Lymphocytic infundibulo-neurohypophysitis: An unusual cause of recurrent optic neuropathy in a child

Al‐Mujaini, Abdullah; Ganesh, Anuradha; Al-Zuhaibi, Sana; Al-Dhuhli, Humoud; Al-Mashani, Ali; Al-Kindi, Hunaina; Al-Memari, Ali; Futaisi, Amna Al; Al‐Asmi, Abdullah

doi:10.1016/j.jaapos.2008.11.003

Cited by 13 publications

(4 citation statements)

References 9 publications

(8 reference statements)

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“…Cases which involve anterior and posterior pituitary (25%) are twice as common in women, and inflammation involving only the neurohypophysis (10%) occurs equally in both sexes. The condition also occurs rarely in childhood 7,8 . Headache (46%) and visual disturbance (33%) are common presenting features.…”

Section: What Are the Clinical Features Of Autoimmune Hypophysitis?mentioning

confidence: 99%

How reliably can autoimmune hypophysitis be diagnosed without pituitary biopsy

Howlett

Levy

Robertson

2010

Clinical Endocrinology

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SummaryAutoimmune hypophysitis is a rare chronic inflammatory condition of the pituitary gland which typically presents with hypopituitarism and a pituitary mass. Cases involving anterior pituitary alone (65%) are six times more common in women, typically presenting during pregnancy or postpartum (57%). Anterior and posterior pituitary involvement (25%) are twice as common in women, and neurohypophysis alone (10%) occurs equally in both sexes. It has a prevalence of around 5 per million, an annual incidence of 1 in 7 to 9 million and in our experience represents the known or suspected cause of 0AE5% of cases of hypopituitarism, <1% of pituitary masses and 2% of nonfunctioning macro lesions presenting to an endocrine clinic. However, 'missed' cases of autoimmune hypophysitis may be the aetiology of some other unexplained cases of hypopituitarism. Clinically, headache and visual disturbance are common. Anterior hypopituitarism shows a characteristic but atypical pattern of deficiency of ACTH followed by TSH, gonadotrophins and prolactin deficiency or hyperprolactinaemia. Eighteen percent of cases have evidence of another autoimmune condition. On magnetic resonance imaging (MRI), autoimmune hypophysitis is typically symmetrical and homogeneous with thickened but undisplaced stalk in contrast to typical findings with pituitary tumours. Ultimately, the histological diagnosis of autoimmune hypophysitis can only be confirmed by surgery but a presumptive diagnosis can often be made on the basis of a combination of context and clinical features, and pituitary biopsy is not always clinically necessary for effective clinical management of the patient.

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Section: What Are the Clinical Features Of Autoimmune Hypophysitis?mentioning

confidence: 99%

How reliably can autoimmune hypophysitis be diagnosed without pituitary biopsy

Howlett

Levy

Robertson

2010

Clinical Endocrinology

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“…There has been no report regarding the combination of optic neuritis and hypophysitis which would be related with COVID-19 or vaccination against COVID-19. Before the COVID-19 pandemic, the combination of optic neuritis and hypophysitis, especially with the diagnosis of diabetes insipidus, has been reported as case reports: 15 patients with sufficient description [19][20][21][22][23][24][25][26][27][28][29][30][31][32][33] are summarized, together with the present case, in Table 2.…”

Section: Discussionmentioning

confidence: 99%

Bilateral Optic Neuritis and Hypophysitis With Diabetes Insipidus 1 Month After COVID-19 mRNA Vaccine: Case Report and Literature Review

Matsuo

Okubo

Mifune³

et al. 2023

Journal of Investigative Medicine High Impact Case Reports

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Either optic neuritis (neuropathy) or hypopituitarism has been known to occur separately after COVID-19 vaccination. In this report, we describe the rare combination of hypophysitis and optic neuritis which occurred after COVID-19 vaccination. A 74-year-old woman began to have thirst, polydipsia, and polyuria, and was diagnosed as central diabetes insipidus 1 month after the fourth COVID-19 mRNA vaccine. Head magnetic resonance imaging (MRI) disclosed the thickened pituitary stalk and enlarged pituitary gland with high contrast enhancement as well as the absence of high-intensity signals in the posterior pituitary lobe on the T1-weighted image, leading to the diagnosis of lymphocytic hypophysitis. She was well with desmopressin nasal spray until further 2 months later, when she developed bilateral optic neuritis, together with gait disturbance, intention tremor of the upper extremities, urinary retention, constipation, abnormal sensation in the distal part of the lower extremities, and moderate hemiplegia on the left side. Autoantibodies, including anti-aquaporin 4 (AQP4) and anti-myelin oligodendrocyte glycoprotein (MOG), were all negative. She showed multifocal spinal cord lesions on MRI and oligoclonal bands in the cerebrospinal fluid obtained by spinal tap, and so underwent steroid pulse therapy with methylprednisolone in the tentative diagnosis of multiple sclerosis, resulting in visual acuity recovery and alleviation of neurological symptoms. In the literature review, the combination of optic neuritis and hypophysitis, mostly with diabetes insipidus, was reported in 15 patients as case reports before the years of COVID-19 pandemic. The COVID-19 vaccination would trigger the onset of hypophysitis and optic neuritis in this patient.

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“…Supporting evidence includes its frequent occurrence during the peripartum period, its histopathological appearance, and the presence of circulating pituitary autoantibodies or coexisting autoimmune conditions (Hashimoto's thyroiditis most commonly, Graves' disease, subacute thyroiditis, adrenalitis, pernicious anemia, Type 1 diabetes mellitus, celiac disease, and vitiligo, to name a few) [1][2][3]. Very few cases of LYH have been reported previously in association with ON, and each of those patients presented with CDI initially, was diagnosed with a subtype of LYH, and later developed ON [4][5][6]. There is one case report of a middle-aged man with LIPH presenting with multiple progressive symptoms who initially had unilateral blurry vision four months prior that spontaneously resolved in a few weeks, and while the publication appears to attribute the patient's visual symptoms to optic chiasmal involvement as seen on MRI, these images were not obtained until months after his vision returned [7].…”

Section: Discussionmentioning

confidence: 99%

Lymphocytic hypophysitis in a patient presenting with sequential episodes of optic neuritis

et al. 2010

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A 41-year-old man presented with left optic neuritis (ON) without evidence of other autoimmune disease or hormonal imbalance. MRI showed enlargement of the left optic nerve but no sellar lesion. The patient recovered after steroid therapy but later developed right ON and required treatment again. Follow-up MRI revealed an ill-defined, enlarging sellar lesion with enhancement extending into the right cavernous sinus, and the patient developed symptoms of fatigue and loss of libido. Hormonal studies revealed hypogonadism and hypocortisolism. All laboratory investigation for autoimmune and infectious diseases remained negative. A transsphenoidal biopsy of the lesion revealed lymphocytic hypophysitis. The concomitant development of lymphocytic hypophysitis and optic neuritis suggests a common and likely autoimmune etiology. Visual loss in patients with LYH can sometimes be due to ON rather than compression of the optic apparatus, with significant implications for treatment strategies.

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Lymphocytic infundibulo-neurohypophysitis: An unusual cause of recurrent optic neuropathy in a child

Cited by 13 publications

References 9 publications

How reliably can autoimmune hypophysitis be diagnosed without pituitary biopsy

How reliably can autoimmune hypophysitis be diagnosed without pituitary biopsy

Bilateral Optic Neuritis and Hypophysitis With Diabetes Insipidus 1 Month After COVID-19 mRNA Vaccine: Case Report and Literature Review

Lymphocytic hypophysitis in a patient presenting with sequential episodes of optic neuritis

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