Iso older dominant female

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Iso-Kikuchi syndrome, or congenital onychodysplasia of the index finger, is an uncommon condition characterized by total anonychia or dysplasia of the nail of the index finger. It is occasionally accompanied by underlying Iso older dominant female abnormalities and is rarely associated with other conditions.

Although various hypotheses have been put forward to explain the pathophysiology of the syndrome, its etiology remains unknown. We report the cases of 3 pediatric patients 2 boys and 1 girl with nail changes and bone abnormalities consistent with Iso-Kikuchi syndrome. We highlight the importance of recognizing this entity early to avoid the need for additional tests and unnecessary treatment.

Iso-Kikuchi syndrome is characterized by anonychia or onychodysplasia of the index finger, accompanied or not by underlying bone abnormalities. It is a benign condition that is not associated with alterations to other organs or systems. Very few cases have been reported to date. Two boys and 1 girl presented with Iso-Kikuchi syndrome for the first time between the ages of 2 and 4 years. Patient 1, a 2-year-old boy, had bilateral index finger involvement and a Y-shaped deformity of the underlying phalanx Fig. His past Iso older dominant female history was remarkable for extreme preterm birth 30 weekswhich is why we have included other diseases associated with this condition in Table 1.

Patient 1. Note the micronychia on the radial side of both index fingers. Patient 2, a 4-year-old girl, had involvement of the left index finger and a Y-shaped bifurcation of the affected phalanx Fig. Patient 2. Unilateral involvement of the left index finger with micronychia on the ulnar and radial sides of the nail bed.

The rest of the nails are unaltered. Patient 3 was 2 years old and had bilateral index finger involvement Fig. Patient 3. Polyonychia of the left index finger and nail dystrophy of the right index finger. All the parents denied consanguinity and reported that their children's nail alterations had been present since birth. Iso-Kikuchi syndrome was first described by Dr. Iso in and Dr. Kikuchi in1—3 and very few cases have been reported since Table 2.

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It affects both sexes equally and can Iso older dominant female sporadic or familial. Iso-Kikuchi syndrome runs an indolent course that is characterized by anonychia or onychodysplasia of the index finger possibly accompanied by bone abnormalities in the underlying phalanx. Transmission of Iso-Kikuchi syndrome is autosomal dominant. No associations with systemic diseases have been reported. Onychodysplasia has multiple presentations Table 2the most common of which are anonychia, micronychia characteristic deviation of the nail to the ulnar side of the nail bed6 polyonychia 2 nails, 1 on each side of the nail bedhemionychogryphosis, irregular lunula, and nail malalignment.

Narrowing of the distal phalanx is a characteristic radiographic finding, but it is not always present. This narrowing will be seen on anteroposterior radiographs, whereas lateral projections will show the corresponding Y-shaped bifurcation. The etiologic and pathogenic mechanisms of Iso-Kikuchi syndrome are not fully understood, and while several theories have been put forward, none of them have been fully accepted.

Notable causes are mutations in the Wnt aling pathway during embryogenesis, in utero damage to arterioles that depend on the radial artery which has a smaller diameter than the ulnar arteryosteodystrophy of the phalanx during embryogenesis, and consumption of teratogenic drugs anticonvulsants during pregnancy. Diagnosis is mostly clinical. Iso-Kikuchi syndrome should be suspected in patients with congenital nail changes affecting one or both index fingers in the absence of other hand alterations, particularly when characteristic radiographic alterations are observed.

The differential diagnosis should include other congenital nail changes, such as ectodermal dysplasia, pachyonychia congenita, nail-patella syndrome, and deafness-onychodystrophy syndrome. Although nail changes are present from an early age in all these diseases, they have distinctive clinical characteristics and are often accompanied by extracutaneous alterations. The differential diagnosis may also include acquired nail changes due to trauma or infection, but these can be distinguished by their postnatal onset. Iso-Kikuchi syndrome is a rare, benign syndrome with characteristic clinical features.

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We have presented 3 new pediatric cases and highlight the importance of early recognition to avoid the need for unnecessary tests and treatment. The authors declare that they have no conflicts of interest. We thank Bruno Mentasti for his help with editing the photographs. Actas Dermosifiliogr. ISSN: Open Access Option.

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article Next article. Issue 5. s ee36 June More article options. Download PDF. Corresponding author. Related content. This item has received. Article information. Show more Show less. Although various hypotheses have been put forward to explain the pathophysiology of the syndrome, its etiology remains unknown. We highlight the importance of recognizing this entity early to avoid the need for additional tests and unnecessary treatment.

Palabras clave:. Full Text. Introduction Iso-Kikuchi syndrome is characterized by anonychia or onychodysplasia of the index finger, accompanied or not by underlying bone abnormalities. Case Descriptions Two boys and 1 girl presented with Iso-Kikuchi syndrome for the first time between the ages of 2 and 4 years. Figure 1. Table 1. Cases Presented. Patient 1 2 3 Sex Male Female Male Age 2 y 4 y 2 y Perinatal history Born at 30 wk; nail changes present at birth Term delivery; nail changes present at birth Term delivery; nail changes present at birth Digit involved Both index fingers Left index finger Both index fingers Appearance of other nail Micronychia on radial side Micronychia on radial and ulnar sides One finger with polyonychia, dystrophic homolateral finger Radiographic changes Bilateral Y-shaped deformity Y-shaped deformity in affected finger None Other alterations Grade Iso older dominant female intraventricular hemorrhage, bilateral inguinal bleeding, bilateral pelviureteric junction stenosis None Gastroesophageal reflux Family history No No No.

Figure 2. Figure 3. Table 2. Cases Published in the Literature. Publication Reference No. We thank Bruno Mentasti for his help with editing the photographs. Congenital nail defects of the index finger and reconstructive surgery. Seikei Geka, 20pp. Kikuchi, S. Horikawa, F. Congenital onychodysplasia of the index fingers. Arch Dermatol,pp. Di Chiacchio, J. Jasso-Olivares, N. Jacinto, M. An Bras Dermatol, 90pp. Valerio, F. Favot, I. Mattei, M. Congenital isolated Iso-Kikuchi syndrome in a newborn.

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Clin Case Rep, 3pp. Baran, J. Congenital onychodysplasia of the index fingers-Iso and Kikuchi syndrome. Kikuchi, Y. Ishi, M. Idemori, K. Congenital nail defects of the index finger. A possible explanation of radially pronounced involvement of the nail in this disorder.

J Dermatol, 8pp. Hussein, H. Brandt, T.

Iso older dominant female

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