Table 3_Schimmelpenning-Feuerstein-Mims syndrome: a systematic review of clinical cases to identify genotype-phenotype associations.docx

Background/objectives<p>Schimmelpenning-Feuerstein-Mims syndrome (SFMS) is a rare genodermatosis from the group of epidermal nevus syndromes with a wide range of clinical manifestations, including linear nevus sebaceous, ophthalmological disorders, cardiovascular abnormalities, nervous system...

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Κύριος συγγραφέας: Ekaterina Zelenova (8658819) (author)
Άλλοι συγγραφείς: Tatiana Belysheva (22801718) (author), Elena Sharapova (22801721) (author), Vera Semenova (22801724) (author), Timur Valiev (22801727) (author), Tatiana Nasedkina (22801730) (author)
Έκδοση: 2025
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_version_ 1851480399669100544
author Ekaterina Zelenova (8658819)
author2 Tatiana Belysheva (22801718)
Elena Sharapova (22801721)
Vera Semenova (22801724)
Timur Valiev (22801727)
Tatiana Nasedkina (22801730)
author2_role author
author
author
author
author
author_facet Ekaterina Zelenova (8658819)
Tatiana Belysheva (22801718)
Elena Sharapova (22801721)
Vera Semenova (22801724)
Timur Valiev (22801727)
Tatiana Nasedkina (22801730)
author_role author
dc.creator.none.fl_str_mv Ekaterina Zelenova (8658819)
Tatiana Belysheva (22801718)
Elena Sharapova (22801721)
Vera Semenova (22801724)
Timur Valiev (22801727)
Tatiana Nasedkina (22801730)
dc.date.none.fl_str_mv 2025-12-08T06:18:50Z
dc.identifier.none.fl_str_mv 10.3389/fmed.2025.1681584.s001
dc.relation.none.fl_str_mv https://figshare.com/articles/dataset/Table_3_Schimmelpenning-Feuerstein-Mims_syndrome_a_systematic_review_of_clinical_cases_to_identify_genotype-phenotype_associations_docx/30817592
dc.rights.none.fl_str_mv CC BY 4.0
info:eu-repo/semantics/openAccess
dc.subject.none.fl_str_mv Foetal Development and Medicine
Schimmelpenning-Feuerstein-Mims syndrome
KRAS
HRAS
NRAS
mutation
phenotype
mosaicism
dermal nevus
dc.title.none.fl_str_mv Table 3_Schimmelpenning-Feuerstein-Mims syndrome: a systematic review of clinical cases to identify genotype-phenotype associations.docx
dc.type.none.fl_str_mv Dataset
info:eu-repo/semantics/publishedVersion
dataset
description Background/objectives<p>Schimmelpenning-Feuerstein-Mims syndrome (SFMS) is a rare genodermatosis from the group of epidermal nevus syndromes with a wide range of clinical manifestations, including linear nevus sebaceous, ophthalmological disorders, cardiovascular abnormalities, nervous system and musculoskeletal system lesions. Benign and malignant neoplasms, precocious puberty, lymphedema and malformations of the lymphatic system may also be present. The main cause of the syndrome are postzygotic mutations in the RAS pathway, namely in the HRAS, KRAS, and NRAS genes. The aim of our work was to analyze previously described and genetically confirmed clinical cases of SFMS in order to reveal genotype-phenotype correlations.</p>Methods<p>A systematic literature review (SLR) was conducted in several databases, including PubMed and ResearchGate to search for clinical cases of SFMS with genetic testing data from 1946 to 2025. The criterion for inclusion was considered to be the reference to SFMS or its synonyms in article titles with no limitations on language or date of publication. Also, no restrictions on patient age, ethnicity, presence/absence of photo-materials were applied. Only original articles reporting clinical case or case series with detection of postzygotic mutations in the HRAS, KRAS or NRAS genes were included.</p>Results<p>Currently, 50 patients with SFMS and identified pathogenic variants in the HRAS (n = 17), KRAS (n = 30), or NRAS (n = 3) genes have been described in the literature. Analyzing presented phenotypic features of genetically confirmed cases of SFMS depending on the affected gene, we found that ophthalmological and central nervous system anomalities were more common for the KRAS-mut patients (p < 0.05). In contrast, patients with HRAS mutations were more likely to have skeletal abnormalities (p < 0.05). Benign and malignant tumors were diagnosed with equal frequency in both genetic groups, but renal tumors were strongly associated with KRAS mutations.</p>Conclusion<p>The data obtained indicates the necessity of genetic testing for all patients suspected of SFMS to determine the individual risk of comorbidities and to form a personalized plan for dynamic follow-up.</p>Systematic review registration<p>https://www.crd.york.ac.uk/PROSPERO/view/CRD420251085872, identifier CRD420251085872.</p>
eu_rights_str_mv openAccess
id Manara_f27f74e66a3e23cd71023b34e60f9625
identifier_str_mv 10.3389/fmed.2025.1681584.s001
network_acronym_str Manara
network_name_str ManaraRepo
oai_identifier_str oai:figshare.com:article/30817592
publishDate 2025
repository.mail.fl_str_mv
repository.name.fl_str_mv
repository_id_str
rights_invalid_str_mv CC BY 4.0
spelling Table 3_Schimmelpenning-Feuerstein-Mims syndrome: a systematic review of clinical cases to identify genotype-phenotype associations.docxEkaterina Zelenova (8658819)Tatiana Belysheva (22801718)Elena Sharapova (22801721)Vera Semenova (22801724)Timur Valiev (22801727)Tatiana Nasedkina (22801730)Foetal Development and MedicineSchimmelpenning-Feuerstein-Mims syndromeKRASHRASNRASmutationphenotypemosaicismdermal nevusBackground/objectives<p>Schimmelpenning-Feuerstein-Mims syndrome (SFMS) is a rare genodermatosis from the group of epidermal nevus syndromes with a wide range of clinical manifestations, including linear nevus sebaceous, ophthalmological disorders, cardiovascular abnormalities, nervous system and musculoskeletal system lesions. Benign and malignant neoplasms, precocious puberty, lymphedema and malformations of the lymphatic system may also be present. The main cause of the syndrome are postzygotic mutations in the RAS pathway, namely in the HRAS, KRAS, and NRAS genes. The aim of our work was to analyze previously described and genetically confirmed clinical cases of SFMS in order to reveal genotype-phenotype correlations.</p>Methods<p>A systematic literature review (SLR) was conducted in several databases, including PubMed and ResearchGate to search for clinical cases of SFMS with genetic testing data from 1946 to 2025. The criterion for inclusion was considered to be the reference to SFMS or its synonyms in article titles with no limitations on language or date of publication. Also, no restrictions on patient age, ethnicity, presence/absence of photo-materials were applied. Only original articles reporting clinical case or case series with detection of postzygotic mutations in the HRAS, KRAS or NRAS genes were included.</p>Results<p>Currently, 50 patients with SFMS and identified pathogenic variants in the HRAS (n = 17), KRAS (n = 30), or NRAS (n = 3) genes have been described in the literature. Analyzing presented phenotypic features of genetically confirmed cases of SFMS depending on the affected gene, we found that ophthalmological and central nervous system anomalities were more common for the KRAS-mut patients (p < 0.05). In contrast, patients with HRAS mutations were more likely to have skeletal abnormalities (p < 0.05). Benign and malignant tumors were diagnosed with equal frequency in both genetic groups, but renal tumors were strongly associated with KRAS mutations.</p>Conclusion<p>The data obtained indicates the necessity of genetic testing for all patients suspected of SFMS to determine the individual risk of comorbidities and to form a personalized plan for dynamic follow-up.</p>Systematic review registration<p>https://www.crd.york.ac.uk/PROSPERO/view/CRD420251085872, identifier CRD420251085872.</p>2025-12-08T06:18:50ZDatasetinfo:eu-repo/semantics/publishedVersiondataset10.3389/fmed.2025.1681584.s001https://figshare.com/articles/dataset/Table_3_Schimmelpenning-Feuerstein-Mims_syndrome_a_systematic_review_of_clinical_cases_to_identify_genotype-phenotype_associations_docx/30817592CC BY 4.0info:eu-repo/semantics/openAccessoai:figshare.com:article/308175922025-12-08T06:18:50Z
spellingShingle Table 3_Schimmelpenning-Feuerstein-Mims syndrome: a systematic review of clinical cases to identify genotype-phenotype associations.docx
Ekaterina Zelenova (8658819)
Foetal Development and Medicine
Schimmelpenning-Feuerstein-Mims syndrome
KRAS
HRAS
NRAS
mutation
phenotype
mosaicism
dermal nevus
status_str publishedVersion
title Table 3_Schimmelpenning-Feuerstein-Mims syndrome: a systematic review of clinical cases to identify genotype-phenotype associations.docx
title_full Table 3_Schimmelpenning-Feuerstein-Mims syndrome: a systematic review of clinical cases to identify genotype-phenotype associations.docx
title_fullStr Table 3_Schimmelpenning-Feuerstein-Mims syndrome: a systematic review of clinical cases to identify genotype-phenotype associations.docx
title_full_unstemmed Table 3_Schimmelpenning-Feuerstein-Mims syndrome: a systematic review of clinical cases to identify genotype-phenotype associations.docx
title_short Table 3_Schimmelpenning-Feuerstein-Mims syndrome: a systematic review of clinical cases to identify genotype-phenotype associations.docx
title_sort Table 3_Schimmelpenning-Feuerstein-Mims syndrome: a systematic review of clinical cases to identify genotype-phenotype associations.docx
topic Foetal Development and Medicine
Schimmelpenning-Feuerstein-Mims syndrome
KRAS
HRAS
NRAS
mutation
phenotype
mosaicism
dermal nevus