Wolfram Syndrome and WFS1-related Disorders International Registry and Clinical Study

Description

In this study, the investigators hypothesize that studying monogenic variants with strong effect associated with severe insulin deficiency of Wolfram syndrome will provide important insights into the more complex type 1 and type 2 diabetes mellitus. Aim 1. Establish and maintain a registry of patients with Wolfram syndrome. An Internet based registry will be employed to enroll participants with the clinical diagnosis of Wolfram syndrome (insulin dependent DM and bilateral OA). Clinical information regarding age of diagnosis and progression of the disease will be collated and analyzed to better define its natural history, along with potential metabolic phenotypes such as glucose intolerance of heterozygous parents and unaffected sibs. If not already completed, blood for WFS1 sequence analysis will be obtained on the participants (parents and sibs also for control purposes) and sent to a CLIA certified lab to define the mutation. This information will benefit patient families and referring physicians by providing a genetic diagnosis and where indicated. The Wolfram Syndrome Registry will foster international collaborations to more efficiently and systematically collect Wolfram syndrome patients and their clinical and experimental data.

Conditions

Wolfram Syndrome, Diabetes Mellitus, Optic Nerve Atrophy, Deafness, Diabetes Insipidus, Ataxia

Study Overview

Study Details

Study overview

In this study, the investigators hypothesize that studying monogenic variants with strong effect associated with severe insulin deficiency of Wolfram syndrome will provide important insights into the more complex type 1 and type 2 diabetes mellitus. Aim 1. Establish and maintain a registry of patients with Wolfram syndrome. An Internet based registry will be employed to enroll participants with the clinical diagnosis of Wolfram syndrome (insulin dependent DM and bilateral OA). Clinical information regarding age of diagnosis and progression of the disease will be collated and analyzed to better define its natural history, along with potential metabolic phenotypes such as glucose intolerance of heterozygous parents and unaffected sibs. If not already completed, blood for WFS1 sequence analysis will be obtained on the participants (parents and sibs also for control purposes) and sent to a CLIA certified lab to define the mutation. This information will benefit patient families and referring physicians by providing a genetic diagnosis and where indicated. The Wolfram Syndrome Registry will foster international collaborations to more efficiently and systematically collect Wolfram syndrome patients and their clinical and experimental data.

Wolfram Syndrome and WFS1-related Disorders International Registry and Clinical Study

Wolfram Syndrome and WFS1-related Disorders International Registry and Clinical Study

Condition
Wolfram Syndrome
Intervention / Treatment

-

Contacts and Locations

Saint Louis

Washington University School of Medicine, Saint Louis, Missouri, United States, 63110

Participation Criteria

Researchers look for people who fit a certain description, called eligibility criteria. Some examples of these criteria are a person's general health condition or prior treatments.

For general information about clinical research, read Learn About Studies.

Eligibility Criteria

  • * Diabetes mellitus \<16 yrs
  • * Optic atrophy \<16 yrs
  • * Diabetes insipidus
  • * Diabetes mellitus \>16yrs
  • * Optic atrophy \>16 yrs
  • * Sensorineural deafness
  • * Neurological signs (ataxia, epilepsy, cognitive impairment)
  • * Renal tract abnormalities (structural or functional)
  • * 1 loss of function mutation in WFS1/CISD2 AND/OR family history of Wolfram syndrome
  • * 2 major OR
  • * 1 major plus 2 minor criteria OR
  • * 2 pathological WFS1 or CISD2 mutations are identified
  • * Hypogonadism (males)
  • * An absence of type 1 diabetes auto-antibodies
  • * Bilateral cataracts
  • * Psychiatric disorder
  • * Gastrointestinal

Ages Eligible for Study

0 Years to

Sexes Eligible for Study

ALL

Accepts Healthy Volunteers

No

Collaborators and Investigators

Washington University School of Medicine,

Study Record Dates

2026-04