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SOFT syndrome with kohlschutter-Tonz syndrome.
Mondkar, S A; Khadilkar, V; Kasegaonkar, P; Khadilkar, A.
Afiliación
  • Mondkar SA; Growth and Pediatric Endocrinology Unit, Hirabai Cowasji Jehangir Medical Research Institute, Jehangir Hospital, Pune, Maharashtra, India.
  • Khadilkar V; Growth and Pediatric Endocrinology Unit, Hirabai Cowasji Jehangir Medical Research Institute, Jehangir Hospital, Pune, Maharashtra, India.
  • Kasegaonkar P; Department of Neurology, CNS Hospital, Solapur, Maharashtra, India.
  • Khadilkar A; Growth and Pediatric Endocrinology Unit, Hirabai Cowasji Jehangir Medical Research Institute, Jehangir Hospital, Pune, Maharashtra, India.
J Postgrad Med ; 70(1): 56-59, 2024.
Article en En | MEDLINE | ID: mdl-37706418
We report a 2.2 year-old-boy, born of consanguineous marriage, referred for short stature, with history of neonatal death and skeletal deformities in his older sibling. Rhizo-mesomelic dwarfism was detected antenatally. Within 24 hours of birth, he developed multiple seizures. Examination revealed severe short stature, dolichocephaly, broad forehead, deep set eyes, low set ears, bulbous nose, small, irregular teeth, pointed chin, and triangular facies. He had rhizomelic shortening, stubby fingers, pes planus, and scanty hair. Neurological evaluation revealed ataxia, hypotonia, and global developmental delay. Skeletal survey radiograph revealed shallow acetabuli, short femurs and humerus, short, broad metacarpals and short cone-shaped phalanges with cupping of phalangeal bases. Clinical exome analysis revealed homozygous mutations involving the POC1A gene and the SLC13A5 gene responsible for SOFT syndrome and Kohlschutter-Tonz syndrome respectively, which were inherited from the parents. Both these syndromes are extremely rare, and their co-occurrence is being reported for the first time.
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Texto completo: 1 Bases de datos: MEDLINE Asunto principal: Osteocondrodisplasias / Anomalías Múltiples / Simportadores / Demencia / Enanismo / Epilepsia / Amelogénesis Imperfecta Idioma: En Revista: J Postgrad Med Año: 2024 Tipo del documento: Article País de afiliación: India

Texto completo: 1 Bases de datos: MEDLINE Asunto principal: Osteocondrodisplasias / Anomalías Múltiples / Simportadores / Demencia / Enanismo / Epilepsia / Amelogénesis Imperfecta Idioma: En Revista: J Postgrad Med Año: 2024 Tipo del documento: Article País de afiliación: India