Panhypopituitarism Following Hemorrhagic Fever with Renal Syndrome Due to Hantavirus

Kayabaş, Abdülcelil; Akgün, Ali Oğuz; Atilla, Aynur; Güzel, Murat; Erenler, Ali Kemal; Gürses, Gül Kefeli

doi:10.5152/jaemcr.2015.799

Cited by 1 publication

(3 citation statements)

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“…Table 3 summarises eleven previous case reports (2, 3, 4, 5, 6, 8, 14, 15, 16, 17, 18) with 14 well-characterised cases of HFRS-related panhypopituitarism. Five patients were diagnosed with hypopituitarism requiring chronic hormonal replacement, starting from the acute phase of HFRS (3, 6, 15, 17, 18).…”

Section: Discussionmentioning

confidence: 99%

“…Five patients were diagnosed with hypopituitarism requiring chronic hormonal replacement, starting from the acute phase of HFRS (3, 6, 15, 17, 18). Nine patients developed late-onset panhypopituitarism months or years after HFRS (2, 3, 4, 5, 8, 14, 16).…”

Section: Discussionmentioning

confidence: 99%

“…(17)xxxx b –AcuteHaemorrhage, atrophy (MRI)16 moNoKaybas et al . (18)xN/Ax–N/AAcuteNormal (MRI)N/AN/AACTH, adrenocorticotropic hormone; AVP, arginine vasopressin; d, days; FSH, follicle-stimulating hormone; GH, growth hormone; LH, luteinizing hormone; mo, months; N/A, data not available; TSH, thyroid-stimulating hormone; y, years.*GH/IGF-1 axis deficiency was diagnosed by determining a serum GH level, or b serum IGF-1 level or by conducting a c GH axis stimulation test.**Adrenocortical and thyroid axis recovered, and gonadal axis did not. …”

Section: Discussionmentioning

confidence: 99%

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Autoimmune polyendocrinopathy and hypophysitis after Puumala hantavirus infection

Tarvainen

Mäkelä

Mustonen

et al. 2016

Endocrinology, Diabetes &Amp; Metabolism Case Reports

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SummaryPuumala hantavirus (PUUV) infection causes nephropathia epidemica (NE), a relatively mild form of haemorrhagic fever with renal syndrome (HFRS). Hypophyseal haemorrhage and hypopituitarism have been described in case reports on patients with acute NE. Chronic hypopituitarism diagnosed months or years after the acute illness has also been reported, without any signs of a haemorrhagic aetiology. The mechanisms leading to the late-onset hormonal defects remain unknown. Here, we present a case of NE-associated autoimmune polyendocrinopathy and hypopituitarism presumably due to autoimmune hypophysitis. Thyroid peroxidase antibody seroconversion occurred between 6 and 12 months, and ovarian as well as glutamate decarboxylase antibodies were found 18 months after acute NE. Brain MRI revealed an atrophic adenohypophysis with a heterogeneous, low signal intensity compatible with a sequela of hypophysitis. The patient developed central (or mixed central and peripheral) hypothyroidism, hypogonadism and diabetes insipidus, all requiring hormonal replacement therapy. This case report suggests that late-onset hormonal defects after PUUV infection may develop by an autoimmune mechanism. This hypothesis needs to be confirmed by prospective studies with sufficient numbers of patients.Learning points:Pituitary haemorrhage resulting in hypopituitarism has been reported during acute HFRS caused by PUUV and other hantaviruses.Central and peripheral hormone deficiencies developing months or years after HFRS have also been found, with an incidence higher than that in the general population. The pathogenesis of these late-onset hormonal defects remains unknown.This case report suggests that the late-onset hypopituitarism and peripheral endocrine defects after HFRS could evolve via autoimmune mechanisms.The sensitivity of current anti-pituitary antibody (APA) tests is low. A characteristic clinical course, together with typical brain MRI and endocrine findings may be sufficient for a non-invasive diagnosis of autoimmune hypophysitis, despite negative APAs.

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