Skip to main content
SpringerLink
Log in

Isolated hypohidrosis: pathogenesis and treatment

  • Clinical report
  • Published:
European Journal of Dermatology

Abstract

Background

Hypohidrosis can result in heat injury, a potentially fatal condition. The majority of hypohidrosis cases have no associated abnormalities or secondary causes, and are termed “isolated hypohidrosis”. These are clinically divided into miliaria profunda (MP), acquired idiopathic generalized anhidrosis (AIGA) and idiopathic partial hypohidrosis (IPH). The pathogenesis of isolated hypohidrosis remains largely unknown and there is no established effective treatment.

Objectives

To elucidate the pathogenesis of isolated hypohidrosis using in vivo high-definition optical coherence tomography (HD-OCT) imaging and assess the therapeutic profile of oral retinoids for this condition.

Materials & Methods

We conducted a retrospective analysis on all patients with isolated hypohidrosis in our neuro-dermatology clinic over a 5.75–year period. All patients routinely underwent standardised exercising and whole-body starch-iodine testing, followed by non-invasive HD-OCT skin imaging. Patients’ demographics, disease characteristics, histology and treatment history were analysed.

Results

Of the 51 patients identified with isolated hypohidrosis; 23 were diagnosed with MP, 14 with AIGA, and 14 with IPH. In these patients, HD-OCT imaging led to the identification of sub-stratum corneal hypo-refractile material with underlying dilated sweat ducts, not present in healthy controls. The size of this material was most pronounced in MP, followed by AIGA, and then IPH. Post-treatment, the material decreased in size. Treatment response was reported in 90.6% patients with isotretinoin and 75.0% with acitretin. No recurrence has been reported to date. Side effects were largely anticipated and common.

Conclusion

The pathogenesis of isolated hypohidrosis involves obstruction of sweat orifices at the stratum corneum. Treatment with oral retinoids, particularly isotretinoin, is effective and safe.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Log in via an institution

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Institutional subscriptions

Similar content being viewed by others

References

  1. Thami GP, Kaur S, Kanwar AJ. Acquired idiopathic generalized anhidrosis: a rare cause of heat intolerance. Clin Exp Dermatol 2003;28:262–4.

    Article  CAS  Google Scholar 

  2. Lim JH, Kok WL, Ali NB, Chong WS, Tey HL. Hypohidrosis in individuals with exertional heat injury: a prospective open cohort study. Dermatology 2016; 232:50–6.

    Article  Google Scholar 

  3. Hu Y, Converse C, Lyons MC, Hsu WH. Neural control of sweat secretion: a review. Br J Dermatol 2018; 178: 1246–56.

    Article  CAS  Google Scholar 

  4. Roskind JL, Petrofsky J, Lind AR, Paletta FX. Quantitation of thermoregulatory impairment in patients with healed burns. Ann Plast Surg 1978; 1: 172–6.

    Article  CAS  Google Scholar 

  5. Chia KY, Tey HL. Approach to hypohidrosis. J Eur Acad Dermatol Venereol 2013; 27:799–804.

    Article  CAS  Google Scholar 

  6. Tey HL, Tay EY, Cao T. in vivo imaging of miliaria profunda using high-definition optical coherence tomography: diagnosis, pathogenesis, and treatment. JAMA Dermatol 2015; 151: 346–8.

    Article  Google Scholar 

  7. Kirk JF, Wilson BB, Chun W, Cooper PH. Miliaria profunda. J Am Acad Dermatol 1996; 35: 854–6.

    Article  CAS  Google Scholar 

  8. Chen YC, Wu CS, Chen GS, Khor GT, Chen CH, Huang P. Identification of subgroups of acquired idiopathic generalized anhidrosis. Neurologist 2008; 14:318–20.

    Article  Google Scholar 

  9. Munetsugu T, Fujimoto T, Oshima Y, et al. Revised guideline for the diagnosis and treatment of acquired idiopathic generalized anhidrosis in Japan. J Dermatol 2017;44: 394400.

    Google Scholar 

  10. Ohshima Y, Yanagishita T, Ito K, et al. Treatment of patients with acquired idiopathic generalized anhidrosis. Br J Dermatol 2013; 168:430–2.

    Article  CAS  Google Scholar 

  11. Palm F, Löser C, Gronau W, Voigtländer V, Grau AJ. Successful treatment of acquired idiopathic generalized anhidrosis. Neurology 2007; 68:532–3.

    Article  CAS  Google Scholar 

  12. Fukunaga A, Hatakeyama M, Tsujimoto M, Oda Y, Washio K, Nishigori C. Steroid treatment can improve the impaired quality of life of patients with acquired idiopathic generalized anhidrosis. Br J Dermatol 2015;172:537.

    Article  CAS  Google Scholar 

  13. Yoritaka A, Hishima T, Akagi K, Kishida S. Successful steroid treatment of acquired idiopathic partial hypohidrosis. J Dermatol 2006;33:265–7.

    Article  Google Scholar 

  14. Fritsch PO. Retinoids in psoriasis and disorders of keratinization. J Am Acad Dermatol 1992; 27: S8–14.

    Article  CAS  Google Scholar 

  15. von Elm E, Altman DG, Egger M, Pocock SJ, Gøtzsche PC, Vandenbroucke JP. The strengthening the reporting of observational studies in epidemiology (strobe) statement: guidelines for reporting observational studies. Lancet 2007; 370: 1453–7.

    Article  Google Scholar 

  16. Narita K, Sakamoto S, Mizushige K, Senda S, Matsuo H. Development and evaluation of a new target heart rate formula for the adequate exercise training level in healthy subjects. J Cardiol 1999; 33: 265–72.

    CAS  PubMed  Google Scholar 

  17. Lim JL, Choo W, Chang JH, Tey HL, Chong WS. Application of iodinated starch powder using an atomizer spray gun-a new and effective tool to evaluate hypohidrosis. Skin Res Technol 2016; 22: 370–4.

    Article  Google Scholar 

  18. Wallace AB. The exposure treatment of burns. Lancet 1951; 1: 501–4.

    Article  CAS  Google Scholar 

  19. Schneider CA, Rasband WS, Eliceiri KW. NIH Image to ImageJ: 25 years of image analysis. Nat Methods 2012; 9: 671.

    Article  CAS  Google Scholar 

  20. R Core Team. R: A language and environment for statistical computing. Vienna: R Foundation for Statistical Computing; 2019. Available at: https://www.R-project.org.(accessed 11 March 2019).

  21. Haenssle HA, Finkenrath A, Hausser I, et al. Effective treatment of severe thermodysregulation by oral retinoids in a patient with recessive congenital lamellar ichthyosis. Clin Exp Dermatol 2008; 33: 578–81.

    Article  CAS  Google Scholar 

  22. Elias PM. Epidermal effects of retinoids: supramolecular observations and clinical implications. J Am Acad Dermatol 1986; 15: 797–809.

    Article  CAS  Google Scholar 

  23. Törmä H. Regulation of keratin expression by retinoids. Dermatoendocrinol 2011; 3: 136–40.

    Article  Google Scholar 

  24. Ormerod AD, Campalani E, Goodfield MJ. British Association of Dermatologists guidelines on the efficacy and use of acitretin in dermatology. Br J Dermatol 2010; 162:952–63.

    Article  CAS  Google Scholar 

Download references

Author information

Authors and Affiliations

  1. National Skin Centre, Singapore, Singapore

    Melissa Mei Hsia Chan, Xiahong Zhao & Hong Liang Tey

  2. Duke-NUS Medical School, Singapore, Singapore

    Gim Hui Lim

  3. Yong Loo Lin School of Medicine, National University of Singapore, Singapore, Singapore

    Hong Liang Tey

  4. Lee Kong Chian School of Medicine, Nanyang Technological University, Singapore, Singapore

    Hong Liang Tey

Authors
  1. Melissa Mei Hsia Chan
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. Gim Hui Lim
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. Xiahong Zhao
    View author publications

    You can also search for this author in PubMed Google Scholar

  4. Hong Liang Tey
    View author publications

    You can also search for this author in PubMed Google Scholar

Corresponding author

Correspondence to Hong Liang Tey.

Additional information

Disclosure

Financial support: this study was supported by the Ministry of Health’s National Medical Research Council, Singapore, under the Clinician Scientist Award (NMRC/CSA-INV/0023/2017). Conflict of interests: none.

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Chan, M.M.H., Lim, G.H., Zhao, X. et al. Isolated hypohidrosis: pathogenesis and treatment. Eur J Dermatol 30, 680–687 (2020). https://doi.org/10.1684/ejd.2020.3931

Download citation

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1684/ejd.2020.3931

Key words

Search

Navigation