Post-Typhoid Anhidrosis: a Clinical Curiosity

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Post-typhoid anhidrosis 435 Postgrad Med J: first published as 10.1136/pgmj.71.837.435 on 1 July 1995. Downloaded from Post-typhoid anhidrosis: a clinical curiosity

V Raveenthiran

Summary family physician. Shortly after convalescence A 19-year-old girl developed generalised she felt vague discomfort and later recognised anhidrosis following typhoid fever. Elab- that she was not sweating as before. In the past orate investigations disclosed nothing seven years she never noticed sweating in any abnormal. A skin biopsy revealed the part ofher body. During the summer and after presence of atrophic as well as normal physical exercise she was disabled by an eccrine glands. This appears to be the episodic rise of body temperature (41.4°C was third case of its kind in the English recorded once). Such episodes were associated literature. It is postulated that typhoid with general malaise, headache, palpitations, fever might have damaged the efferent dyspnoea, chest pain, sore throat, dry mouth, pathway of sweating. muscular cramps, dizziness, syncope, inability to concentrate, and leucorrhoea. She attained Keywords: anhidrosis, hypohidrosis, sweat gland, menarche at the age of 12 and her menstrual typhoid fever cycles were normal. Hypothalamic functions such as hunger, thirst, emotions, libido, and sleep were normal. Two years before admission Anhidrosis is defined as the inability of the she had been investigated at another centre. A body to produce and/or deliver sweat to the skin biopsy performed there reported normal skin surface in the presence of an appropriate eccrine sweat glands. stimulus and environment' and has many forms An elaborate physical examination ofgeneral (box 1). The localised form, despite giving a physique, heart, lungs, thyroid, abdomen, vital diagnostic clue to the underlying disease genitalia, eyes, mouth, salivary and lacrimal (eg, leprosy, Homer's syndrome) does not glands, lymph nodes, ear, nose and throat cause bodily discomfort to the patient. The disclosed nothing abnormal. There were no generalised form upsets the thermoregulatory evidences of sensory, motor or autonomic mechanisms and causes thermal intolerance; it disorders of the nervous system. The skin was may result in heat stroke and even death in dry and scaly. Other ectodermal derivatives tropical climates.2 This report describes a such as teeth, hair, nail and breast were normal. http://pmj.bmj.com/ curious case ofgeneralised anhidrosis following A 10 x hand lens revealed normal sweat pores typhoid fever. The extreme rarity ofthis comp- in the palmar creases. A continuous body lication of typhoid prompted this report. temperature recording showed normal early morning temperature and a tendency for this to Case report rise towards evenings. Haematological and biochemical investiga-

A 19-year-old woman was investigated in May tions as well as radiographs of skull were on September 25, 2021 by guest. Protected copyright. 1992 for generalised absence of sweating. She normal. Elaborate screening tests for occult was the only female child of her consan- infections, tumours, metabolic and autoim- guineous parents and was fourth of seven mune diseases were negative. Starch-iodine siblings. None of her family members had any test,8 thermal sweating test and exercise test evidence of ectodermal dysplasias or neuro- demonstrated a total absence of sweating. Dur- endocrine diseases. She had a history of high ing the thermal sweating test she developed (39-40.5°C) intermittent febrile illness for diffuse flushing of the skin, tachycardia, dys- three weeks when she was 12 years old. It was pnoea and fine rales over both lung fields. A diagnosed and treated as typhoid fever by her pilocarpine test was suggested but not performed. A skin biopsy from the axilla revealed normal epidermis, dermis, hair follicles, sebaceous and apocrine glands. Most of the Department of eccrine glands were of normal morphology Medicine, Anhidrosis while a few were atrophic. There was no Stanley Medical immune cell infiltration around the atrophic College, * congenital or acquired glands (figure). Madras, India * primary or secondary She was diagnosed as suffering from V Raveenthiran * localised or generalised * partial (hypohidrosis) or complete acquired idiopathic generalised anhidrosis and Correspondence to (anhidrosis) she was reassured. Her symptoms have V Raveenthiran, 68 Seventh * permanent or temporary (eg, sweat gland remained unaltered during the subsequent Street, Sayee Nagar, Madras fatigue, miliaria) two-year follow-up. She accepts a sedentary 600 092, India life-style and has learnt to use cold water to Accepted 2 February 1995 Box 1 reduce body temperature. 436 Raveenthiran

Causes of physiological anhidrosis

* newborn period Postgrad Med J: first published as 10.1136/pgmj.71.837.435 on 1 July 1995. Downloaded from * dehydration due to vomiting, diarrhoea or diabetes * senile skin

Box 2

Causes of generalised anhidrosis * heat stroke * hysteria * anorexia nervosa Figure Skin biopsy showing an atrophic eccrine * poisons and drugs (eg, atropine, arsenic, gland. Haematoxylin-eosin stain. Magnification 100 x morphine) * post exfoliative dermatitis * miliaria Discussion * sweating fatigue (tropical anhidrosis) * sun burn Generalised anhidrosis without any organic * Sjogren's syndrome * diabetes mellitus lesion is extremely rare (see boxes 2-4). It has * orthostatic hypotension been variously named acquired generalised * occult malignancy especially lymphomas idiopathic anhidrosis, chronic idiopathic * autoimmune anhidrosis anhidrosis and pure progressive sudomotor * scleroderma failure.5 It is generally believed to be due to an * idiopathic interruption in the neural pathway ofsweating, although the exact cause and mechanism ofthis Box 3 neural interruption is largely unknown. A history of typhoid fever preceding the onset of anhidrosis in the above reported case may Causes of localised anhidrosis provide a clue to this mystery. On a survey ofthe literature, only two similar * myelitis * alcoholic or diabetic polyneuritis cases could be found in which anhidrosis was * oedema and urticaria reported to follow typhoid. In 1936 Fog * dermatitis (seborrheic, atopic, contact, reported generalised anhidrosis in a 25-year- exfoliative, etc) old man following an attack of paratyphoid * pressure effect fever.6 Fog assumed the lesion to be at the * neurodermatitis hypothalamic sweating centre and implicated * leprosy

* iatrogenic (eg, sympathectomy, skin grafts, http://pmj.bmj.com/ paratyphoid fever as the cause. Engelhardt and etc) Melvin reported the second case, a 49-year-old * radiodermatitis woman, in whom generalised anhidrosis fol- * antiperspirant chemicals and cosmetics lowed an attack of typhoid fever.' This case appears to be the third of its kind in English Box 4 literature. Sweating is controlled by the anterior nucleus of the hypothalamus. The efferent Experimental data suggest that nerve cells on September 25, 2021 by guest. Protected copyright. sudomotor pathway starting from the hypo- can tolerate a maximum of 42°C for 40 to 60 thalamus is relayed through the brainstem, minutes. Neural complications were reported intermediolateral horn of the spinal cord, sym- after whole body hyperthermia at a maximum pathetic ganglia and non-myelinated C fibres.8 ofbetween 40 and 43°C for six hours.'0 Hence it There are enough data to state that neural cells is probable that the prolonged high fever in the of the sweating pathway undergo destructive reported case might have damaged the neural changes at high body temperature. Shelley et al cells of the sweating pathway. The generalised cited 13 cases of heat stroke in whom a reduc- nature of the illness indicates that the lesion is tion in the cell count of the hypothalamus was unlikely to be at the level ofthe spinal cord or at demonstrated.' Delgado et al reported lesions the peripheral neurons. Selective neuronal in the intermediolateral horn of the spinal cord damage at the sweating centre of the hypo- in a case of fatal heat stroke.9 thalamus appears to be the correct explanation.

1 Shelley WB, Horvath PN, Pillsbury DM. Anhidrosis. 7 Engelhardt HT, Melvin JP. General acquired anhydrosis. Medicine 1950; 29: 195-224. Am J Med Sci 1945; 210: 323-8. 2 Dann EJ, Berkman N. Chronic idiopathic anhydrosis - a rare 8 Champion RH. Disorders of sweat gland. In: Champion cause of heat stroke. Postgrad Med J 1992; 68: 750-2. RH, Burton JL, Ebling FJG, eds. Rook's textbook of 3 Low PA, Fealey RD, Sheps SG, Su WP, Trautmann JC, dermatology 5th edn. London: Blackwell Scientific, 1992; Kuntz NL. Chronic idiopathic anhidrosis. Ann Neurol 1985; pp 1745-62. 18: 344-8. 9 Delgado G, Tunon T, Gallego J, Villanueva JA. Spinal cord 4 Murakami K, Sobue G, Terao S, Mitsuma T. Acquired lesions in heat stroke. J Neurol Neurosurg Psychiatry 1985; idiopathic generalized anhidrosis, a distinctive clinical syn- 48: 1065-7. drome. J Neurol 1988; 235: 428-31. 10 Sminia P, van der Zee J, Wondergem J, Haveman J. Effect of 5 Nakazato Y, Shimazu K, Tamura N, Hamaguchi K. hyperthermia on the central nervous system - a review. IntJ (Idiopathic pure sudomotor failure). Rinsho Shinkeigaku Hyperthermia 1994; 10: 1-30. 1994; 34: 12-5 (English abstract). 6 Fog M. General acquired anhidrosis. JAMA 1936; 107: 2040-5.