Literature DB >> 17849153

Rabson-Mendenhall syndrome: medullary sponge kidney, a new component.

Andrew M Harris1, Bryan Hall, Vesna M Kriss, John L Fowlkes, Stefan G Kiessling.   

Abstract

Rabson-Mendenhall syndrome is a rare genetic disorder characterized by severe insulin resistance, extreme hyperinsulinemia, postprandial hyperglycemia, growth retardation, and dysmorphisms. Enlargement of the kidneys and nephrocalcinosis have been described previously. We report a 10-year-old boy who presented with gross hematuria, unilateral hydronephrosis, and the initial diagnosis of bilateral extensive medullary nephrocalcinosis. Medullary sponge kidney (MSK) was included in the differential diagnosis given the ultrasound findings. Further evaluation by intravenous pyelogram confirmed the suspected bilateral MSK. Given the patient's history of hydronephrosis due to an obstructing renal stone and MSK, urine calcium excretion was assessed and found to be markedly increased at 9.5 mg/kg per day. To our knowledge, this is the first report of Rabson-Mendenhall syndrome and an association with MSK. We recommend evaluation for nephrocalcinosis, MSK, and hypercalciuria in all children diagnosed with Rabson-Mendenhall syndrome.

Entities:  

Mesh:

Year:  2007        PMID: 17849153     DOI: 10.1007/s00467-007-0600-5

Source DB:  PubMed          Journal:  Pediatr Nephrol        ISSN: 0931-041X            Impact factor:   3.714


  21 in total

Review 1.  Insulin resistance--mechanisms, syndromes, and implications.

Authors:  D E Moller; J S Flier
Journal:  N Engl J Med       Date:  1991-09-26       Impact factor: 91.245

2.  Tumor of the pineal body with high insulin resistance.

Authors:  E N MENDENHALL
Journal:  J Indiana State Med Assoc       Date:  1950-01

3.  Association of medullary sponge kidney and medullary dysplasia in Beckwith-Wiedemann syndrome.

Authors:  R W Chesney; R Kaufman; F B Stapleton; M L Rivas
Journal:  J Pediatr       Date:  1989-11       Impact factor: 4.406

4.  Wiedemann-Beckwith syndrome: presentation of clinical and cytogenetic data on 22 new cases and review of the literature.

Authors:  M J Pettenati; J L Haines; R R Higgins; R S Wappner; C G Palmer; D D Weaver
Journal:  Hum Genet       Date:  1986-10       Impact factor: 4.132

5.  Ehlers-Danlos syndrome with renal tubular acidosis and medullary sponge kidneys. A report of a case and studies of renal acidification in other patients with the Ehlers-Danlos syndrome.

Authors:  A S Levine; A F Michael
Journal:  J Pediatr       Date:  1967-07       Impact factor: 4.406

6.  A primary defect in insulin receptor in a young male patient with insulin resistance.

Authors:  Y Takata; M Kobayashi; H Maegawa; N Watanabe; O Ishibashi; Y Shigeta; A Fujinami
Journal:  Metabolism       Date:  1986-10       Impact factor: 8.694

7.  A syndrome of insulin resistance resembling leprechaunism in five sibs of consanguineous parents.

Authors:  L I al-Gazali; M Khalil; K Devadas
Journal:  J Med Genet       Date:  1993-06       Impact factor: 6.318

Review 8.  Medullary sponge kidneys and unilateral Wilms tumour in a child with Beckwith-Wiedemann syndrome.

Authors:  R Beetz; O Schofer; H Riedmiller; R Schumacher; P Gutjahr
Journal:  Eur J Pediatr       Date:  1991-05       Impact factor: 3.183

9.  Decreased insulin binding to cultured cells from a patient with the Rabson-Mendenhall syndrome: dichotomy between studies with cultured lymphocytes and cultured fibroblasts.

Authors:  S I Taylor; L H Underhill; J A Hedo; J Roth; M S Rios; R M Blizzard
Journal:  J Clin Endocrinol Metab       Date:  1983-04       Impact factor: 5.958

Review 10.  Mutant insulin receptors in syndromes of insulin resistance.

Authors:  A Krook; S O'Rahilly
Journal:  Baillieres Clin Endocrinol Metab       Date:  1996-01
View more
  5 in total

Review 1.  Rabson-Mendenhall syndrome: two case reports and a brief review of the literature.

Authors:  Renuka J Bathi; Sameena Parveen; Sunil Mutalik; Reema Rao
Journal:  Odontology       Date:  2010-02-16       Impact factor: 2.634

2.  Metreleptin improves blood glucose in patients with insulin receptor mutations.

Authors:  Rebecca J Brown; Elaine Cochran; Phillip Gorden
Journal:  J Clin Endocrinol Metab       Date:  2013-08-22       Impact factor: 5.958

3.  Two Novel Variants and One Previously Reported Variant in the Insulin Receptor Gene in Two Cases with Severe Insulin Resistance Syndrome.

Authors:  Aydilek Dagdeviren Cakir; Said Saidov; Hande Turan; Serdar Ceylaner; Yavuz Özer; Tufan Kutlu; Oya Ercan; Olcay Evliyaoglu
Journal:  Mol Syndromol       Date:  2020-03-18

4.  Insulin Receptor and the Kidney: Nephrocalcinosis in Patients with Recessive INSR Mutations.

Authors:  Arabella Simpkin; Elaine Cochran; Fergus Cameron; Mehul Dattani; Martin de Bock; David B Dunger; Gun Forsander; Tulay Guran; Julie Harris; Iona Isaac; Khalid Hussain; Robert Kleta; Catherine Peters; Velibor Tasic; Rachel Williams; Fabian Yap Kok Peng; Stephan O''Rahilly; Philipp Gorden; Robert K Semple; Detlef Bockenhauer
Journal:  Nephron Physiol       Date:  2014-10-24

5.  Renal manifestations of severe Rabson-Mendenhall syndrome: a case report.

Authors:  Yih Harng Chong; Barry J Taylor; Benjamin J Wheeler
Journal:  J Diabetes Metab Disord       Date:  2013-01-22
  5 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.