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DOI 10.1007/s00467-021-05374-4

Медицинская библиотека · Pediatric nephrology (Berlin, Germany) · 2022

A multidisciplinary nephrogenetic referral clinic for children and adults-diagnostic achievements and insights.

Pode-Shakked Ben, Ben-Moshe Yishay, Barel Ortal, Regev Lilach C, Kagan Maayan, Eliyahu Aviva, Marek-Yagel Dina, Atias-Varon Danit, Lahav Einat, Issler Naomi, Shlomovitz Omer, Semo Oz Rotem, Kol Nitzan, Mor Nofar, Bar-Joseph Ifat, Khavkin Yulia, Javasky Elisheva, Beckerman Pazit, Greenberg Meidad, Volovelsky Oded, Borovitz Yael, Davidovits Miriam, Haskin Orly, Alfandary Hadas, Levi Shely, Kaidar Maital, Katzir Ze'ev, Angel-Korman Avital, Becker-Cohen Rachel, Ben-Shalom Efrat, Leiba Adi, Mor Eytan, Dagan Amit, Pessach Itai M, Lotan Danny, Shashar Moshe, Anikster Yair, Raas-Rothschild Annick, Rechavi Gideon, Dekel Benjamin, Vivante Asaf

СтатьяПоддержка исследования: вне правительства СШАПеревод в очереди

Аннотация

BACKGROUND

Genetic kidney diseases contribute a significant portion of kidney diseases in children and young adults. Nephrogenetics is a rapidly evolving subspecialty; however, in the clinical setting, increased use of genetic testing poses implementation challenges. Consequently, we established a national nephrogenetics clinic to apply a multidisciplinary model.

METHODS

Patients were referred from different pediatric or adult nephrology units across the country if their primary nephrologist suspected an undiagnosed genetic kidney disease. We determined the diagnostic rate and observed the effect of diagnosis on medical care. We also discuss the requirements of a nephrogenetics clinic in terms of logistics, recommended indications for referral, and building a multidisciplinary team.

RESULTS

Over 24 months, genetic evaluation was completed for a total of 74 unrelated probands, with an age range of 10 days to 72 years. The most common phenotypes included congenital anomalies of the kidneys and urinary tract, nephrotic syndrome or unexplained proteinuria, nephrocalcinosis/nephrolithiasis, tubulopathies, and unexplained kidney failure. Over 80% of patients were referred due to clinical suspicion of an undetermined underlying genetic diagnosis. A molecular diagnosis was reached in 42/74 probands, yielding a diagnostic rate of 57%. Of these, over 71% of diagnoses were made via next generation sequencing (gene panel or exome sequencing).

CONCLUSIONS

We identified a substantial fraction of genetic kidney etiologies among previously undiagnosed individuals which influenced subsequent clinical management. Our results support that nephrogenetics, a rapidly evolving field, may benefit from well-defined multidisciplinary co-management administered by a designated team of nephrologist, geneticist, and bioinformatician. A higher resolution version of the Graphical abstract is available as Supplementary information.

Для цитирования: Pode-Shakked Ben, Ben-Moshe Yishay, Barel Ortal, Regev Lilach C, Kagan Maayan, Eliyahu Aviva, Marek-Yagel Dina, Atias-Varon Danit, Lahav Einat, Issler Naomi, Shlomovitz Omer, Semo Oz Rotem, Kol Nitzan, Mor Nofar, Bar-Joseph Ifat, Khavkin Yulia, Javasky Elisheva, Beckerman Pazit, Greenberg Meidad, Volovelsky Oded, Borovitz Yael, Davidovits Miriam, Haskin Orly, Alfandary Hadas, Levi Shely, Kaidar Maital, Katzir Ze'ev, Angel-Korman Avital, Becker-Cohen Rachel, Ben-Shalom Efrat, Leiba Adi, Mor Eytan, Dagan Amit, Pessach Itai M, Lotan Danny, Shashar Moshe, Anikster Yair, Raas-Rothschild Annick, Rechavi Gideon, Dekel Benjamin, Vivante Asaf. A multidisciplinary nephrogenetic referral clinic for children and adults-diagnostic achievements and insights.. Pediatric nephrology (Berlin, Germany). 2022. DOI: 10.1007/s00467-021-05374-4.

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