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PubMed2006

Botulinum toxin: clinical use.

Since its development for the use of blepharospasm and strabismus more than 2.5 decades ago, botulinum neurotoxin (BoNT) has become a versatile drug in various fields of medicine. It is the standard of care in different disorders such as cervical dystonia, hemifacial spasm, focal spasticity, hyperhidrosis, ophthalmological and otolaryngeal disorders. It has also found widespread use in cosmetic applications. Many other indications are currently under investigation, including gastroenterologic and urologic indications, analgesic management and migraine. This paper is an extensive review of the spectrum of BoNT clinical applications.

Animals↗

PubMed2005

[Spontaneous hypothermia: a series of ten cases, place of Shapiro's syndrome].

SUBJECT: Hypothermia (defined as a core temperature lower than 35 degrees C) may result from accidental causes (exposure to cold, drug intoxications), from endocrine disorders (hypothyroidism), or from central or peripheral neurological disease. Among the causes of spontaneous hypothermia, the place of spontaneous periodic hypothermia or Shapiro's syndrome, of which less than 50 cases in children or adults have been reported, remains unclear. METHODS: Case series of spontaneous hypothermia in adults, from a register of the French Society of Internal Medicine (SNFMI). RESULTS: The ten collected cases of spontaneous hypothermia are heterogeneous. In half of the cases, an often ill-labeled psychiatric illness and/or epilepsy and/or anti-psychotic medication were found contributive. Only 5 cases at best seem to conform to the pattern of spontaneous periodic hypothermia or Shapiro's syndrome (in which agenesis of corpus callosum is typically found). In such cases, the episodes of hypothermia start with profuse hyperhidrosis suggesting a paroxystic reset of the hypothalamic thermostat with a lower temperature set point. In none of the cases was found a significant encephalic lesion. None of the treatment trials with anti-epileptics or cyproheptadine were found useful. Spontaneous hypothermia, whether periodic or not, seems to have an unpredictable course, with long periods of remission, and a benign long-term outcome. CONCLUSIONS: Spontaneous hypothermia is a symptom of likely multifactorial etiology. Even in cases conforming to the definition of Shapiro's syndrome, central nervous system anomalies are not unequivocal. No specific treatment for spontaneous hypothermia, whether periodic or not, can be recommended in the current state of knowledge.

Adult↗

PubMed2026

Sleep disturbances in children and adolescents with iron deficiency: Questionnaire-based and actigraphic findings.

OBJECTIVE: Iron deficiency (ID), without or with anemia (IDA), has been linked to restless sleep and sleep-related movement disorders in childhood, but objective correlates are incompletely defined. We aimed to describe caregiver-reported sleep disturbances, restless legs syndrome (RLS) - related symptoms, and actigraphic sleep patterns in children and adolescents with ID/IDA, and to compare these findings with available sleep-asymptomatic control datasets. We finally explored short-term changes after iron supplementation. METHODS: In this single-center pilot observational study, 31 children with ID/IDA underwent baseline clinical/laboratory assessment and caregiver-reported sleep evaluation with the Sleep Disturbance Scale for Children (SDSC) plus RLS-oriented items. Sixteen also completed home actigraphy, and 8 had follow-up after iron treatment prescribed in routine care. Baseline findings were compared with historical healthy control datasets without reported sleep disturbances; iron-status data were not available for the questionnaire control group; within-subject changes were explored in the follow-up subgroup. RESULTS: Compared with healthy controls, the ID/IDA cohort had higher SDSC total scores (43.97 ± 9.63 vs 34.61 ± 7.50; p < 0.001), with significant differences in the subscales difficulty in initiating and maintaining sleep (DIMS), sleep-wake transition disorders (SWTD), and sleep hyperhidrosis (SHY). Ten of 31 screened children (32.25%) had clinically plausible RLS-related symptoms and higher SWTD scores. Actigraphy showed shorter sleep duration, lower sleep efficiency, longer wake after sleep onset, and greater fragmentation. After iron treatment, parent reported restlessness improved, whereas actigraphic parameters showed only partial normalization. CONCLUSIONS: Pediatric ID/IDA was associated with caregiver-reported and actigraphic sleep disruption, characterized by restless and fragmented sleep. These findings support systematic sleep assessment in children with low iron stores and consideration of iron status in the work-up of restless or nonrestorative sleep.

Humans↗

PubMed2000

Chronic regional pain syndrome, type 1: Part II.

Chronic regional pain syndrome, type 1 (CRPS1) is a complex neurologic disease characterized by chronic, severe, burning pain; hyperesthesia; soft tissue swelling; dystrophy; hyperhidrosis; vasomotor and sudomotor instability; joint stiffness; and patchy osteoporosis. Five to six million people in the United States alone suffer from CRPS1. To date, CRPS1 is poorly understood and often is not recognized clinically. This syndrome requires early detection, pain control, and treatment in tandem with physical therapy to the affected area. Part I (published in September) discussed background information on CRPS1 and sympathetic nerve blocks. Part II focuses on the remaining treatment modalities (e.g., sympathectomy, physical therapy, stimulators, trigger point injections, acupuncture, tourniquet effects, placebo effects, amputation).

Chronic Disease↗

PubMed2005

[Segmental anhidrosis associated with Adie's pupil: an incomplete case of Ross syndrome].

We present the case of a 43-year-old woman who presented with a localized area of anhidrosis on the upper left trunk and ipsilateral upper limb. It was associated with Adie's tonic pupil and an area of compensatory hyperhidrosis on the contralateral side. After 9 years of follow up, the alterations have remained stable. These alterations of the autonomous nervous system correspond to an incompletely expressed Ross syndrome.

Adult↗

PubMed2005

[Reflex sympathetic dystrophy: description of a case with skin lesions].

Reflex sympathetic dystrophy or algodystrophy is a poorly defined syndrome in which the patient develops pain disproportionate to the cause. It is included among the complex regional pain syndromes. The symptoms are triggered by some type of trauma, at times trivial, and consist of burning pain, edema, changes in skin color, alterations in vascularization, temperature changes, hyperhidrosis and skin disorders, which primarily consist of atrophic changes. Other less frequent cutaneous manifestations have been described in patients with this syndrome. These include papules, blisters, inflammatory lesions and reticulated hyperpigmentation. We discuss the case of a patient with reflex sympathetic dystrophy who presented with superficial ulcers on the affected limb, which mimicked dermatitis artefacta.

Adult↗

PubMed1995

Gustatory facial sweating subsequent to upper thoracic sympathectomy.

Gustatory facial sweating has been described as a consequence of upper thoracic sympathectomy. Patients may also develop compensatory hyperhidrosis, sensory deficits, nipple hypersensitivity, and Horner's syndrome. In this article, we have reviewed three patients with reflex sympathetic dystrophy who developed gustatory facial sweating subsequent to endoscopic T2 and T3 ganglionectomy. This article also discusses the possible mechanisms of gustatory facial sweating.

Adult↗

PubMed1992

Asymmetrical autonomic dysfunction of the feet after retroperitoneal surgery in patients with testicular cancer: 2 case reports.

We report on 2 patients with hyperhidrosis and decreased temperature of the leg on the unoperated side after unilateral retroperitoneal lymph node dissection. Both patients had a 4 to 5C difference in skin temperature of the feet, with the operated side being warm and dry compared to the nonoperated side. This condition is most likely due to a lesion of sympathetic fibers or ganglia located in close proximity to the retroperitoneal lymph nodes, resembling a unilateral lumbar sympathectomy. In addition, both patients had profuse sweating and a subjective feeling of coldness of the leg on the nonoperated side, which caused considerable discomfort. This latter phenomenon most likely represents a compensatory sympathetic hyperfunction due to the decreased sympathetic function in the other leg.

Adult↗

PubMed2006

[Factors associated with palmoplantar or plantar pompholyx: a case-control study].

BACKGROUND: Disydrosis or pompholyx is a chronic and recurrent form of dermatosis that poses a number of therapeutic issues. The etiopathology of the condition is unclear. The aim of this study was to identify factors associated with the palmoplantar and plantar dysidrosis. PATIENTS AND METHODS: This was a prospective case-control study conducted between June 2001 and February 2004 at the University Hospital of Lome (Togo). Each case (palmoplantar or plantar pompholyx) was matched for age (+/- 5 years) and sex with two controls. Examination for tinea pedis was performed in all participants (patients and controls) but mycologic culture alone was done in patients with interdigital-plantar intertrigo. RESULTS: One hundred patients with pompholyx were matched with 200 controls. Mean age was 32.8 +/- 14.8 years in the patient group and 31.4 +/- 14.8 years in the control group. For univariate analysis, the main factors associated with pompholyx were: personal atopy (OR = 12.6; CI95%: 6.4 - 25.1) and familial atopy (OR = 5.8; CI95%: 3.2 - 10.5); history of eczema (OR = 5.4; CI95%: 2.6 - 11.4); hyperhidrosis (OR=4.5; CI 95%: 5.5 - 40.7), sport (OR = 8.8; CI 95%: 3.9 - 20.8); tinea pedis (OR = 15.6; CI 95%: 7.5 - 32.9). In multivariate analysis, atopy (OR = 10.5; CI95%: 8.4 - 20.8) and tinea pedis (OR = 18; CI95%: 10.5 - 25.2) were the only factors associated with pompholyx. Trichophyton rubrum was the most common etiology of tinea pedis in both patients and controls. DISCUSSION: The results of this study show atopy and tinea pedis as factors statistically associated with palmoplantar or plantar pompholyx. However, only cohort studies can determine the precise causal relationship between tinea pedis and pompholyx.

Adult↗

PubMed1988

Pachyonychia congenita.

Pachyonychia congenita is a rare hereditary disorder characterized mainly by nail hypertrophy and dyskeratoses of skin and mucous membranes. A thorough literature survey since its first description in 1904 up to 1985 revealed 168 cases of pachyonychia congenita. There were no indications of any sex or ethnic group predilection. Based on this survey the following classification is suggested: type I (56.2% of cases), hyperkeratosis of nails, palmoplantar keratosis, follicular keratosis, and oral leukokeratosis; type II (24.9% of cases), clinical findings of type I plus bullae of palms and soles, palmar and plantar hyperhidrosis, natal or neonatal teeth, and steatocystoma multiplex; type III (11.7% of cases), clinical findings of types I and II plus angular cheilosis, corneal dyskeratosis, and cataracts; and type IV (7.2% of cases), clinical findings of types I, II, and III plus laryngeal lesions, hoarseness, mental retardation, hair anomalies, and alopecia.

Humans↗

PubMed1989

Biology of sweat glands and their disorders. I. Normal sweat gland function.

The basic mechanisms of sweat gland function and an updated review of some relatively common disorders of sweat secretion, are presented. Although sweat secretion and ductal absorption are basically biophysical and biologic cellular processes, a detailed description of the basic biophysical principles of membrane transport has been avoided to make the discussion more readable. The cited references will, however, help those readers primarily interested in the basic details of sweat gland function. Part I of this article includes a discussion of morphologic characteristics, central and peripheral nervous control of sweat secretion, neurotransmitters, intracellular mediators and stimulus secretion coupling, Na-K-Cl cotransport model for the ionic mechanism of sweat secretion, ingredients of sweat, ductal function, the pathogenesis of abnormal sweat gland function in cystic fibrosis, and the discovery of the apoeccrine sweat gland. Part II, to be published in the May issue of the Journal, reviews reports of all those major disorders of hyperhidrosis and hypohidrosis that have appeared in the literature during the past 10 years. It is hoped that this review will serve as a resource for clinicians who encounter puzzling disorders of sweating in their patients, as well as for investigators who wish to obtain a quick update on sweat gland function.

Humans↗

PubMed1999

Eccrine angiomatous hamartoma: report of a case and literature review.

Eccrine angiomatous hamartoma is a rare condition characterized histologically by increased numbers of eccrine structures and numerous capillary channels. Patients characteristically have a solitary, congenital nodule that may be painful and that may show hyperhidrosis. It is important to recognize this condition because it is a benign lesion for which aggressive treatment is not indicated. We report the case of a congenital eccrine angiomatous hamartoma that had a firm nodule studded with blue papules.

Eccrine Glands↗

PubMed1989

Morvan's fibrillary chorea. A case with possible manganese poisoning.

The clinical picture of Morvan's fibrillary chorea includes a. spontaneous muscular activity resulting from repetitive motor unit action potentials of peripheral origin (multiplets), b. autonomic dysregulation with profuse hyperhidrosis, and c. central nervous system involvement as shown by severe insomnia and hallucinosis. A case featuring all these symptoms is presented. Whereas known causative factors range from gold or mercury poisoning to autoimmune disorders, the presented case is the first one in which chronic manganese intoxication (occupational exposure) seems to be implicated. Manganese has been found to inhibit acetylcholine esterase, and, as a consequence, may produce peripheral and central cholinergic hyperactivity.

Aged↗

PubMed2001

Despite clinical similarities there are significant differences between acute limb trauma and complex regional pain syndrome I (CRPS I).

In order to analyze the pathophysiology behind the clinical similarity acutely after limb trauma and in acute stages of complex regional pain syndrome (CRPS), 20 patients with external fixation after distal radius fracture (3.5 days after surgery) without signs of CRPS and 24 patients suffering from acute CRPS I (without nerve lesion; duration, 5 weeks) were investigated. Hyperalgesia to heat was tested by a feedback-controlled thermode, and to mechanical stimuli by an impact stimulator. The sympathetic nervous system was examined by measuring skin temperature (infra-red thermography), testing different sympathetic vasoconstrictor reflexes (laser-Doppler flowmetry) and quantitative sudometry after thermal load (thermoregulatory sweat test). We found hyperalgesia to heat after trauma (P<0.001), but not in CRPS, whereas mechanical hyperalgesia was present in both patient groups (trauma: P<0.001; CRPS: P<0.005). Skin temperature was significantly increased on the affected side in both patient groups (acute trauma: P<0.001; CRPS: P<0.005). However, sympathetic failure, as indicated by impairment of sympathetic vasoconstrictor reflexes (P<0.02) and hyperhidrosis (P<0.01), was found exclusively in CRPS patients. Our results indicate that pain and vasomotor disturbances may be generated by different mechanisms acutely after trauma and in acute CRPS. Despite the clinical similarity, additional changes in the peripheral or central nervous system are required for CRPS. In the light of our observations, it seems unlikely that CRPS is a simple exaggeration of post-traumatic inflammation.

Acute Disease↗

PubMed1999

The role of botulinus toxin type A in treatment--with special reference to children.

Although botulinum toxin A was first introduced to treat strabismus and blepherospasm it is now used in an increasing number of conditions, many in the field of pediatrics. Its action results from a prevention of the release of acetylcholine from nerve terminals. A number of studies recording the effects of the toxin in the treatment of spastic cerebral palsy are reviewed, and although these can be criticized, there seems to be no doubt that it can be of benefit. It is few side effects, but it may reveal an underlying weakness. Other disadvantages are its cost and the need for repeated injections. It can be used for the relief of rigidity, although the effects in the extrapyramidal form of cerebral palsy are not so dramatic. Also it can be beneficial in some forms of dystonia, rarely if this is generalized, but certainly if it is focal, and especially if there is accompanying pain. There are several conditions seen in children, such as strabismus, blepherospasm and tremors, in which this form of treatment will rarely be indicated; but they will be mentioned. An exception may be spasmodic torticollis during adolescence if it does not respond to other therapy, as it is so disabling. Botulinum toxin can be used to block the discharges from cholinergic sympathetic and parasympathetic terminals. Focal hyperhidrosis can be very distressing among older children, and the use of the toxin should sometimes be considered in this and other autonomic disorders.

Autonomic Nervous System Diseases↗

PubMed2004

Complications with the use of botulinum toxin.

This article discusses complications with the use of botulinum toxin. The following topics are explored: conditions caused by muscle spasms, resistance to botulinum toxin, cosmetic use of botulinum toxin, complications in treating hyperhidrosis, treatment of migraine headaches, and informed consent.

Botulinum Toxins, Type A↗

PubMed1993

Sympathetic nervous system disorders in man.

The sympathetic nervous system innervates most organs in the body and controls their function. A variety of disease processes, surgery or drugs can result in disordered sympathetic nerve function, which can be either localized or more generalized. Malfunction can result in either sympathetic underactivity (causing postural hypotension, impotence or anhidrosis) or overactivity (causing paroxysmal hypertension or hyperhidrosis). The investigation of sympathetic disorders depends upon the system and organs involved and should include, where relevant, investigation of the possible aetiological processes. The clinical features and management of some of the major disorders affecting the sympathetic nervous system, including the recently described syndrome of DBH deficiency, are described.

Dopamine beta-Hydroxylase↗

PubMed1999

Multiple paraneoplastic diseases associated with thymoma.

Here we report a patient with a lymphoepithelial thymoma who developed in chronological sequence limbic encephalitis, neuromyotonia and myasthenia gravis. The patient presented with limbic encephalitis associated with an invasive thymoma and improved after surgery and cytotoxic therapy. Two months after thymectomy, neuromyotonia associated with hyperhidrosis and mild motor neuropathy occurred and the patient was given plasma-exchange and prednisone therapy. Five months later he developed mild generalised myasthenia gravis. Anti-acetylcholine receptor antibodies, previously repeatedly negative, were found positive at the onset of clinical signs of myasthenia gravis.

Adult↗