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SHH – Solitary Median Maxillary Central Incisor Syndrome

Solitary median maxillary central incisor syndrome (SMMCI) represents the mildest manifestation of the holoprosencephaly spectrum and occurs in approximately 1 in 50 000 live births. In a cohort of six patients, array comparative genomic hybridization identified a heterozygous deletion at 7q36 encompassing SHH in one individual, implicating SHH haploinsufficiency in SMMCI pathogenesis (PMID:26080100).

No additional SHH‐specific variants or familial segregation data have been reported in SMMCI, and to date there are no functional studies directly assessing SHH in this context. Thus, current evidence for a SHH–SMMCI association remains limited and warrants further investigation.

References

  • American journal of medical genetics. Part A | 2015 | Genotypic and phenotypic variation in six patients with solitary median maxillary central incisor syndrome. PMID:26080100

Evidence Based Scoring (AI generated)

Gene–Disease Association

Limited

Single patient with heterozygous deletion at 7q36 including SHH, no segregation ([PMID:26080100])

Genetic Evidence

Limited

One proband with SHH‐containing deletion, lacking additional cases or segregation data ([PMID:26080100])

Functional Evidence

No evidence

No functional studies linking SHH to solitary median maxillary central incisor syndrome