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Shapiro syndrome: a cause of episodic hyperhidrosis, hypothermia and altered mental status

QJM. 2023 Jun 20:hcad145. doi: 10.1093/qjmed/hcad145. Online ahead of print.

ABSTRACT

INTRODUCTION: Shapiro syndrome is characterized by recurrent episodes of hypothermia and hyperhidrosis, with agenesis of the corpus callosum. This is a rare condition with only approximately 60 cases described worldwide. We describe a case of Shapiro syndrome.

METHODS/RESULTS: A 50-year-old Indian man with diabetes and hypertension presented with a 3-month history of frequent episodic profuse hyperhidrosis, accompanied by postural giddiness and confusion. He had episodes of isolated hyperhidrosis 20 years ago which had spontaneously resolved. These episodes re-emerged 3 years prior to presentation, with increasing frequency over the last 3 months. Previous extensive investigations, including a positron emission tomography (PET) scan were normal and he was treated for anxiety. During his inpatient stay, he was observed to have recurrent episodes of hypothermia (lowest temperature recorded 31.3 degrees Celsius), labile blood pressure (systolic ranging from 71mmHg to 175mmHg) and pulse rate (38/min to 214/min). Apart from slow responses to routine questioning, the rest of his neurological examination was normal. Extensive investigations looking for malignancy, autoimmune diseases and infections were unremarkable. CSF studies were negative for inflammation or infection. Magnetic resonance imaging of the brain demonstrated agenesis of the corpus callosum and schizencephaly. Based on his symptoms of hyperhidrosis, hypothermia and imaging findings, a diagnosis of Shapiro syndrome was made. He was treated with clonidine and levetiracetam with good response.

CONCLUSION: Shapiro syndrome is characterized by a triad of episodic hyperhidrosis, hypothermia and agenesis of the corpus callosum. Recognition of this rare condition is important to direct effective treatment.

PMID:37338563 | DOI:10.1093/qjmed/hcad145

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Comparison of CT-guided thoracic sympathetic nerve block and radiofrequency in the treatment of primary palmar hyperhidrosis

Front Surg. 2023 May 31;10:1126596. doi: 10.3389/fsurg.2023.1126596. eCollection 2023.

ABSTRACT

BACKGROUND: Primary palmar hyperhidrosis (PPH) is a condition marked by an overactive secretion of the hand’s exocrine glands and is frequently hereditary. The profuse sweating associated with this condition can significantly impair the patient’s daily activities and quality of life.

OBJECTIVE: The objective of this study was to compared the benefits and drawbacks of thoracic sympathetic block and thoracic sympathetic radiofrequency in the treatment of PPH.

METHODS: A retrospective analysis was conducted on 69 patients. They were divided into groups A and B according to their treatment. Group A (34 cases) received CT-guided percutaneous thoracic sympathetic nerve chain anhydrous alcohol chemical damage block, and group B (35 cases) received CT-guided percutaneous thoracic sympathetic nerve chain radiofrequency thermocoagulation.

RESULTS: Palmar sweating disappeared immediately after the operation. The recurrence rates at 1, 3, 6, 12, 24, and 36 months were 5.88% vs. 2.86% (P > 0.05), 20.59% vs. 5.71% (P > 0.05), 32.35% vs. 11.43% (P < 0.05),32.35% vs. 11.43% (P < 0.05), 25% vs. 14.71% (P < 0.05), and 68.75% vs. 20.59% (P < 0.05), respectively. The incidence of intercostal neuralgia and compensatory hyperhidrosis was higher in group A compared with of group B (52.94% vs. 22.86%, P < 0.05; 55.88% vs. 22.86%, P < 0.05).

CONCLUSION: Both methods were found to be effective in treating PPH, but thoracic sympathetic radiofrequency had a longer-term effect, a lower recurrence rate, and a lower incidence of intercostal neuralgia and compensatory hyperhidrosis than a thoracic sympathetic block.

PMID:37325414 | PMC:PMC10264635 | DOI:10.3389/fsurg.2023.1126596

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Attempted treatment of primary axillary hyperhidrosis with one session of either Neodymium YAG laser or Intense Pulsed Light: A within patient randomized trial of treated versus untreated contralateral axilla

Lasers Surg Med. 2023 Jun 14. doi: 10.1002/lsm.23697. Online ahead of print.

ABSTRACT

BACKGROUND: Primary axillary hyperhidrosis (PAH) affects 1-5% of the world’s population who has an unmet need for improved treatments. The heating of sweat glands with specific microwave therapy has shown promising results, yet, treatment with widely available devices such as long-pulsed Neodymium Yttrium Aluminum Garnet (Nd:YAG) lasers, diode lasers or Intense Pulsed Light (IPL) may serve as pragmatic alternatives.

OBJECTIVES: To compare sweat secretion of treated versus untreated contralateral control axilla 1-3 months after one session of Nd:YAG laser or IPL in patients with PAH.

METHODS: A within-person randomized controlled trial. Patients were randomized to receive either one session of Nd:YAG laser or IPL in one axilla with the contra-lateral serving as control. Sweat production was assessed by gravimetry, trans-epidermal water loss, hyperhidrosis disease severity scale and dynamic optical coherence tomography. Mixed-effects models were used to handle the within-person design, containing both fixed effect factors (side, group, and subgroup), and random effects (patients), while also adjusting for the level at baseline.

RESULTS: A total of 20 patients were enrolled. At follow-up 1-3 months after treatment, sweat secretion was not affected in the treated axilla when compared to the control axillae (0.01 [95%CI: -0.04 to 0.05]; p = 0.68). In the Nd:YAG subgroup (10 patients), least squares means for sweat secretion was 0.18 mg/5 min in the treated versus 0.15 mg/5 min in the control axilla, respectively, corresponding to a statistically insignificant mean difference of 0.02 mg/5 min (95% CI: -0.06 to 0.11; p = 0.54). In the IPL subgroup (10 patients), sweat secretion was 0.06 mg/5 min in the treated axilla versus 0.07 mg/5 min in the control axilla with a statistically insignificant difference of -0.01 points (95% CI: -0.03 to 0.02; p = 0.46). Likewise, none of the secondary outcomes were significantly affected by treatment. However, both treatments appeared safe and well tolerated with no adverse effects reported at follow-up.

CONCLUSIONS: One treatment with external 1064 nm Nd:YAG laser or 640 nm IPL at commercially available settings, failed to demonstrate clinical benefit in treating PAH, with narrow confidence intervals implying that this was not due to a type-2 error.

PMID:37313834 | DOI:10.1002/lsm.23697

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Primary Palmar Hyperhidrosis (PPH) Accompanied With Nevus Flammeus: A Case Report

Cureus. 2023 May 8;15(5):e38723. doi: 10.7759/cureus.38723. eCollection 2023 May.

ABSTRACT

Palmar hyperhidrosis (PH) is a medical condition characterized by excessive sweating in the palms of the hands, which can result in significant distress and impairment in daily activities. Flammeus nevus, on the other hand, is a benign vascular lesion that appears as a red or purplish discoloration on the skin, commonly found on the face, neck, or trunk. In some cases, flammeus nevus can co-occur with PH, leading to increased sweating in the affected area. This condition can cause significant psychosocial impact, affecting an individual’s quality of life (QoL) and self-esteem. We present a case report of a patient presenting with PPH with flammeus nevus. There is currently limited information available on the relationship between PH and flammeus nevus, and more research is needed to better understand this phenomenon; here we have reported the presentation of a patient. In conclusion, PH accompanied with flammeus nevus is a condition that requires prompt attention and management to mitigate its adverse effects. We have used ChatGPT to aid in structuring and writing this case report.

PMID:37292575 | PMC:PMC10246863 | DOI:10.7759/cureus.38723

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Hormonal Causes for Excessive Sweating

Praxis (Bern 1994). 2023 Jun;112(7-8):398-402. doi: 10.1024/1661-8157/a004025.

ABSTRACT

Hormonal Causes for Excessive Sweating Abstract: Excessive sweating is a frequent symptom in the general practice, but quite a few patients report their sweating problems only when explicitly asked. The differentiation into night sweats on the one hand and general sweating on the other hand can give us first diagnostic hints. Based on their frequency, night sweats should also trigger questions about panic attacks or sleeping disorders. The most frequent hormonal causes for excessive sweating are the menopause and hyperthyroidism. Hypogonadism in the aging male is a rather rare cause for excessive sweating and must be associated with sexual problems and a repeatedly low morning testosterone. This article provides an overview about the most frequent hormonal causes of excessive sweating and the diagnostic approach.

PMID:37282519 | DOI:10.1024/1661-8157/a004025

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Hyperhidrosis quality of life index (HidroQoL): further validation by applying classical test theory and item response theory using data from a phase III clinical trial

J Patient Rep Outcomes. 2023 Jun 6;7(1):55. doi: 10.1186/s41687-023-00596-6.

ABSTRACT

BACKGROUND: The Hyperhidrosis Quality of Life Index (HidroQoL ©) is a well-developed and validated patient-reported outcome measure assessing the quality-of-life impacts in hyperhidrosis with 18 items. Our aim was to extend the already existing validity evidence for the HidroQoL, especially in relation to structural validity. Especially Rasch analysis has not been applied to the final 18-item HidroQoL before.

METHODS: Data from a phase III clinical trial were used. Confirmatory factor analysis was conducted to confirm the two a priori HidroQoL scales within classical test theory. Furthermore, the assumptions of the Rasch model (model fit, monotonicity, unidimensionality, local independence) and Differential Item Functioning (DIF) were assessed using item response theory.

RESULTS: The sample included 529 patients with severe primary axillary hyperhidrosis. The two-factor structure could be confirmed by the confirmatory factor analysis (SRMR = 0.058). The item characteristic curves showed mainly optimally functioning response categories, indicating monotonicity. The overall fit to the Rasch model was adequate and unidimensionality for the HidroQoL overall scale could be confirmed, since the first factor had an eigenvalue of 2.244 and accounted for 18.7%. Local independence was below assumed thresholds (residual correlations ≤ 0.26). DIF analysis, controlling for age or gender, was critical for four and three items, respectively. However, this DIF could be explained.

CONCLUSION: Using classical test theory and item response theory/Rasch analyses, this study provided further evidence for the structural validity of the HidroQoL. This study confirmed several specific (measurement) properties of the HidroQoL questionnaire in patients with physician-confirmed severe primary axillary hyperhidrosis: the HidroQoL is a unidimensional scale allowing the summation of scores to generate a single score, and simultaneously it has a dual structure, also allowing the calculation of separate domain scores for daily activities and psychosocial impacts. With this study, we provided new evidence of the structural validity of the HidroQoL in the context of a clinical trial. Trial registration The study was registered (ClinicalTrials.gov identifier: NCT03658616, 05 September 2018, https://clinicaltrials.gov/ct2/show/NCT03658616?term=NCT03658616&draw=2&rank=1 ).

PMID:37280417 | DOI:10.1186/s41687-023-00596-6

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The differentiating effect of COVID-19-associated stress on the morbidity of blood donors with symptoms of hidradenitis suppurativa, hyperhidrosis, or psoriasis

Qual Life Res. 2023 Jun 3. doi: 10.1007/s11136-023-03448-4. Online ahead of print.

ABSTRACT

PURPOSE: The burden of different skin diseases may vary leading individuals to have different sensitivity to stress. Therefore, we compared the health-related quality of life (HRQoL) and stress before and during the universal stress from the severe acute respiratory syndrome coronavirus-2-pandemic in individuals with and without hyperhidrosis, hidradenitis suppurativa, or psoriasis.

METHODS: The study cohort was the Danish Blood Donor Study. Overall, 12,798 participants completed a baseline questionnaire before the pandemic, in 2018-2019, and a follow-up questionnaire during the pandemic, in 2020. Regression determined the association between the skin diseases and outcomes. Outcomes were the physical and mental component summary (MCS, PCS, respectively), which assess the mental and physical HRQoL, and the perceived stress scale, which assesses stress in the past four weeks.

RESULTS: Overall, 1168 (9.1%) participants had hyperhidrosis, 363 (2.8%) had hidradenitis suppurativa, and 402 (3.1%) had psoriasis. At follow-up, the participants with hyperhidrosis had worse MCS (coefficient -0.59 [95% confidence interval (CI) -1.05, -0.13]) and higher odds of moderate-to-severe stress (odds ratio 1.37 [95% CI 1.13, 1.65]) and the participants with hidradenitis suppurativa worse PCS (coefficient -0.74 [95% CI -1.21, -0.27]) than the control groups. The associations were independent of baseline HRQoL, stress, the Connor-Davidson Resilience scale, and other covariables. Psoriasis was not associated with the outcomes.

CONCLUSION: Individuals with hyperhidrosis or hidradenitis suppurativa experienced worse mental or physical well-being and individuals with hyperhidrosis also had higher stress during the pandemic compared to healthy individuals. This suggests that individuals with these skin diseases are particularly susceptible to external stress.

PMID:37270451 | DOI:10.1007/s11136-023-03448-4