Evaluation of the Patients with the Diagnosis of Pontocerebellar Hypoplasia: A Multicenter National Study
Open accessAbstract
Pontocerebellar hypoplasia (PCH) is a heterogeneous group of neurodegenerative disorders characterized by hypoplasia and degeneration of the cerebellum and pons. We aimed to identify the clinical, laboratory, and imaging findings of the patients with diagnosed PCH with confirmed genetic analysis. We collected available clinical data, laboratory, and imaging findings in our retrospective multicenter national study of 64 patients with PCH in Turkey. The genetic analysis included the whole-exome sequencing (WES), targeted next-generation sequencing (NGS), or single gene analysis. Sixty-four patients with PCH were 28 female (43.8%) and 36 (56.3%) male. The patients revealed homozygous mutation in 89.1%, consanguinity in 79.7%, pregnancy at term in 85.2%, microcephaly in 91.3%, psychomotor retardation in 98.4%, abnormal neurological findings in 100%, seizure in 63.8%, normal biochemistry and metabolic investigations in 92.2%, and dysmorphic findings in 51.2%. The missense mutation was found to be the most common variant type in all patients with PCH. It was detected as CLP1 (n = 17) was the most common PCH related gene. The homozygous missense variant c.419G > A (p.Arg140His) was identified in all patients with CLP1. Moreover, all patients showed the same homozygous missense variant c.919G > T (p.A307S) in TSEN54 group (n = 6). In Turkey, CLP1 was identified as the most common causative gene with the identical variant c.419G > A; p.Arg140His. The current study supports that genotype data on PCH leads to phenotypic variability over a wide phenotypic spectrum.
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PCH1B patients who are carriers of compound heterozygous EXOSC3 D132A/G191D variants present with milder disease, without involving the pons, and show intrafamilial variability.
“PCH1B patients who are carriers of compound heterozygous EXOSC3 D132A/G191D variants present with milder disease, without involving the pons, and show intrafamilial variability23,53,54.”
From EXOSC3 G191 Variants Trigger System-Wide Recalibration of RNA Processing Machinery (2026) · cites this paper as [53]
The Discussion (page unavailable) identifies D132A and G191D in patients with PCH1B and describes phenotypes associated with both variants as moderate. It reports those variants as homozygous in its patients, not as a D132A/G191D compound-heterozygous pair. The cited passages do not establish absence of pontine involvement or intrafamilial variability. (LLM, conf=0.86)
Experimental — accuracy not yet measuredThe lesser thermodynamic severity of the G191C variant compared to G191D correlates well with less severe clinical outcomes observed in G191C homozygous patients compared to G191D homozygous patients.
“Notably, the lesser thermodynamic severity of the G191C variant compared to G191D correlates well with less severe clinical outcomes observed in G191C homozygous patients (previously classified with complicated hereditary spastic paraplegia) compared to G191D homozygous patients (classified with moderate PCH1b) 23,53,54.”
From EXOSC3 G191 Variants Trigger System-Wide Recalibration of RNA Processing Machinery (2026) · cites this paper as [53]
The Discussion (page unavailable) reports three patients homozygous for Gly191Asp and describes the phenotype associated with that variant as moderate, supporting the G191D/PCH1B part of the sentence. It does not discuss G191C, thermodynamic severity, or a clinical comparison between G191C and G191D. (LLM, conf=0.88)
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“…severity of the G191C variant compared to G191D correlates well with less severe clinical outcomes observed in G191C homozygous patients (previously classified with complicated hereditary spastic paraplegia) compared to G191D homozygous patients (classified with moderate PCH1b) 23,53,54 .”
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- Authors
- Cavusoglu, D., Ozturk, G., Turkdogan, D., Kurul, S. H., Yis, U., Komur, M., Incecik, F., Kara, B., Sahin, T., Unver, O., Dilber, C., Mert, G. G., Gunay, C., Uzan, G. S., Ersoy, O., Oktay, Y., Mermer, S., Tuncer, G. O., Gungor, O., Ozcora, G. D. K., Gumus, U., Sezer, O., Cetin, G. O., Demir, F., Yilmaz, A., Gurbuz, G., Topcu, M., Topaloglu, H., Ceylan, A. C., Ceylaner, S., Gleeson, J. G., Icagasioglu, D. F., & Sonmez, F. M.
- Affiliations
- Bezmiâlem Vakıf Üniversitesi (TR)
- Marmara University (TR)
- Kahramanmaraş Sütçü İmam University (TR)
- Dokuz Eylül University (TR)
- Mersin Üniversitesi (TR)
- Ankara Bilkent City Hospital (TR)
- Intergen (Turkey) (TR)
- Howard Hughes Medical Institute (US)
- Karadeniz Technical University (TR)
- Corresponding author
- Sonmez, F. Mujgan
- ORCID
- Venue
- The Cerebellum
- Published
- 2024-04-15
- Volume
- 23
- Issue
- 5
- Pages
- 1950-1965
- ISSN
- 1473-4222 (ISSN-L)
- 1473-4230
- PMC
- PMC11489189
- OpenAlex
- W4394804744
- Cited by
- 8
- References
- 52
- Citations by year
- 2026: 3 · 2025: 4 · 2024: 1
- FWCI
- 5.42
- Type
- journal article
- Language
- en
- Open access
- hybrid
- doi.org
- Locations
- The Cerebellum (publishedVersion, cc-by)
- PubMed Central (publishedVersion, cc-by)
- PubMed (publishedVersion)
- AYBU AVESIS (submittedVersion, cc-by-nc-nd)
- Hasan Kalyoncu University Institutional Repository (Hasan Kalyoncu University) (submittedVersion)
- Yeditepe University Institutional Repository (submittedVersion, other-oa)
- Funders
- Karadeniz Technical University
- Topics
- Health Sciences › Medicine › Pediatrics, Perinatology and Child Health › Fetal and Pediatric Neurological Disorders
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Clinical Biochemistry › Metabolism and Genetic Disorders
- Life Sciences › Neuroscience › Neurology › Neurological diseases and metabolism
- Keywords
- pontocerebellar hypoplasia, Clp1, homozygous missense mutation, consanguinity, microcephaly, psychomotor retardation, seizures, dysmorphic features, genotype-phenotype correlation, phenotypic variability, neurodegenerative diseases, Turkey
- MeSH terms
- Adolescent, Adult, Cerebellar Diseases, Child, Child, Preschool, Consanguinity, Female, Humans, Infant, Male, Mutation, Retrospective Studies, Turkey, Young Adult
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