Open Access

Compensatory quadrant-hyperhidrosis after contralateral intrathoracic surgery: a case report

  • Stefan Brodoehl1Email author,
  • Otto Wilhelm Witte1 and
  • Albrecht Guenther1
Journal of Medical Case Reports20137:24

DOI: 10.1186/1752-1947-7-24

Received: 24 August 2012

Accepted: 4 December 2012

Published: 18 January 2013

Abstract

Introduction

Unilateral hyperhidrosis can be a neurological manifestation of irritations of the central or peripheral nervous system.

Case presentation

We present the case of a 67-year-old German man who had hyperhidrosis of his right upper body quadrant (including face, arm, and chest) following intrathoracic surgery of a left-sided pleural lipoma.

Conclusion

An isolated unilateral hyperhidrosis might occur after intrathoracic surgery. Besides anticholinergic drugs the use of botulinum toxin should be considered.

Keywords

Botulinum toxin Intrathoracic surgery Unilateral hyperhidrosis

Introduction

Anhidrosis and hyperhidrosis can be neurological manifestations of irritations of the central or peripheral nervous system [1, 2]. There are described causes such as injuries, tumors, infarcts or hemorrhages of the brain or the medulla.

Case presentation

A 67-year-old German man admitted to our botulinum toxin clinic complained about extensive sweating of his right upper body including his right facial area, right arm, and chest, whereas the left side of his body appeared to be ‘summer-dry’. At the time of his presentation at our clinic the patient reported a subjective reduction of quality of life. The massive hyperhidrosis made him feel uncomfortable in the company of strangers and therefore kept him from taking part in social activities.

Symptoms started some weeks after tumor extirpation of a left-sided pleural lipoma via an anterior-posterior thoracotomy at the 5th intercostal space in conjunction with a partial pleurectomy. After being released from hospital, he experienced contralateral hyperhidrosis which started in the face spreading downwards.

Clinical neurological examination together with ophthalmological tests yielded a normal status appropriate to the patient’s age. Sympathetic skin nerve action potentials [3] were recorded in both arms and legs and revealed a significantly decreased sympathetic activity in the left arm. Pharmacological pupil function test (cocaine and phenylephrine eye drops) showed no discrepancy on the left side. In particular, Horner’s syndrome or hypesthesia were not found. In addition, scintigraphy showed no proof of a reduced function of salivary glands. After physical activity, a starch-iodine preparation was performed, revealing significant hyperhidrosis of the right upper body together with a relevant temperature difference measured on the chest (right 30.5°C, left 33.7°C). Lung auscultation, before and after physical exertion, yielded right-sided bronchial spasm post-exercise.

Treatment was started with bornaprine: a central anticholinergic drug. A starting dose of two mg per day increasing to six mg did not lead to a relevant clinical improvement but produced intolerable side effects.

Due to a considerable subjective impairment, we decided on a test injection of botulinum toxin type A. A total of 20 units at three injection points on the right side of the patient’s forehead and a total of 40 units at six injection points on his right upper body were administered. Some improvement was achieved and therapy was repeated in a three-month interval.

Discussion

The localized unilateral hyperhidrosis in the present case developed after intrathoracic surgery. Therefore, we hypothesize that the quadrant-hyperhidrosis of the right upper body is a (hyper-) compensation of a disturbed sympathetic innervation of the contralateral left side due to a surgical lesion of the cervical sympathetic chain. The lesion is assumed to be situated in the endothoracic fascia, where the cervical sympathetic fibers are closely related to the apical pleura [4]. Presumably caused by a disturbed negative feedback of afferent sympathetic signals, the sweating center in the hypothalamus generates an increased positive feedback signal that induces severe sweating on the body side contralateral to the sympathetic lesion [5].

Table 1 provides an overview of known cases of localized unilateral hyperhidrosis in the literature and (if included) briefly describes applied therapies and their effectiveness. Taken together most successful therapy strategies included the application of botulinum toxin type A. Besides sympathectomy treatment with antidepressants or local therapy with aluminum hexachloride have proven effective. Although not effective in the present case, the use of oral anticholinergic drugs is a good therapeutic alternative especially because Wolosker et al. [6] have recently shown the potential of oxybutynin in treating localized hyperhidrosis.
Table 1

Cases of localized unilateral hyperhidrosis in the literature excluding isolated facial hyperhidrosis

Cause of hyperhidrosis

Reference

Cases

Therapy

Idiopathic unilateral hyperhidrosis

 

Cunliffe et al. [7]

3

local anticholinergic*

 

van de Kerkhof et al. [8]

1

no

 

Dworin and Sober [9]

1

local Al2Cl6 *

 

Fernández and Armijo [10]

1

local Al2Cl6 *

 

Querol Nasarre et al. [11]

1

no

 

Köse and Baloglu [12]

1

no

 

Boyvat et al. [13]

1

antidepressant

 

Kreyden et al. [14]

1

botulinum toxin A *

 

Kocyigit et al. [15]

1

no

Intrathoracic neoplasia

 

Middleton [16]

1

no

 

Walsh et al. [17]

2

no

 

McCoy [18]

1

no

 

Wang et al. [19]

2

no

 

McEvoy et al. [20]

1

no

 

Lindsay et al. [4]

2

no

 

Umeki et al. [21]

1

no

 

Lambert et al. [22]

1

local radiotherapy *

 

Yamauchi et al. [23]

1

no

 

Slabbynck et al. [24]

1

no

 

Lee and Greenstone [25]

1

no

 

Nishimura et al. [26]

1

local radiotherapy *

 

Sribnick and Boulis [27]

1

sympathectomy *

Strokebrainstem infarction

 

Mon and Mizotani [28]

1

no

 

Korpelainen et al. [29]

16

no

 

Rey et al. [30]

1

no

 

Rousseaux et al. [31]

5

no

 

Sato and Nitta [32]

1

no

 

Pellecchia et al. [33]

1

no

Strokecerebral infarction

 

Labar et al. [34]

2

no

 

Sakashita et al. [35]

1

no

 

Kim et al. [36]

5

no

 

Bassetti and Staikov [37]

1

no

 

Smith [38]

1

no

 

Faruqi et al. [39]

1

no

Postinterventional complications

 

Belin and Polo [40]

1

botulinum toxin A *

 

Aşik et al. [41]

1

local radiotherapy *

Spinal cord pathology

 

Pool [42]

1

hemilaminectomy *

 

Baskan et al. [43]

1

botulinum toxin A *

 

Kilinçer et al. [44]

1

no

 

Gorman [45]

1

improved bedding *

Sweat glands pathology

 

Kopera and Soyer [46]

1

appetite depressant *

 

Ruiz de Erenchun et al. [47]

1

antidepressant *

 

Parslew and Lewis-Jones [48]

1

no

Others (Buerger's disease)

   
 

Baker [49]

1

no

(*) indicates successful therapy, (Al2Cl6) aluminum hexachloride.

Another relevant clinical aspect might be a bronchial spasm of the right lung in the course of physical exertion due to an overwhelming activity of the left-sided parasympathetic nervous system. In our case, the patient had no medical history of chronic lung disease; although an obstructive lung disease cannot be excluded beyond doubt. This additional feature might be an interesting physiological aspect that has – to the best of our knowledge – never been reported before.

Conclusion

An isolated unilateral hyperhidrosis is a rare complication after intrathoracic surgery that potentially reduces the quality of life. As therapeutic options anticholinergic drugs or the use of botulinum toxin could be considered.

Consent

Written informed consent was obtained from the patient for publication of this manuscript and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.

Declarations

Acknowledgments

We thank our patient who gave written permission to publish his case. No external funding was obtained.

Authors’ Affiliations

(1)
Hans Berger Department of Neurology, Jena University Hospital

References

  1. Leung AK, Chan PY, Choi MC: Hyperhidrosis. Int J Dermatol. 1999, 38: 561-567. 10.1046/j.1365-4362.1999.00609.x.View ArticlePubMed
  2. Wasner G, Maag R, Ludwig J, Binder A, Schattschneider J, Stingele R, Baron R: Harlequin syndrome–one face of many etiologies. Nat Clin Pract Neurol. 2005, 1: 54-59. 10.1038/ncpneuro0040.View ArticlePubMed
  3. Lidberg L, Wallin BG: Sympathetic skin nerve discharges in relation to amplitude of skin resistance responses. Psychophysiology. 1981, 18: 268-270. 10.1111/j.1469-8986.1981.tb03033.x.View ArticlePubMed
  4. Lindsay DC, Freeman JG, Record CO: Unilateral hyperhidrosis associated with underlying intrathoracic neoplasia. Thorax. 1986, 41: 814-815. 10.1136/thx.41.10.814.PubMed CentralView ArticlePubMed
  5. Chou SH, Kao EL, Lin CC, Chang YT, Huang MF: The importance of classification in sympathetic surgery and a proposed mechanism for compensatory hyperhidrosis: experience with 464 cases. Surg Endosc. 2006, 20: 1749-1753. 10.1007/s00464-005-0829-7.View ArticlePubMed
  6. Wolosker N, Campos JR, Kauffman P, Munia MA, Neves S, Jatene FB, Puech-Leao P: The use of oxybutynin for treating facial hyperhidrosis. An Bras Dermatol. 2011, 86: 451-456. 10.1590/S0365-05962011000300005.View ArticlePubMed
  7. Cunliffe WJ, Johnson CE, Williamson DM: Localized unilateral hyperhidrosis–a clinical and laboratory study. Br J Dermatol. 1972, 86: 374-378. 10.1111/j.1365-2133.1972.tb05050.x.View ArticlePubMed
  8. van de Kerkhof PC, den Arend JA, Bousema MT, Stolz E: Localized unilateral hyperhidrosis. Br J Dermatol. 1987, 117: 779-782. 10.1111/j.1365-2133.1987.tb07360.x.View ArticlePubMed
  9. Dworin A, Sober AJ: Unilateral segmental hyperhidrosis. Response to 20% aluminum chloride solution and plastic wrap. Arch Dermatol. 1978, 114: 770-771. 10.1001/archderm.1978.01640170070015.View ArticlePubMed
  10. Fernández G, Armijo M: Unilateral facial circumscribed hyperhidrosis. Acta Derm Venereol. 1985, 65: 445-447.PubMed
  11. Querol Nasarre I, Jarauta Salvador F, Alberdi Ibáñez I, Cisneros Lanuza T, Expósito Rando A: Paroxysmal unilateral hyperhidrosis. Neurologia. 1994, 9: 419-422.PubMed
  12. Köse O, Baloglu H: Idiopathic unilateral circumscribed hyperhidrosis. Int J Dermatol. 1997, 36: 209-210.View ArticlePubMed
  13. Boyvat A, Pişkin G, Erdi H: Idiopathic unilateral localized hyperhidrosis. Acta Derm Venereol. 1999, 79: 404-405. 10.1080/000155599750010508.View ArticlePubMed
  14. Kreyden OP, Schmid-Grendelmeier P, Burg G: Idiopathic localized unilateral hyperhidrosis: case report of successful treatment with botulinum toxin type A and review of the literature. Arch Dermatol. 2001, 137: 1622-1625.View ArticlePubMed
  15. Kocyigit P, Akay BN, Saral S, Akbostanci C, Bostanci S: Unilateral hyperhidrosis with accompanying contralateral anhidrosis. Clin Exp Dermatol. 2009, 34: e544-e546. 10.1111/j.1365-2230.2008.03070.x.View ArticlePubMed
  16. Middleton WG: Bronchial carcinoma with pleural spread causing unilateral thoracic hyperhidrosis. Br Med J. 1976, 2: 563-PubMed CentralView ArticlePubMed
  17. Walsh JC, Low PA, Allsop JL: Localized sympathetic overactivity: an uncommon complication of lung cancer. J Neurol Neurosurg Psychiatry. 1976, 39: 93-95. 10.1136/jnnp.39.1.93.PubMed CentralView ArticlePubMed
  18. McCoy BP: Apical pulmonary adenocarcinoma with contralateral hyperhidrosis. Arch Dermatol. 1981, 117: 659-661. 10.1001/archderm.1981.01650100061031.View ArticlePubMed
  19. Wang YT, Singh D, Poh SC: Hemihyperhidrosis and intrathoracic malignancy. Singapore Med J. 1981, 22: 86-88.PubMed
  20. McEvoy M, Ryan E, Neale G, Prichard J: Unilateral hyperhydrosis–an unusual presentation of bronchial carcinoma. Ir J Med Sci. 1982, 151: 51-52. 10.1007/BF02940144.View ArticlePubMed
  21. Umeki S, Tamai H, Yagi S, Soejima R, Higashi Y: Harlequin syndrome (unilateral flushing and sweating attack) due to a spinal invasion of the left apical lung cancer. Rinsho Shinkeigaku. 1990, 30: 94-99.PubMed
  22. Lambert M, Kanyinda JM, Richard F, Sindic C: Unilateral hyperhidrosis associated with intrathoracic IgD lambda myelomatous tumour. Clin Oncol (R Coll Radiol). 1993, 5: 65-66. 10.1016/S0936-6555(05)80705-8.View Article
  23. Yamauchi Y, Kobayashi T, Nagaro T, Yamamoto H, Kimura S, Arai T: A case of hemifacial hyperhidrosis on the opposite side of the pancoast tumor. Masui. 1994, 43: 924-926.PubMed
  24. Slabbynck H, Bedert L, De Deyn PP, Galdermans D, Coolen D: Unilateral segmental hyperhidrosis associated with pulmonary adenocarcinoma. Chest. 1998, 114: 1215-1217. 10.1378/chest.114.4.1215.View ArticlePubMed
  25. Lee WY, Greenstone M: Unilateral hyperhidrosis in malignant mesothelioma. Am J Med Sci. 2011, 342: 332-10.1097/MAJ.0b013e31821d6d1a.View ArticlePubMed
  26. Nishimura J, Tamada Y, Iwase S, Kubo A, Watanabe D, Matsumoto Y: A case of lung cancer with unilateral anhidrosis and contralateral hyperhidrosis as the first clinical manifestation. J Am Acad Dermatol. 2011, 65: 438-440. 10.1016/j.jaad.2010.01.043.View ArticlePubMed
  27. Sribnick EA, Boulis NM: Treatment of Harlequin syndrome by costotransversectomy and sympathectomy: case report. Neurosurgery. 2011, 69: E257-E259. 10.1227/NEU.0b013e3182186829.View ArticlePubMed
  28. Mon Y, Mizotani M: A case of hemi-hyperhidrosis and non-paralytic pontine exotropia due to brainstem infarction. Rinsho Shinkeigaku. 1992, 32: 718-721.PubMed
  29. Korpelainen JT, Sotaniemi KA, Myllylä VV: Ipsilateral hypohidrosis in brain stem infarction. Stroke. 1993, 24: 100-104. 10.1161/01.STR.24.1.100.View ArticlePubMed
  30. Rey A, Martí-Vilalta JL, Abellán MT: Contralateral hyperhidrosis secondary to the pontine infarct. Rev Neurol. 1996, 24: 459-460.PubMed
  31. Rousseaux M, Hurtevent JF, Benaim C, Cassim F: Late contralateral hyperhidrosis in lateral medullary infarcts. Stroke. 1996, 27: 991-995. 10.1161/01.STR.27.5.991.View ArticlePubMed
  32. Sato K, Nitta E: Pontine hemorrhage presenting with Foville syndrome and transient contralateral hyperhidrosis. Rinsho Shinkeigaku. 2000, 40: 271-273.PubMed
  33. Pellecchia MT, Criscuolo C, De Joanna G, D'Amico A, Santoro L, Barone P: Pure unilateral hyperhidrosis after pontine infarct. Neurology. 2003, 61: 1305-10.1212/WNL.61.9.1305.View ArticlePubMed
  34. Labar DR, Mohr JP, Nichols FT, Tatemichi TK: Unilateral hyperhidrosis after cerebral infarction. Neurology. 1988, 38: 1679-1682. 10.1212/WNL.38.11.1679.View ArticlePubMed
  35. Sakashita Y, Kakuta K, Kakuma K, Matsuda H: Unilateral persistent hyperhidrosis after ischemic stroke. Rinsho Shinkeigaku. 1992, 32: 454-456.PubMed
  36. Kim BS, Kim YI, Lee KS: Contralateral hyperhidrosis after cerebral infarction. Clinicoanatomic correlations in five cases. Stroke. 1995, 26: 896-899. 10.1161/01.STR.26.5.896.View ArticlePubMed
  37. Bassetti C, Staikov IN: Hemiplegia vegetativa alterna (ipsilateral Horner's syndrome and contralateral hemihyperhidrosis) following proximal posterior cerebral artery occlusion. Stroke. 1995, 26: 702-704. 10.1161/01.STR.26.4.702.View ArticlePubMed
  38. Smith CD: A hypothalamic stroke producing recurrent hemihyperhidrosis. Neurology. 2001, 56: 1394-1396. 10.1212/WNL.56.10.1394.View ArticlePubMed
  39. Faruqi S, Redmond G, Ram P, Owens VB, Sangster G, Barrett JA: Hemihyperhidrosis in cerebral infarction. Age Ageing. 2004, 33: 514-515. 10.1093/ageing/afh179.View ArticlePubMed
  40. Belin EE, Polo J: Treatment of compensatory hyperhidrosis with botulinum toxin type A. Cutis. 2003, 71: 68-70.PubMed
  41. Aşik ZS, Orbey BC, Aşik I: Sympathetic radiofrequency neurolysis for unilateral lumbar hyperhidrosis: a case report. Agri. 2008, 20: 37-39.PubMed
  42. Pool JL: Unilateral thoracic hyperhidrosis caused by osteoma of the tenth dorsal vertebra; case report. J Neurosurg. 1956, 13: 111-115. 10.3171/jns.1956.13.1.0111.View ArticlePubMed
  43. Baskan EB, Karli N, Baykara M, Cikman S, Tunali S: Localized unilateral hyperhidrosis and neurofibromatosis type 1: case report of a new association. Dermatology. 2005, 211: 286-289. 10.1159/000087026.View ArticlePubMed
  44. Kilinçer C, Oztürk L, Hamamcioglu MK, Altunrende E, Cobanoglu S: An upper thoracic spinal cord tumor presenting as hemifacial hyperhidrosis. Surg Neurol. 2007, 68: 461-463. 10.1016/j.surneu.2006.10.067. discussion 463View ArticlePubMed
  45. Gorman PH: Unilateral hyperhidrosis from a contralateral source in an individual with C4 complete tetraplegia. J Spinal Cord Med. 2010, 33: 428-430.PubMed CentralPubMed
  46. Kopera D, Soyer HP: Eccrine hamartoma of the sweat glands simulating localized unilateral hyperhidrosis. Hautarzt. 1992, 43: 587-589.PubMed
  47. Ruiz de Erenchun F, Vázquez-Doval FJ, Contreras Mejuto F, Quintanilla E: Localized unilateral hyperhidrosis: eccrine nevus. J Am Acad Dermatol. 1992, 27: 115-116. 10.1016/S0190-9622(08)80822-5.View ArticlePubMed
  48. Parslew R, Lewis-Jones MS: Localized unilateral hyperhidrosis secondary to an eccrine naevus. Clin Exp Dermatol. 1997, 22: 246-247. 10.1111/j.1365-2230.1997.tb01080.x.View ArticlePubMed
  49. Baker H: Unilateral hyperhidrosis. Br J Dermatol. 1988, 118: 588-589. 10.1111/j.1365-2133.1988.tb02480.x.View ArticlePubMed

Copyright

© Brodoehl et al.; licensee BioMed Central Ltd. 2013

This article is published under license to BioMed Central Ltd. This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://​creativecommons.​org/​licenses/​by/​2.​0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

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