Literature DB >> 15754355

Fibular aplasia, tibial campomelia, and oligosyndactyly in a male newborn infant: a case report and review of the literature.

Winnie Courtens1, Ann Jespers, Inge Harrewijn, Dirk Puylaert, Filip Vanhoenacker.   

Abstract

We report on a male newborn with a rarely described congenital limb deficiency syndrome consisting of shortening and anterior bowing of the right lower limb at the distal third of the tibia with associated overlying soft tissue dimpling, oligodactyly of the right foot, and a left-sided oligosyndactyly of the hand. The right hand and left lower limb were clinically normal. Radiographic examination revealed complete absence of the right fibula, absence of the right-sided Vth ray, and anterior bowing and shortening of the right-sided tibia. Femora, humeri, ulnae, and radii were normal. The infant had neither facial dysmorphia nor other associated anomalies. A limb deficiency syndrome comparable to this case has been reported in a female by Hecht and Scott, the only report classified under OMIM 246570 so far. We found two other reports describing three cases comparable to our case and the female reported by Hecht and Scott, and reviewed these cases. The major common findings in all the five cases consist of fibular aplasia, tibial campomelia, and oligosyndactyly. Therefore, we propose to name it fibular aplasia-tibial campomelia-oligosyndactyly (FATCO) syndrome. Additional case reports are needed for further delineation of this rare limb deficiency syndrome.

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Year:  2005        PMID: 15754355     DOI: 10.1002/ajmg.a.30441

Source DB:  PubMed          Journal:  Am J Med Genet A        ISSN: 1552-4825            Impact factor:   2.802


  8 in total

1.  A Turkish Female Twin Sister Patient with Fibular Aplasia, Congenital Tibia Pseudoarthrosis, Oligosyndactyly, and Negative WNT7A Gene Mutation.

Authors:  Hale Önder Yılmaz; Duran Topak; Orkun Yılmaz; Seda Çakmaklı
Journal:  J Pediatr Genet       Date:  2018-11-18

2.  A rare association of congenital absence of femur and fibular hemimelia with maternal hyperpyrexia.

Authors:  Devendra Kumar; Sriram Krishnamurthy
Journal:  Indian J Pediatr       Date:  2009-11       Impact factor: 1.967

3.  Overlap between Fibular Aplasia, Tibial Campomelia, and Oligosyndactyly and Fuhrmann's Syndromes in an Egyptian Female Infant.

Authors:  Ebtesam M Abdalla; Ahmed A El-Beheiry
Journal:  J Pediatr Genet       Date:  2017-01-02

4.  FATCO Syndrome Variant - Fibular Hypoplasia, Tibial Campomelia and Oligosyndactyly -- A Case Report.

Authors:  Navendu Goyal; Randeep Kaur; Manu Gupta; Shiraz Bhatty; Rajesh Paul
Journal:  J Clin Diagn Res       Date:  2014-09-20

5.  Hemimelia and absence of the peroneal artery.

Authors:  S Huda; G Sangster; A Pramanik; S Sankararaman; H Tice; H Ibrahim
Journal:  J Perinatol       Date:  2014-02       Impact factor: 2.521

6.  A Japanese male patient with 'fibular aplasia, tibial campomelia and oligodactyly': an additional case report.

Authors:  Taichi Kitaoka; Noriyuki Namba; Ji Yoo Kim; Takuo Kubota; Kohji Miura; Yoko Miyoshi; Haruhiko Hirai; Mikihiko Kogo; Keiichi Ozono
Journal:  Clin Pediatr Endocrinol       Date:  2009-08-01

7.  A Female Newborn Infant with FATCO Syndrome Variant (Fibular Hypoplasia, Tibial Campomelia, Oligosyndactyly) - A Case Report.

Authors:  Gitte Smets; Yoeri Vankan; Annick Demeyere
Journal:  J Belg Soc Radiol       Date:  2016-02-26       Impact factor: 1.894

8.  Fibular Hypoplasia, Talar Aplasia, Absent Proximal Tibial Growth Plate and Oligosyndactyly (Variant of Fibular Aplasia, Tibial Campomelia, and Oligosyndactyly Syndrome) - Paucity of Case Reports with Evolving Definition.

Authors:  Pankaj Kumar Mishra; Maneesh Verma
Journal:  J Orthop Case Rep       Date:  2021-08
  8 in total

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