Evidence map›Paper›PMID 41536045›Full record

ArticleThe American journal of case reports2026

Phenotypic Spectrum of Type 2-3 Gaucher Disease: A Case Study in the Balkan Genotype.

Paskal Cullufi, Virtut Velmishi, Ermira Dervishi, Sonila Tomori, Gladiola Hoxha, Eda Jazexhiu-Postoli, Entela Basha, Aferdita Kumaraku Tako, Mirela Tabaku

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Article in The American journal of case reports, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

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0cells of the map it votes in
1citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

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2 · The registry

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3 · Its place in the literature

Who cites it

1 citing paper in PubMed.

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4 · The record

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5 · Who and what money

Authors and funding

9 authors.

Paskal CullufiDepartment of Pediatric, Mother Teresa Hospital, University of Medicine of Tirana, Tirana, Albania.ORCID 0000-0001-5678-9926
Virtut VelmishiDepartment of Pediatric, Mother Teresa Hospital, University of Medicine of Tirana, Tirana, Albania.ORCID 0000-0003-1648-7258
Ermira DervishiDepartment of Pediatric, Mother Teresa Hospital, University of Medicine of Tirana, Tirana, Albania.
Sonila TomoriDepartment of Pediatric, Mother Teresa Hospital, University of Medicine of Tirana, Tirana, Albania.ORCID 0000-0001-6451-9088
Gladiola HoxhaDepartment of Pediatric, Mother Teresa Hospital, University of Medicine of Tirana, Tirana, Albania.
Eda Jazexhiu-PostoliNeonatology Service, International Hospital Hygeia, Tirana, Albania.ORCID 0009-0004-4783-9679
Entela BashaDepartment of Neuro Science, Mother Teresa Hospital, Tirana, Albania.ORCID 0009-0005-4118-9203
Aferdita Kumaraku TakoDepartment of Pediatric, Mother Teresa Hospital, University of Medicine of Tirana, Tirana, Albania.
Mirela TabakuDepartment of Pediatric, Mother Teresa Hospital, University of Medicine of Tirana, Tirana, Albania.ORCID 0000-0001-8985-6785

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

BACKGROUND Gaucher disease (GD) is a lysosomal storage disorder caused by mutations in the glucosylceramidase beta 1 (GBA1) gene, with a wide range of clinical manifestations. Type 2 GD, the acute neuronopathic form, is the most severe and has historically been considered a distinct clinical entity. Emerging evidence suggests a broader phenotypic spectrum, including intermediate forms that do not fully align with the diagnostic criteria for either type 2 or type 3 GD. Here, we describe an Albanian patient with an unusual intermediate type 2-3 phenotype, homozygous for the rare GBA1 complex allele p.[His294Gln;Asp448His] that is frequently found in Balkan populations. Although this genotype is typically associated with early-onset type 2 GD and death within the first 2 years of life, our patient developed neurologic symptoms at 15 months and survived until age 5. CASE REPORT A male Albanian infant presented at 4 months of age with hepatosplenomegaly and thrombocytopenia. Genetic testing revealed homozygosity for the rare GBA1 complex allele p.[His294Gln;Asp448His]. Enzyme replacement therapy, initiated at 7 months, led to temporary improvement of visceral and hematological symptoms. Neurologic manifestations - including oculomotor apraxia, dystonia, and seizures - emerged at 15 months. Despite continued enzyme replacement therapy and supportive care, the patient's neurologic condition progressively worsened, and he died at age 5 due to disease progression and pulmonary complications. CONCLUSIONS This case reinforces the concept that type 2 GD exists on a phenotypic spectrum, rather than as a uniform clinical entity, and underscores the broad genotype-phenotype variability in GD.

Indexed as

Gaucher DiseaseGlucosylceramidaseAlbaniaGenotypeHomozygoteHumansInfantMaleMutationPhenotypeGlucosylceramidase

Identifiers

PMID41536045
PMCPMC12822607

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.