Congenital onychodysplasia of the toenails

Isolated Congenital Nail Dysplasia

Peculiar facial appearance and generalized brachydactyly in a patient with congenital onychodysplasia of the index fingers (Iso-Kikuchi syndrome)

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