Case report: Hereditary sensory autonomic neuropathy presenting as bifid deformity to the tongue
Hereditary sensory autonomic neuropathy (HSAN) is a group of rare genetic disorders in which affected patients have a diminished capacity to feel pain. Patients with HSAN may present with a wide range of factitial injuries, where injury to the oral cavity may be an early presenting sign. While exist...
Main Authors: | , , , |
---|---|
Format: | Article |
Language: | English |
Published: |
Frontiers Media S.A.
2023-10-01
|
Series: | Frontiers in Dental Medicine |
Subjects: | |
Online Access: | https://www.frontiersin.org/articles/10.3389/fdmed.2023.1179795/full |
_version_ | 1797655882684694528 |
---|---|
author | Kelsey O’Hagan-Wong Dana Smith Hernan Gonorazky Michael Casas |
author_facet | Kelsey O’Hagan-Wong Dana Smith Hernan Gonorazky Michael Casas |
author_sort | Kelsey O’Hagan-Wong |
collection | DOAJ |
description | Hereditary sensory autonomic neuropathy (HSAN) is a group of rare genetic disorders in which affected patients have a diminished capacity to feel pain. Patients with HSAN may present with a wide range of factitial injuries, where injury to the oral cavity may be an early presenting sign. While existing literature on HSAN is scant, many reports highlight the long-term outcomes that may include enucleation of eyes, amputation of fingers and limbs, and disfigurement of the tongue. This case describes a five-month-old female with repetitive injury to the tongue causing it to heal with a bifid deformity. The patient was later diagnosed with HSAN type 4. This case highlights the importance of recognition of extensive oral trauma as one of the early signs of HSAN that should provoke a timely referral for neurological assessment. |
first_indexed | 2024-03-11T17:21:04Z |
format | Article |
id | doaj.art-ed4dbf765848487194674353a7a5c979 |
institution | Directory Open Access Journal |
issn | 2673-4915 |
language | English |
last_indexed | 2024-03-11T17:21:04Z |
publishDate | 2023-10-01 |
publisher | Frontiers Media S.A. |
record_format | Article |
series | Frontiers in Dental Medicine |
spelling | doaj.art-ed4dbf765848487194674353a7a5c9792023-10-19T12:23:44ZengFrontiers Media S.A.Frontiers in Dental Medicine2673-49152023-10-01410.3389/fdmed.2023.11797951179795Case report: Hereditary sensory autonomic neuropathy presenting as bifid deformity to the tongueKelsey O’Hagan-Wong0Dana Smith1Hernan Gonorazky2Michael Casas3Department of Dentistry, University of Toronto, Toronto, CanadaDepartment of Dentistry, The Hospital for Sick Children, University of Toronto, Toronto, ON, CanadaDepartment of Dentistry, The Hospital for Sick Children, University of Toronto, Toronto, ON, CanadaDepartment of Dentistry, The Hospital for Sick Children, University of Toronto, Toronto, ON, CanadaHereditary sensory autonomic neuropathy (HSAN) is a group of rare genetic disorders in which affected patients have a diminished capacity to feel pain. Patients with HSAN may present with a wide range of factitial injuries, where injury to the oral cavity may be an early presenting sign. While existing literature on HSAN is scant, many reports highlight the long-term outcomes that may include enucleation of eyes, amputation of fingers and limbs, and disfigurement of the tongue. This case describes a five-month-old female with repetitive injury to the tongue causing it to heal with a bifid deformity. The patient was later diagnosed with HSAN type 4. This case highlights the importance of recognition of extensive oral trauma as one of the early signs of HSAN that should provoke a timely referral for neurological assessment.https://www.frontiersin.org/articles/10.3389/fdmed.2023.1179795/fullpediatric dentistrycongenital insensitivity to painhereditary sensory autonomic neuropathytongue ulcerationbifid deformity of the tongue |
spellingShingle | Kelsey O’Hagan-Wong Dana Smith Hernan Gonorazky Michael Casas Case report: Hereditary sensory autonomic neuropathy presenting as bifid deformity to the tongue Frontiers in Dental Medicine pediatric dentistry congenital insensitivity to pain hereditary sensory autonomic neuropathy tongue ulceration bifid deformity of the tongue |
title | Case report: Hereditary sensory autonomic neuropathy presenting as bifid deformity to the tongue |
title_full | Case report: Hereditary sensory autonomic neuropathy presenting as bifid deformity to the tongue |
title_fullStr | Case report: Hereditary sensory autonomic neuropathy presenting as bifid deformity to the tongue |
title_full_unstemmed | Case report: Hereditary sensory autonomic neuropathy presenting as bifid deformity to the tongue |
title_short | Case report: Hereditary sensory autonomic neuropathy presenting as bifid deformity to the tongue |
title_sort | case report hereditary sensory autonomic neuropathy presenting as bifid deformity to the tongue |
topic | pediatric dentistry congenital insensitivity to pain hereditary sensory autonomic neuropathy tongue ulceration bifid deformity of the tongue |
url | https://www.frontiersin.org/articles/10.3389/fdmed.2023.1179795/full |
work_keys_str_mv | AT kelseyohaganwong casereporthereditarysensoryautonomicneuropathypresentingasbifiddeformitytothetongue AT danasmith casereporthereditarysensoryautonomicneuropathypresentingasbifiddeformitytothetongue AT hernangonorazky casereporthereditarysensoryautonomicneuropathypresentingasbifiddeformitytothetongue AT michaelcasas casereporthereditarysensoryautonomicneuropathypresentingasbifiddeformitytothetongue |