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PubMedDate not supplied

Symptomatic and asymptomatic hypohidrosis in children under topiramate treatment.

Topiramate (TPM) has peculiar side effects such as speech difficulties, weight loss, oligohidrosis and hyperthermia. We present the frequency and severity of hypohidrosis in our patients under TPM treatment. One hundred and two patients treated with TPM were evaluated retrospectively. Five (8 months-15 years of age) of them experienced symptomatic hypohidrosis manifested with prolonged or intermittent fever. Pilocarpine iontophoresis sweat test had been performed on the five patients before they were managed, and no sweat had been collected in 4/5 cases. Of the 102 patients, 42 who started TPM treatment when the study was established were evaluated prospectively regarding oligohidrosis. First, they were questioned about whether hypohidrosis occurred after TPM. Of 42, 11 patients complained of hypohidrosis. A sweat test was then performed on these 11 patients. Sweat could not be obtained in 5/11, and increased chloride concentration was found in 4/11. However, sweat could be obtained in the patients who had no complaint of hypohidrosis. Pediatricians should be aware of this side effect of TPM to prevent nonrelevant and cumbersome investigations in patients with prolonged or intermittent fever onset during TPM treatment. Our findings suggest that 5% of patients would experience hyperthermia during TPM treatment. Hypohidrosis without hyperthermia would be more frequent. If it is possible to collect sweat in patients who get fever during TPM treatment, it would be highly probable that the fever is not due to hypohidrosis. Increased chloride concentration alone does not seem to be significant for the hyperthermia risk.

Adolescent↗
PubMed2006

Successful steroid treatment of acquired idiopathic partial hypohidrosis.

The pathogenesis of idiopathic-acquired hypohidrosis remains unknown, and no specific causes have yet been established. We report a 34-year-old man with acquired idiopathic hypohidrosis successfully treated with prednisolone. The patient noticed heat intolerance and hypohidrosis of the pectoral and back during the summer. No systemic disease or neurological findings were identified. Eccrine sweat glands displayed infiltration by inflammatory cells, with immunoglobulin G and C3 deposition in the basement membrane. Steroid therapy improved the hypohidrosis. An immunological pathogenesis could be a major factor in idiopathic-acquired hypohidrosis.

Adult↗
PubMed2004

Monitoring perspiration from palms of hypohidrosis patients with a stopped-flow conductometric mini-system.

BACKGROUND: Sensitive and real-time methods are required to characterize the symptoms and resolve the complicated pathology of hypohidrosis. METHOD: We constructed a conductometric humidity sensor and a stopped-flow manifold with suitable dynamic range for monitoring impaired perspiration. The mini-sensor was coated with a water-absorbing polymer of poly-(2-acrylamido-2-methylpropane sulfonate). RESULTS: Perspiration from palms of normal individuals and hypohidrosis patients were monitored and compared. Tangent slopes of the sensorograms were capable of discriminating hypohidrosis patients from normal individuals. CONCLUSION: The conductometric min-sensor and the stopped-flow manifold were proven to be useful as a diagnostic tool for hypohidrosis.

Electric Conductivity↗
PubMed2002

Cholinergic urticaria, a new pathogenic concept: hypohidrosis due to interference with the delivery of sweat to the skin surface.

BACKGROUND: Acetylcholine has been suspected to be a pathogenetic factor for cholinergic urticaria (CU), without definite evidence. In contrast, there are scattered reports of CU associated with acquired generalized hypohidrosis. We have recently examined 2 patients with CU in both of whom we noticed the presence of extensive hypohidrosis that occurred only in winter. OBJECTIVE: In these 2 patients, acquired hypohidrosis due to superficial obstruction of the acrosyringium was suspected as the cause. Both case 1, a 22-year-old Japanese man, and case 2, a 21-year-old Japanese man, began to have anhidrosis and numerous red macules on their body whenever they felt hot in winter. These symptoms ceased to appear in summer. METHODS: We studied histologically their lesional skin in addition to provocation tests for CU. RESULTS: The diagnosis of CU was confirmed by the provocation of typical wheals after physical exercise in both cases. Histological study revealed findings suggestive of the presence of occlusion of the superficial acrosyringium. CONCLUSION: We think that such a hypohidrosis due to occlusion of superficial sweat ducts may also play a role in many other patients with CU of unknown etiology that becomes exacerbated in winter when sweating is not a frequent event.

Acetylcholine↗
PubMed2006

Hypohidrosis during topiramate treatment: a rare and reversible side effect.

Topiramate is an antiepileptic drug with a beneficial clinical effect on various seizure types. Topiramate does not seem to be associated with serious adverse effects and is also well tolerated in pediatric patients. Only few cases of hypohidrosis have been described. This report presents one young patient with complex partial seizures who was medicated with topiramate when she developed fatigue, headache, intermittent hyperthermia, inability to produce sweat secretion, and dryness of the skin. Reduced sweat response was determined using the Wescor Macroduct collection procedure. Topiramate was discontinued, and within 3 weeks a repeat sweat test was completely normal. At that time, clinical signs had also disappeared. Hypohidrosis is an uncommon and reversible side effect reported in association with topiramate therapy. It is rare in patients on monotherapy. Although a definite causal relationship still needs to be established, this side effect might be attributed to an autonomic dysfunction by inhibition of isoenzymes of carbonic anhydrase localized in human eccrine sweat glands.

Anticonvulsants↗
PubMed1998

The ultrastructural characteristics of eccrine sweat glands in a Fabry disease patient with hypohidrosis.

As an investigation of the pathogenetic mechanism of diminished sweating in Fabry disease, an electron microscopy ultrastructural study was conducted on specimens of eccrine sweat glands from a typical patient with Fabry disease who had hypohidrosis, a low skin moisture content, and diminished thermoregulation ability. Numerous characteristic cytoplasmic inclusions were observed in the eccrine sweat glands, the lamellar pattern of which was considerably variable in various types of gland cells. Large vacuolar inclusions predominated in clear cells of secretory coil; lesser vacuoles were also seen in the coiled duct, and the basal cells of the straight duct toward the coiled duct displayed mulberry-like figures. There were some clear cells showing cell damage and necrosis in the secretory coil. Lamellated inclusions were noted in the unmyelinated axons innervating the eccrine sweat glands. The small blood vessels around the eccrine glands were narrowed by swollen endothelial cells with heavy inclusions. These intracytoplasmic deposits may be responsible for the decreased sweating ability in Fabry disease. The factors related to hypohidrosis are also discussed.

Eccrine Glands↗
PubMed2003

Transient hypohidrosis induced by topiramate.

PURPOSE: Hypohidrosis during topiramate (TPM) treatment was recently reported in children. We describe an adult epilepsy patient who developed inability to sweat as well as heat intolerance while undergoing treatment with TPM. METHODS: To detect the site of the sweat block, patient underwent examination of sweat gland function, cardiovascular autonomic test, and body temperature rhythm determination. RESULTS: During TPM treatment, cardiovascular autonomic function and circadian rhythm of body core temperature were normal, whereas thermoregulatory sweat test (TST) showed anhydrosis. This adverse drug effect was quickly resolved after drug discontinuation. CONCLUSIONS: Because of normal cardiovascular autonomic function and central and peripheral thermoregulatory mechanisms, we hypothesize that hypohidrosis during TPM treatment could be due to a carbonic anhydrases (CA) block at the level of sweat gland.

Adult↗
PubMed2000

Familial mydriasis, cardiac arrhythmia, respiratory failure, muscular weakness and hypohidrosis.

OBJECTIVES: To describe a family with some sort of progressive autonomic failure in one generation (2 affected of a sibship of 7 sisters). The main features were: mydriasis, cardiac arrhythmia, cardiomegaly, hypohidrosis, respiratory failure, and muscular weakness. METHODS: Pupillometry, evaporimetry, and isokinetic power measurements were carried out. RESULTS: The autonomic dysfunction pattern (mainly cardiac abnormalities, mydriasis) seems to differ somewhat from that of progressive autonomic failure (Shy-Drager syndrome). "Lewy body-like" inclusions were present, in particular in substantia nigra, but also in locus ceruleus and raphe nuclei (cell loss only in locus ceruleus). There were no oligodendroglial, cytoplasmatic inclusions, apparently a marker in multiple system atrophy. Proper Lewy bodies were also present. Differences seemed to prevail vs the Shy-Drager syndrome. Various traits: muscular weakness pattern (e.g. preferential peroneal distribution), minor elbow contractures, and arrhythmia were reminiscent of Emery-Dreifuss muscle dystrophy (E-D). Distinguishing features included: hereditary pattern, mydriasis, and hypohidrosis. CONCLUSION: Conceivably, this disorder is close to, but still not identical with E-D.

Adult↗
PubMed1993

Ipsilateral hypohidrosis in brain stem infarction.

BACKGROUND AND PURPOSE: The brain stem is the most important autonomic processing center, but very little attention has been given to clinical manifestations of autonomic failure in brain stem stroke. Our purpose was to evaluate the prevalence, characteristics, and prognostic significance of sweating dysfunction in brain stem infarctions. METHODS: We carried out a prospective study using quantitative evaporimetry to investigate spontaneous and heat-stimulated sweating in 18 healthy control subjects and 18 patients with ischemic brain stem stroke in the acute phase and at 1 and 6 months after infarction. RESULTS: The sweating response induced by a heating stimulus was significantly lower on the ipsilateral side to the infarction than on the contralateral side. Constant ipsilateral hypohidrosis was established in 83% of the patients in the acute phase, in 100% at 1 month, and in 76% at 6 months after infarction. No differences of sweating response were found between medullary and pontine infarcts. CONCLUSIONS: Hypohidrosis throughout the whole ipsilateral side of the body, a long-lasting phenomenon that has not previously been described, is an essential feature of autonomic failure in brain stem infarction.

Adult↗
PubMed1998

Congenital ichthyosis, follicular atrophoderma, hypotrichosis, and hypohidrosis: a new genodermatosis?

Follicular atrophoderma is a rare anomaly observed mainly in the X-dominant form of chondrodysplasia punctata (Conradi-Hünermann-Happle syndrome) and in the X-linked dominant Bazex syndrome. We report on five Emirati sibs (three girls and two boys), 4-18 years old, with normal stature, diffuse congenital ichthyosis, patchy follicular atrophoderma, generalized and diffuse non-scarring hypotrichosis, and marked hypohidrosis. Steroid sulfatase activity, assessed in the two boys, was found to be normal. Electron microscopic studies of ichthyotic skin did not show any specific abnormality. The association of congenital diffuse ichthyosis with follicular atrophoderma and hypotrichosis has not been reported before. The patients were reminiscent of Bazex syndrome; however, ichthyosis is not a component of Bazex syndrome. We conclude that this syndrome of congenital ichthyosis with follicular atrophoderma represents a new autosomal recessive genodermatosis.

Abnormalities, Multiple↗
PubMed1988

Familial simple hypohidrosis with abnormal palmar dermal ridges.

A brother and sister born to nonconsanguineous, Iranian Jewish parents were found to have simple hypohidrosis. Pilocarpine produced only little sweating, and external heat and physical effort were associated with elevation of body temperature. Abnormal palmar dermal ridges were considered to be associated with paucity of sweat pores and glands. Biopsy supported this view since only one normal sweat gland and duct was found in a 5 mm punch.

Adolescent↗
PubMed1987

Anhidrosis (hypohidrosis) in Sjögren's syndrome.

There has been a relative lack of literature on the association of hypohidrosis in Sjögren's syndrome with any lesion having specific histologic findings. We looked at a recent case presentation of a 55-year-old man with complaints of dry mouth and dry eyes, becoming easily overheated in direct sunlight, and having difficulty in perspiring. Physical examination showed fissuring and atrophy of the tongue and angular cheilitis. A punch biopsy of the skin showed a moderate number of eccrine gland and ductal structures in the lower reticular dermis, each surrounded by a dense cellular infiltrate of plasma and lymphocytic cells. Our patient also had a markedly decreased sweating response to methacholine. In reviewing the literature as far back as 1951 and on the basis of findings in our present case study, we conclude that it seems probable that the severity of the skin disease is an important determining factor in predicting whether the sweat gland lesion does exist.

Humans↗
PubMed2000

Sporadic Bazex-Dupré-Christol-like syndrome: early onset basal cell carcinoma, hypohidrosis, hypotrichosis, and prominent milia.

BACKGROUND: We present the case of a 32-year-old woman with a large recurrent multifocal basal cell carcinoma on the scalp. Conspicuous accompanying symptoms were multiple periorbital milia, hypotrichosis of the body and the scalp, and hypohidrosis. The sparse hair of the scalp showed further abnormalities such as pili torti, as well as flattened, irregularly curly hairs. OBJECTIVE: In 1964, Bazex et al. described a syndrome characterized by congenital hypotrichosis, follicular atrophoderma, and basocellular neoplasms that included basal cell nevi and early onset basal cell carcinomas. The Bazex-Dupré-Christol syndrome is a rare X-linked dominant disease. A sporadic occurrence with the typical constellation of these symptoms has not yet been reported. The lack of a positive family history and no signs of follicular atrophoderma argues for a sporadic occurrence of a Bazex-Dupré-Christol-like syndrome. The case reported shares several features with the classic Bazex-Dupré-Christol syndrome. CONCLUSION: Our report documents the necessity to look for early development of basal cell carcinomas in patients who show signs of the epidermal malformations described.

Acneiform Eruptions↗
PubMed2001

Hypohidrosis related to the administration of topiramate to children.

PURPOSE: Topiramate (TPM) is an antiepileptic agent, first licensed in the United Kingdom in 1994, that is used in the treatment of patients with refractory seizure disorders. TPM is a monosaccharide d-fructose derivate, with sulfamate function, and so far, few adverse side effects have been reported. METHODS: We describe three patients with epilepsy who were treated with TPM and developed hypohidrosis, heat and exercise intolerance, as well as fever. The sudomotor function was assessed after peripheral stimulation with pilocarpine iontophoresis. RESULTS: Sweat response was reduced in all three patients. Signs and symptoms ceased after drug suppression. CONCLUSIONS: This side effect associated with TPM, which has not been described previously, can be clinically significant during heat stress and exercise challenge.

Adolescent↗
PubMed1977

An ectodermal dysplasia syndrome of alopecia, onychodysplasia, hypohidrosis, hyperkeratosis, deafness and other manifestations.

A girl is reported with a hitherto apparently undescribed ectodermal dysplasia syndrome. The main findings include: alopecia, onychodysplasia, hypohidrosis, sensorineural deafness, skin with a tan color and hyperkeratosis (involving also plams and soles), unusual facies (with slight auricle and nose abnormalities), pectus excavatum, severe hyperopia, EEG abnormalities, and retarded bone age. The patient also presents mongoloid palpebral slanting, narrow palpebral fissures, bilateral esotropia, photophobia and dermatoglyphics with extensive ridge dissociation. The etiology is unknown but presumed to be genetic, possibly due to the homozygous state of an autosomal recessive mutation.

Alopecia↗
PubMed1999

Hypohidrosis in children.

Hypohidrosis has many causes. The condition may be generalised or segmental, acute or progressive and central or peripheral in origin. Most of the causes can be diagnosed from the history and physical examination. Laboratory investigations are usually not necessary. Treatment should be directed at the underlying cause whenever possible.

Child↗
PubMed1976

Hyperhidrosis and hypohidrosis.

Sweating has an important physiological function concerned with both temperature and fluid balance. Hypohidrosis is uncommon and rarely recognized by the patient. It usually represents damage to the central nervous system or peripheral nerves. Hyperhidrosis may be a feature of general medical diseases such as thyrotoxicosis and fevers. It may also be due to damage of the central of peripheral sympathetic nerves. Localized hyperhidrosis tends to occur on the palms, the soles, the axillae and, to a lesser extent, the face. Treatment of local hyperhidrosis with topical aluminium salts, or with topical or oral anticholinergics, may help some patients but surgery may be indicated in those with severe hyperhidrosis.

Age Factors↗