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Medical Genetics · DNAJC21-Related Shwachman-Diamond Syndrome

Understanding DNAJC21-Related Shwachman-Diamond Syndrome

At a Glance

DNAJC21-related Shwachman-Diamond syndrome is an ultra-rare genetic condition that disrupts the structures cells use to make proteins and may cause bone marrow failure, pancreatic problems, growth or developmental differences. Monitoring is individualized because cases vary widely.

Receiving a diagnosis for an ultra-rare disease like DNAJC21-related Shwachman-Diamond Syndrome (SDS) can feel overwhelming. You may find that many doctors you encounter have never seen a case before, which can lead to a sense of isolation [1]. Please know that while the name is long and the condition is rare, there is a dedicated community of researchers and families working to understand and manage this disease [NCT06999954].

DNAJC21-related SDS is a genetic condition that belongs to a group of disorders called ribosomopathies—diseases caused by problems with how cells build ribosomes, the “factories” that make proteins [2]. Because proteins are essential for almost every function in the body, these defects can lead to a wide range of symptoms affecting the blood, growth, and various organs [3].

How Rare is This Condition?

DNAJC21-related SDS is significantly rarer than the more common version of the syndrome (caused by the SBDS gene). As of late 2023, fewer than 20 cases had been documented in the global medical literature, though true prevalence is unknown [1][3].

Because so few cases exist, doctors are still learning about the full spectrum of the disease. While early reports focused on severe childhood cases, more recent research has identified adults with milder symptoms, such as low platelet counts, suggesting the condition may be more variable than originally thought [1][4]. Keep in mind that clinical features and prognosis described below are drawn from small case series rather than large, reliable population categories.

The Biological “Glitch”: DNAJC21 vs. SBDS

To understand why this condition happens, it helps to look at how cells build their protein factories. Ribosomes are made of two main parts: the small subunit (40S) and the large subunit (60S). Both forms of SDS involve problems making the 60S subunit, but they happen at different times:

  • Standard SDS (SBDS gene): Think of this as a problem late in the assembly line. A factor called eIF6 must be removed before the factory can start working. The SBDS protein works with EFL1 to promote this release [5].
  • DNAJC21-related SDS: This involves a problem in early 60S ribosomal maturation. The DNAJC21 protein is needed for pre-rRNA processing and gathering the early components for the 60S subunit [2].

When DNAJC21 is missing or not working correctly, the cell cannot mature enough 60S subunits. This leads to aberrant ribosome profiles (malformed factories) and increased cell death, particularly in the bone marrow [2].

What to Expect: Clinical Features

DNAJC21-related SDS is a “multisystem” disorder, meaning it can affect many parts of the body. Because it is so rare, not everyone will have every symptom. Features reported in the literature include:

  • Bone Marrow Failure: This is a prominent feature. The bone marrow may not produce enough white blood cells (neutropenia), red blood cells (anemia), or platelets (thrombocytopenia) [3][6].
  • Pancreatic Issues: Some individuals have exocrine pancreatic insufficiency (EPI), where the pancreas doesn’t produce enough enzymes to digest food properly [3][6].
  • Growth and Development: Many individuals experience growth retardation, difficulty gaining weight, or developmental delays [3][4].
  • Physical Findings: Some individuals have unique physical features involving the skin, teeth, or hair. Abnormalities in the retina (back of the eye) or skeletal system, such as hip dysplasia, have also been reported in small case studies [3][4].

Prognosis and Long-Term Outlook

Because the number of known cases is so small, there is no “standard” life expectancy or fixed prognosis for DNAJC21-related SDS [7]. Some have severe bone marrow failure early in life, while others may have a more stable course into adulthood [1].

A primary concern is the risk of MDS (myelodysplastic syndrome) or AML (acute myeloid leukemia), which are blood cancers that can develop from marrow stress [2][3]. While the exact frequency of MDS/AML in DNAJC21 is not fully known, regular monitoring of blood counts and bone marrow—extrapolated from classic SDS guidance—is the standard way to manage this risk [3].

Research and Registries

Since this is an ultra-rare condition, participation in research registries is incredibly valuable, though optional.

  • SDS Registry and Study: Based at Boston Children’s Hospital, this registry explicitly accepts patients with DNAJC21 mutations to study the natural history of the disease [NCT06056908].
  • SDS Global Patient Survey: An online platform that allows families to share their experiences [NCT06999954].
  • NCI Natural History Study: The National Cancer Institute (NCI) conducts a long-term study on inherited bone marrow failure syndromes [NCT00027274].

Common questions in this guide

What is DNAJC21-related Shwachman-Diamond syndrome?
DNAJC21-related Shwachman-Diamond syndrome is an ultra-rare genetic disorder in the ribosomopathy group. Changes in the DNAJC21 gene disrupt early development of the 60S part of ribosomes, the cell structures that help make proteins, and can affect bone marrow, the pancreas, growth, development, and other organs.
How does DNAJC21-related SDS differ from the more common SBDS-related form?
Both forms affect production of the 60S part of the cell's protein-making machinery, but at different stages. The SBDS protein acts later with other factors, while DNAJC21 helps with earlier RNA processing and assembly.
What blood problems can DNAJC21-related SDS cause?
The bone marrow may not make enough neutrophils, red blood cells, or platelets. This can appear as neutropenia, anemia, or thrombocytopenia, and the absolute neutrophil count helps clinicians assess infection risk.
Can DNAJC21-related SDS affect digestion, growth, or development?
Yes. Some people have exocrine pancreatic insufficiency, meaning the pancreas does not make enough digestive enzymes, which may contribute to difficulty gaining weight or oily stools. Growth problems, developmental delays, and changes involving the skin, teeth, hair, eyes, or skeleton have also been reported, but not everyone has the same features.
What is the outlook for someone with DNAJC21-related SDS?
There is no established life expectancy or single standard prognosis because only a small number of cases have been reported. Some people develop severe bone marrow failure in childhood, while others have milder or more stable symptoms into adulthood, so outlook depends on the person's findings and ongoing monitoring.
How are people monitored for leukemia-related changes?
Regular blood counts and bone marrow evaluations are used to watch for worsening marrow problems and possible myelodysplastic syndrome or acute myeloid leukemia. Because DNAJC21-related SDS is very rare, the timing of these checks should be individualized with a specialist, using available Shwachman-Diamond syndrome guidance.
Should families join a DNAJC21 or Shwachman-Diamond syndrome registry?
Research participation is optional but can help doctors understand the condition's natural history and range of symptoms. The SDS Registry at Boston Children's Hospital, an SDS global patient survey, and an NCI natural history study are examples of research opportunities mentioned for affected families.

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References

References (7)
  1. 1

    DNAJC21-related thrombocytopenia in a young adult female.

    Aslan D, Akgun-Dogan O, Ay B, et al.

    American journal of medical genetics. Part C, Seminars in medical genetics 2023; (193(2)):193-197 doi:10.1002/ajmg.c.32043.

    PMID: 37186482
  2. 2

    DNAJC21 Mutations Link a Cancer-Prone Bone Marrow Failure Syndrome to Corruption in 60S Ribosome Subunit Maturation.

    Tummala H, Walne AJ, Williams M, et al.

    American journal of human genetics 2016; (99(1)):115-24.

    PMID: 27346687
  3. 3

    Refining the phenotype associated with biallelic DNAJC21 mutations.

    D'Amours G, Lopes F, Gauthier J, et al.

    Clinical genetics 2018; (94(2)):252-258 doi:10.1111/cge.13370.

    PMID: 29700810
  4. 4

    A Rare Inherited Bone Marrow Failure Syndrome Disclosed by Reanalysis of the Exome Data of a Patient Evaluated for Cytopenia and Dysmorphic Features.

    Durmaz D, Aslanger AD, Yavas Abali Z, et al.

    Journal of pediatric hematology/oncology 2024; (46(3)):e214-e219 doi:10.1097/MPH.0000000000002839.

    PMID: 38408162
  5. 5

    Predisposition to myeloid malignancies in Shwachman-Diamond syndrome: biological insights and clinical advances.

    Reilly CR, Shimamura A

    Blood 2023; (141(13)):1513-1523 doi:10.1182/blood.2022017739.

    PMID: 36542827
  6. 6

    Shwachman Diamond syndrome: narrow genotypic spectrum and variable clinical features.

    Thompson AS, Giri N, Gianferante DM, et al.

    Pediatric research 2022; (92(6)):1671-1680 doi:10.1038/s41390-022-02009-8.

    PMID: 35322185
  7. 7

    Shwachman-Diamond syndromes: clinical, genetic, and biochemical insights from the rare variants.

    Kawashima N, Oyarbide U, Cipolli M, et al.

    Haematologica 2023; (108(10)):2594-2605 doi:10.3324/haematol.2023.282949.

    PMID: 37226705

This page is for informational purposes only and does not constitute medical advice. Your genetics and hematology team can help interpret your specific findings, monitoring needs, and prognosis.