Hyperhidrosis, another hallmark of SS, was present in only 42.3% of the cases and mainly in adult onset. [ncbi.nlm.nih.gov]

Shapiro syndrome presenting with catatonia and thrombocytopenia. [neurologyindia.com]

Case Presentation The patient is a 35-year-old male who presented with a chief complaint of temperature fluctuations dating back to November 2020. [cureus.com]

Patients with Shapiro syndrome present with episodic or periodic hypothermia and whole-body hyperhidrosis 1,3-5. Episodes typically occur without warning and last hours in length 1,3-5. [radiopaedia.org]

We decided to present this case because we believe this syndrome should also be considered as a rare cause of fever of unknown origin. Case Presentation A 6-month-old female patient was admitted to the pediatrics unit for fever of unknown origin. [openaccesspub.org]

• Plethora

  Yet in some case reports, features suggestive of catatonia have been reported, for example, mutism or little speech, immobility, unresponsiveness, blank facial expressions, and withdrawn behaviour.[1],[7],[9],[10],[11],[12],[16],[26] A plethora of conditions [neurologyindia.com]

• Precocious Puberty

  Typically there are no other neuropsychiatric symptoms or other evidence of hypothalamic dysfunction (e.g. hypogonadism, diabetes insipidus, precocious puberty) 1,3-5. [radiopaedia.org]

• Alopecia

  Support Organizations Children’s Alopecia Project, Inc. General Support Organization Retina UK General Support Organization PID UK General Support Organization [globalgenes.org]

A sepsis workup was also performed as well as procalcitonin (<0.12 ng/mL) and C-reactive protein (0.94 mg/L) tests, peripheral blood smear, blood culture (at least 3 times), cerebrospinal fluid culture and analysis, complete urinalysis, urine culture, [openaccesspub.org]

• Epileptiform Activity

  Epileptiform activity was not observed.There was no detectable pathology during MRT examination. sinus rhythm followed in the ECG.30 mq/kg depakin was prescribed to patient and the patient was controlled.After 3 weeks the number of sweating attacks were [adc.bmj.com]

• Hyponatremia

  He was diagnosed with Shapiro syndrome, which may be due to hypothalamic dysfunction and is responsive to clonidine and carbamazepine.1,2 Because of lack of clonidine and hyponatremia, the patient was treated with levetiracetam, and the episodes disappeared [n.neurology.org]

It is recommended that treatment options be discussed with a health care professional. Only a patient's personal health care provider can determine the appropriate course of treatment. [rareguru.com]

This patient did not respond to cyproheptadine treatment 8. [openaccesspub.org]

Although different treatment options have been attempted in some patients, the treatments have been unsuccessful or of doubtful efficacy because of the small number of individuals that have been documented as having this condition. [globalgenes.org]

There is no definitive treatment. [cureus.com]

Treatment and prognosis Management is guided by case report-level evidence, with one notably effective agent being cyproheptadine 1-3. Shapiro syndrome Promoted articles (advertising) [radiopaedia.org]

Treatment and prognosis Management is guided by case report-level evidence, with one notably effective agent being cyproheptadine 1-3. Shapiro syndrome Promoted articles (advertising) [radiopaedia.org]

Evaluation for etiology of central hypogonadism revealed markedly elevated serum ferritin concentration > 1000 ng/dl on more than one occasion. [scirp.org]

In 2012, Guha et al. reported encountering the signs of reverse Shapiro’s syndrome while investigating the etiology of a fever of unknown origin in a 3-month-old female patient. [openaccesspub.org]

[…] complete blood count with differential, parathyroid hormone, thyroid-stimulating hormone, urinary analysis, procalcitonin, quantiferon testing, blood cultures, Covid testing, lipase, liver function tests, urine drug screen, screens for various infectious etiologies [cureus.com]

The absence of an etiology to explain these recurrent episodes of hypothermia with hyperhidrosis led us to suspect the diagnosis of Shapiro’s syndrome variant. Little is known about the pathophysiology of Shapiro’s syndrome. [thejournalofheadacheandpain.biomedcentral.com]

Contemporary epidemiology and characterization of newborn males with prune belly syndrome. Urology 2010;76(1):44-8. PubMed PMID: 20381841. Epub 2010/04/13. eng. 7. Salihu HM, Tchuinguem G, Aliyu MH, Kouam L. [revistapediatria.org]

Genetic epidemiology of hereditary hemorrhagic telangiectasia in a local community in the northern part of Japan. Hum Mutat. 2002;19(2):140-8. https://doi.org/10.1002/humu.10026 Grosse SD, Boulet SL, Grant AM, Hulihan MM, Faughnan ME. [revistagastrocol.com]

In conclusion, episodic headache with spontaneous hypothermia appear to be very rare in clinical practice and little is known about its pathophysiology. [thejournalofheadacheandpain.biomedcentral.com]

Its pathophysiology dwells on the role of dopaminergic hypersensitivity caused by hypothalamic dysfunction. Until now, only 5 cases have been described in the literature as reverse Shapiro’s syndrome. [openaccesspub.org]

Pathophysiologic and anesthetic correlations of the prune-belly syndrome. AANA journal 1989;57(2):137-41. 16. Woodard JR. Prune Belly syndrome. En: King LR, Panayotis KP, Belman AB, editores. Clinical Pediatric Urology. [revistapediatria.org]

We believe that all these manifestations including hypogonadism may be induced by hypothalamic dysfunction implicated in pathophysiology of Shapiro Syndrome. [scirp.org]

L thyroxine was also prescribed to suppress further enlargement of the documented nodules and prevent formation of new ones. [scirp.org]

Journal of Preventive Medicine And Care - 2(4):1-6. [openaccesspub.org]