Functional Neurological Symptom Disorder in Williams Syndrome: Case series and Review of Relevant Literature

A rare manifestation during the post-anesthetic period may include the occurrence of functional neurological symptom disorder (FNSD). FNSD is described as neurological symptoms that are not consistently explained by neurological or medical conditions. We report a case series consisting of six patients who underwent a general anesthetic at a tertiary referral hospital and experienced FNSD in the immediate post-anesthetic period. Life-threatening causes were excluded based on benign physical exam findings and knowledge of past history. Five of six cases manifested with FNSD only in the immediate post-operative setting after exposure to anesthesia, and never otherwise experienced these symptoms during their normal daily lives. MEDLINE and Google Scholar were searched through October 2019 using a highly-sensitive search strategy and identified 38 published cases of post-anesthetic FNSD. Meta-analysis of pooled clinical data revealed that a significant proportion of patients were females (86%), reported a history of psychiatric illness (49%), reported a prior history of FNSD (53%), and underwent general anesthesia as the primary anesthetic (93%). The majority of patients were exposed to diagnostic studies (66% received radiographic tests and 52% received electroencephalogram) as well as pharmacologic therapy (57%). While no deaths occurred, many patients had unanticipated admission to the hospital (53%) or to the intensive care unit (25%). These data may help inform the anesthesia literature on presentation, risk factors, and treatment outcomes of FNSD in the context of anesthetic administration. We contemplate whether anesthetic agents may predispose a vulnerable brain to manifest with involuntary motor and sensory control seen in FNSD.

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A Novel Treatment Protocol (Nocebo Hypothesis Cognitive Behavioural Therapy; NH-CBT) for Functional Neurological Symptom Disorder/Conversion Disorder: A Retrospective Consecutive Case Series

Behavioural and Cognitive Psychotherapy ◽

10.1017/s1352465817000832 ◽

2018 ◽

Vol 46 (4) ◽

pp. 497-503 ◽

Cited By ~ 3

Author(s):

Matt Richardson ◽

Gina Isbister ◽

Brad Nicholson

Keyword(s):

Neurological Symptom ◽

Treatment Protocol ◽

Case Series ◽

Conversion Disorder ◽

Functional Independence ◽

Interdisciplinary Treatment ◽

Controlled Study ◽

Consecutive Case ◽

Symptom Remission ◽

Functional Neurological Symptom Disorder

Background: Theories concerning the aetiology of functional neurological symptom disorder (FNSD; also known as conversion disorder) have historically inferred that psychological factors or dissociative states underlie patients’ symptoms. Current psychological models of functional neurological symptoms suggest that some type of ‘top-down’ representations/beliefs are activated automatically (without conscious awareness), leading to symptoms. It is assumed that these representations or beliefs are similar to the idea ‘I am neurologically damaged’, as in our clinical experience, almost all patients have some reason to doubt the integrity of their neurological system. Aims: It was hypothesized that FNSD arises from a belief of being neurologically damaged (via a mechanism akin to a nocebo response), and an interdisciplinary treatment protocol was developed consistent with this hypothesis, transparently sharing this theory with participants. Method: A retrospective consecutive case series design was utilized, measuring functional independence and symptom remission. Results: Of the 13 episodes of care, 12 resulted in complete or almost complete symptom remission. Length of stay in rehabilitation was also reduced compared with previous treatment attempts. Conclusions: It appears as if the treatment protocol may be very effective, and further controlled study appears warranted.

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Brief Report: Major Depressive Disorder with Psychotic Features in Williams Syndrome: A Case Series

Journal of Autism and Developmental Disorders ◽

10.1007/s10803-017-3384-x ◽

2017 ◽

Vol 48 (3) ◽

pp. 947-952 ◽

Cited By ~ 4

Author(s):

Francisca Valdes ◽

Christopher J. Keary ◽

Jennifer E. Mullett ◽

Michelle L. Palumbo ◽

Jessica L. Waxler ◽

...

Keyword(s):

Major Depressive Disorder ◽

Depressive Disorder ◽

Williams Syndrome ◽

Case Series ◽

Major Depressive ◽

Psychotic Features

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Management of Isolated Capitate Nonunion: A Case Series and Literature Review

Journal of Wrist Surgery ◽

10.1055/s-0038-1651487 ◽

2018 ◽

Vol 07 (05) ◽

pp. 419-423 ◽

Cited By ~ 2

Author(s):

Rishabh Jethanandani ◽

Schneider Rancy ◽

Keith Corpus ◽

Jeffrey Yao ◽

Scott Wolfe

Keyword(s):

Literature Review ◽

Internal Fixation ◽

Bone Grafting ◽

Relevant Literature ◽

Case Series ◽

Rare Condition ◽

Vascular Supply ◽

Autogenous Bone ◽

Rare Injury ◽

Autogenous Bone Grafting

Background Isolated capitate nonunion is rare. No consensus on the appropriate treatment for this condition exists. Case Description We reported two cases of capitate fracture nonunion presenting several months after untreated high-impact wrist trauma. Treatment was delayed as both patients' nonunions were missed on conventional radiographs. Both were ultimately diagnosed with advanced imaging and successfully treated with internal fixation and autogenous bone grafting. The relevant literature pertaining to capitate nonunion was reviewed. Literature Review Immobilization and internal fixation with bone grafting for capitate nonunion have been described in the literature. Loss of vascular supply and progression to avascular necrosis is a concern after capitate nonunion. Clinical Relevance We present two cases and review the literature on the diagnosis and treatment of this rare injury to guide management. Internal fixation with autogenous bone grafting could play a role in management for this rare condition.

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Overview of the Relevant Literature on the Possible Role of Acupuncture in Treating Male Sexual Dysfunction

Acupuncture in Medicine ◽

10.1136/acupmed-2014-010592 ◽

2014 ◽

Vol 32 (5) ◽

pp. 406-410 ◽

Cited By ~ 5

Author(s):

Ming-Yen Tsai ◽

Chun-Ting Liu ◽

Cheng-Chieh Chang ◽

Shih-Yu Chen ◽

Sheng-Teng Huang

Keyword(s):

Sexual Dysfunction ◽

Premature Ejaculation ◽

Relevant Literature ◽

Case Series ◽

Sham Acupuncture ◽

Risk Of Bias ◽

Inclusion Criteria ◽

Male Sexual Dysfunction ◽

Randomised Controlled

Objective To systematically review scientific reports on the effectiveness of acupuncture to treat male sexual dysfunction. Methods The Medline database was searched for published clinical trials of acupuncture for erectile dysfunction (ED) and premature ejaculation (PE) with English abstracts. Risk of bias was assessed for randomised controlled trials (RCTs). Results Seven studies on two conditions of male sexual dysfunction met the inclusion criteria. Three out of four RCTs were patient-blinded, but all had a high risk of bias. Three suggested that acupuncture has a therapeutic effect as compared with sham acupuncture. Comparisons with paroxetine were inconsistent. Other uncontrolled studies and case series suggested satisfactory improvements of ED and PE after acupuncture. Conclusions Acupuncture appears to have promise for treating male sexual dysfunction, but in view of the small number of studies and their variable quality, doubts remain about its effectiveness. Further studies are justified.

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Phosphaturic Mesenchymal Tumors of the Sinonasal Area and Skull Base: Experience at a Single Institution

Annals of Otology Rhinology & Laryngology ◽

10.1177/00034894211037416 ◽

2021 ◽

pp. 000348942110374

Author(s):

Davis P. Argersinger ◽

Catherine T. Haring ◽

John E. Hanks ◽

Kevin J. Kovatch ◽

S. Ahmed Ali ◽

...

Keyword(s):

Skull Base ◽

Surgical Resection ◽

Medical Center ◽

Fibroblast Growth Factor 23 ◽

Academic Medical Center ◽

Relevant Literature ◽

Case Series ◽

Radiological Evidence ◽

Lateral Skull Base ◽

Complete Surgical Resection

Objectives: Phosphaturic mesenchymal tumor (PMT) is a rare, polymorphous neoplasm with a highly variable presentation and natural history and unpredictable clinical course. The primary objective was to describe our clinical experience with and management of 4 markedly different cases of sinonasal and skull base PMT. Methods: A retrospective case series with chart review, and relevant literature review, was performed at a tertiary academic medical center between 1998 and 2020. Adult patients treated for PMTs of the sinonasal area and skull base were included. Our main outcome measures included postoperative laboratory findings and radiological evidence of disease remission. Results: Four patients (2 Males, 2 Females; Mean Age: 63.5 years) with PMTs of the skull base have been managed at our institution since 1998. Patient presentations varied, ranging from severe phosphorus wasting and osteoporosis to symptoms secondary to mass effect, including nasal obstruction and rhinorrhea. All 4 patients were eventually found to have elevated levels of fibroblast growth factor 23. Tumors were located in the sinonasal area (right frontal sinus, right ethmoid sinus, and right nasal cavity, respectively) in 3 patients and in the lateral skull base (right jugular foramen) in 1 patient. All 4 patients underwent complete surgical resection of their tumors. PMT tissue pathology was confirmed in all cases. Gross total resection was achieved in all patients. There was no chemical or radiological evidence of disease recurrence in any patients at follow-up. Conclusions: The presentation of skull base PMT is variable, and it may mimic other mass pathologies of the head and neck. Complete surgical resection with negative margins is potentially curative.

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Thy3 cytology: what to do next?

Annals of The Royal College of Surgeons of England ◽

10.1308/147870811x565232 ◽

2011 ◽

Vol 93 (3) ◽

pp. 225-228 ◽

Cited By ~ 6

Author(s):

Raj Lakhani ◽

Thomas Rourke ◽

Anthony Jefferis ◽

Louise Perry ◽

Sabour Ghiacy ◽

...

Keyword(s):

Relevant Literature ◽

Significant Risk ◽

Case Series ◽

District General Hospital ◽

Needle Aspiration ◽

Thyroid Fine Needle Aspiration ◽

Risk Of Malignancy ◽

Thyroid Fna ◽

The Uk ◽

Lost To Follow Up

INTRODUCTION Cytological analysis of thyroid fine needle aspiration (FNA) is aided by the ‘Thy’ classification. However, there is often confusion surrounding the management of patients with a Thy3 classification. A subdivision of Thy3 has been created to help reduce this dilemma but its use within the UK appears to by infrequent. This paper analyses the management of patients with Thy3 cytology from FNA of a thyroid nodule in a UK case series and reviews the relevant literature. It also describes the results of a survey of selected UK ENT departments on the use of the Thy3 classification and its subdivisions. PATIENTS AND METHODS A retrospective analysis of a case series of patients was undertaken. In addition, a telephone survey of local/regional pathology departments was conducted to assess the utilisation of the Thy classification and to assess the awareness and usage of the Thy3 subdivisions. RESULTS A total of 39 Thy3 results (11 males, 28 females) were identified from 2007 to 2009. Of these, 24 patients went on to have surgery, 8 had a further FNA, 2 had a Tru-cut biopsy and 5 were lost to follow-up. Eleven (28.2%) patients were subsequently diagnosed with a thyroid malignancy. The survey identified that none of the departments had adopted the Thy3 subclassifications and only 40% were aware of them. CONCLUSIONS Thy3 results from thyroid FNA have a significant risk of malignancy but there remains confusion surrounding their management. This district general hospital has adopted and recommends the use of the Thy3 (i) and Thy3 (ii) subdivisions in order to assist decision-making and avoid delays in treatment or unnecessary surgery.

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Long-term stenting for chronic frontal sinus disease: case series and literature review

The Journal of Laryngology & Otology ◽

10.1017/s0022215110001052 ◽

2010 ◽

Vol 124 (11) ◽

pp. 1216-1222 ◽

Cited By ~ 17

Author(s):

B Hunter ◽

S Silva ◽

R Youngs ◽

A Saeed ◽

V Varadarajan

Keyword(s):

Frontal Sinus ◽

Relevant Literature ◽

Case Series ◽

Outflow Tract ◽

Sinus Surgery ◽

Sinus Disease ◽

Narrow Channels ◽

Co Morbidity ◽

Disease Case

AbstractObjective:The frontal sinus outflow tract consists anatomically of narrow channels prone to stenosis. Following both endonasal and external approach surgery, up to 30 per cent of patients suffer post-operative re-stenosis of the frontal sinus outflow tract, with recurrent frontal sinus disease. This paper proposes the surgical placement of a long-term frontal sinus stent to maintain fronto-nasal patency, as an alternative to more aggressive surgical procedures such as frontal sinus obliteration and modified Lothrop procedures.Design:We present a series of three patients with frontal sinus disease and significant co-morbidity, the latter making extensive surgery a significant health risk. We also review the relevant literature and discuss the use of long-term frontal sinus stenting.Results:These three cases were successfully treated with long-term frontal sinus stenting. Stents remained in situ for a period ranging from 48 to over 60 months.Conclusion:Due to the relatively high failure rates for both endonasal and external frontal sinus surgery, with a high post-operative incidence of frontal sinus outflow tract re-stenosis, long-term stenting is a useful option in carefully selected patients.

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