Evidence map›Paper›PMID 32569089›Full record

ArticlePain2020

Loss of SLC9A6/NHE6 impairs nociception in a mouse model of Christianson syndrome.

Hugues Petitjean, Tarheen Fatima, Stephanie Mouchbahani-Constance, Albena Davidova, Catherine E Ferland, John Orlowski, Reza Sharif-Naeini

Open access · greenAbstract read
In one paragraph

Article in Pain, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 16 papers.

0numbers the graph read from it
0cells of the map it votes in
16citing papers in PubMed
1.4field-weighted citation impact, top 19% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

16 citing papers in PubMed, 26 citations in OpenAlex.

  1. Article
  2. The Recycling Endosomal (NaActa physiologica (Oxford, England) · 2026
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  15. The therapeutic importance of acid-base balance.Biochemical pharmacology · 2021
    Review
  16. Article
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

7 authors at 1 institution in 1 country.

Hugues PetitjeanDepartment of Physiology, Cell Information Systems Group, McGill University, Montreal, QC, Canada.
Tarheen FatimaDepartment of Physiology, Cell Information Systems Group, McGill University, Montreal, QC, Canada.
Stephanie Mouchbahani-ConstanceDepartment of Physiology, Cell Information Systems Group, McGill University, Montreal, QC, Canada.
Albena DavidovaDepartment of Physiology, Cell Information Systems Group, McGill University, Montreal, QC, Canada.
Catherine E FerlandDepartment of Anesthesia, Shriners Hospital for Children-Canada, McGill University, Montreal, QC, Canada.
John OrlowskiDepartment of Physiology, Cell Information Systems Group, McGill University, Montreal, QC, Canada.
Reza Sharif-NaeiniDepartment of Physiology, Cell Information Systems Group, McGill University, Montreal, QC, Canada.
McGill University · CA

Funding

CIHR MOP-130471CIHR PJT-166165
6 · The paper itself

Abstract

Children diagnosed with Christianson syndrome (CS), a rare X-linked neurodevelopmental disorder characterized by intellectual disability, epilepsy, ataxia, and mutism, also suffer from hyposensitivity to pain. This places them at risk of sustaining serious injuries that often go unattended. Christianson syndrome is caused by mutations in the alkali cation/proton exchanger SLC9A6/NHE6 that regulates recycling endosomal pH homeostasis and trafficking. Yet, it remains unclear how defects in this transporter lead to altered somatosensory functions. In this study, we validated a Nhe6 knockout (KO) mouse as a model of CS and used it to identify the cellular mechanisms underlying the elevated pain tolerance observed in CS patients. Within the central nervous system, NHE6 immunolabelling is detected in a small percentage of cortical neurons involved in pain processing, including those within the primary somatosensory and the anterior cingulate cortices as well as the periaqueductal gray. Interestingly, it is expressed in a larger percentage of nociceptors. Behaviourally, Nhe6 KO mice have decreased nocifensive responses to acute noxious thermal, mechanical, and chemical (ie, capsaicin) stimuli. The reduced capsaicin sensitivity in the KO mice correlates with a decreased expression of the transient receptor potential channel TRPV1 at the plasma membrane and capsaicin-induced Ca influx in primary cultures of nociceptors. These data indicate that NHE6 is a significant determinant of nociceptor function and pain behaviours, vital sensory processes that are impaired in CS.

Indexed as

AtaxiaEpilepsyGenetic Diseases, X-LinkedIntellectual DisabilityMicrocephalyOcular Motility DisordersAnimalsCapsaicinHumansMiceMice, Inbred C57BLNociceptionNociceptorsSodium-Hydrogen ExchangersTRPV Cation ChannelsCapsaicinNHE6 protein, mouseSodium-Hydrogen ExchangersTRPV Cation Channels

Identifiers

PMID32569089
PMCPMC7566297
OpenAlexW3035881374

What OpenQuestion holds

Textmetadata
LicenceCC BY-NC-ND
Read underepoch 390

Registered trials

None linked

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.