Strongyloides Hyperinfection Syndrome Complicating (Ectopic) Cushing Syndrome

Vadlamudi, Raja; Dort, Martin Van; Barklow, Thomas; Byrd, Ryland P.; Moorman, Jonathan P.

doi:10.1097/smj.0b013e31817a836e

Cited by 4 publications

(5 citation statements)

References 10 publications

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“…After his death, a review of his clinical records showed that he had been previously diagnosed with strongyloidiasis and treated with a 3-day course of albendazole; although serology persisted positive and eosinophilia was still present 6 and 12 months after treatment, the patient did not receive any further therapy. Another patient who died from Strongyloides hyper infection had never been treated previously, despite a positive serology [101]. …”

Section: Resultsmentioning

confidence: 99%

Severe strongyloidiasis: a systematic review of case reports

et al. 2013

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BackgroundStrongyloidiasis is commonly a clinically unapparent, chronic infection, but immuno suppressed subjects can develop fatal disease. We carried out a review of literature on hyperinfection syndrome (HS) and disseminated strongyloidiasis (DS), in order to describe the most challenging aspects of severe strongyloidiasis.MethodsWe conducted a structured search using PubMed to collect case reports and short case series on HS/DS published from 1991 to 2011. We restricted search to papers in English, Spanish, Italian and French. Case reports were classified as HS/DS according to given definitions.ResultsRecords screened were 821, and 311 were excluded through titles and abstract evaluation. Of 510 full-text articles assessed for eligibility, 213 were included in qualitative analysis. As some of them were short case series, eventually the number of cases analyzed was 244.Steroids represented the main trigger predisposing to HS and DS (67% cases): they were mostly administered to treat underlying conditions (e.g. lymphomas, rheumatic diseases). However, sometimes steroids were empirically prescribed to treat signs and symptoms caused by unsuspected/unrecognized strongyloidiasis. Diagnosis was obtained by microscopy examination in 100% cases, while serology was done in a few cases (6.5%). Only in 3/29 cases of solid organ/bone marrow transplantation there is mention of pre-transplant serological screening. Therapeutic regimens were different in terms of drugs selection and combination, administration route and duration. Similar fatality rate was observed between patients with DS (68.5%) and HS (60%).ConclusionsProper screening (which must include serology) is mandatory in high - risk patients, for instance candidates to immunosuppressive medications, currently or previously living in endemic countries. In some cases, presumptive treatment might be justified. Ivermectin is the gold standard for treatment, although the optimal dosage is not clearly defined in case of HS/DS.

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Section: Resultsmentioning

confidence: 99%

Severe strongyloidiasis: a systematic review of case reports

et al. 2013

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“…The one reported by Vadlamudi et al. had combined SCLC, lung adenocarcinoma, and giant cell carcinoma of the lung ( 40 ).…”

Section: Resultsmentioning

confidence: 99%

“…For differential diagnosis of CS, 132 SCLC patients (84.1% of all 157 patients) in 58 reports (95.1% of all 61 reports) were diagnosed with ECS. In 9 (15.5%) reports, 40 (30.3%) patients were diagnosed without strict evidence from combining imaging examinations with laboratory tests or reported with no mention of specific procedures for the diagnosis of ectopic hormone secretion ( 8 , 10 , 22 , 40 , 45 , 49 , 57 , 58 , 72 ). The patient reported by Cabral et al.…”

Section: Resultsmentioning

confidence: 99%

Management of small cell lung cancer complicated with paraneoplastic Cushing’s syndrome: a systematic literature review

Li,

et al. 2023

Front. Endocrinol.

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Paraneoplastic Cushing’s syndrome (PCS) is a rare, but clinically important feature of small cell lung cancer (SCLC) that is associated with even worse prognosis. To identify key considerations in comprehensive management of SCLC patients complicated with PCS, we conducted a systematic review of relevant reports on PubMed and Web of Science, focusing on SCLC with PCS cases. The systematic review analyzed 61 reports published between 1985 and 2022 with a total of 157 SCLC patients included. Out of the 157 patients, 132 (84.1%) patients across 58 (95.1%) reports were diagnosed with ectopic Cushing’s syndrome. The immunohistochemical (IHC) staining for adrenocorticotropic hormone (ACTH) was performed on 30 (19.1%) patients across 22 (36.1%) reports and demonstrated encouraging performance. For treatment, chemotherapy and ketoconazole were utilized in 50 (81.97%) and 24 (39.34%) reports, respectively. Regarding cause of death, infection and cancer were equally frequent, each being recorded in 17 (27.87%) reports. To conclude, the majority of PCS cases in SCLC patients were caused by ectopic hormone secretion. In order to make a differential diagnosis, it is recommended to utilize IHC staining for a specific hormone such as ACTH or corticotropin-releasing hormone. In the comprehensive treatment of SCLC with PCS patients, effective management of hypercortisolism and potent safeguarding against infection play two crucial roles. Ultimately, further confirmations are required regarding the specificity and accuracy of IHC staining technique as well as the efficacy and safety of immunotherapy in the treatment of SCLC with PCS patients.

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“…Two cases of S. stercoralis hyperinfection accompanying Cushing syndrome have been previously reported 4,10 : one had respiratory symptoms and ectopic ACTH syndrome due to a small cell lung cancer 10 , the other had abdominal pain and Cushing syndrome of unknown etiology. 4 Although this is the third documented case, it is the first to highlight recurrent GI bleeding as the primary presenting symptom of hyperinfection in the setting of Cushing syndrome.…”

Section: Discussionmentioning

confidence: 99%

“…3 Widespread, extraintestinal larval invasion beyond the normal pattern of migration and into extraintestinal organs is characteristic of disseminated strongyloidiasis, also known as superinfection, which can lead to serious sequelae such as meningitis, polymicrobial bacteremia, sepsis, and death. 1,5,6,10 Strongyloidiasis is typically diagnosed in mild cases when a patient with a relevant travel history presents with eosinophilia, possibly but not always accompanied by abdominal pain or other complaints. Clinical examination, laboratory testing, imaging, and endoscopy 1,2 are not specific in the absence of an etiological diagnosis, which can be obtained by visualizing parasites in a stool examination (insensitive), histopathology (invasive), or serology (which can have crossreactions with other helminths; the negative schistosomiasis serologies helped to rule out that cross-reaction in this case).…”

Section: Discussionmentioning

confidence: 99%

Strongyloides stercoralis Hyperinfection Syndrome Presenting as Severe, Recurrent Gastrointestinal Bleeding, Leading to a Diagnosis of Cushing Disease

Yee¹,

Chi²,

Hansen³

et al. 2015

The American Society of Tropical Medicine and Hygiene

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Abstract.A 50-year-old male immigrant from Ethiopia presented for consultation after 3 years of hematochezia/melena requiring > 25 units of blood transfusions. Physical examination revealed severe proximal muscle wasting and weakness, central obesity, proptosis, and abdominal striae, accompanied by eosinophilia, elevated hemoglobin A1c, elevated 24-hour urinary cortisol, lack of suppression of 8 AM cortisol levels by 1 mg dexamethasone, and inappropriately elevated random adrenocorticotropic hormone (ACTH) level. Histopathological examination of gastrointestinal biopsies showed large numbers of Strongyloides stercoralis, indicating Strongyloides hyperinfection. Treatment with 2 days of ivermectin led to resolution of gastrointestinal bleeding. This syndrome was due to chronic immunosuppression from a pituitary ACTH (corticotroph) microadenoma, of which resection led to gradual normalization of urine cortisol, improved glycemic control, resolution of eosinophilia, and no recurrence of infection.

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Strongyloides Hyperinfection Syndrome Complicating (Ectopic) Cushing Syndrome

Cited by 4 publications

References 10 publications

Severe strongyloidiasis: a systematic review of case reports

Severe strongyloidiasis: a systematic review of case reports

Management of small cell lung cancer complicated with paraneoplastic Cushing’s syndrome: a systematic literature review

Strongyloides stercoralis Hyperinfection Syndrome Presenting as Severe, Recurrent Gastrointestinal Bleeding, Leading to a Diagnosis of Cushing Disease

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