A case of lymphocytic panhypophysitis (LPH) during pregnancy

Arai, Yasuyuki; Nabe, Koichiro; Ikeda, Hiroki; Honjo, Sachiko; Wada, Yoshiharu; Hamamoto, Yoshiyuki; Nomura, Kazuhiro; Aoki, Tomokazu; Sendo, Toshiaki; Koshiyama, Hiroyuki

doi:10.1007/s12020-007-9001-6

Cited by 12 publications

(5 citation statements)

References 21 publications

(29 reference statements)

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“…The clinical characteristics of subjects are shown in Table 1, which include three cases previously reported by us; case 1,5 case 2,9 and case 3 10. All of these five cases were female, aged 20 to 77 years.…”

Section: Methodsmentioning

confidence: 99%

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Lymphocytic Panhypophysitis: Its Clinical Features in Japanese Cases

Wada

Hamamoto²,

Nakamura

et al. 2011

Jpn Clin Med

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Lymphocytic hypophysitis is divided into three forms according to the involved tissues, lymphocytic adenohypophysitis, lymphocytic infundibulo-neurohypophysitis, and lymphocytic panhypophysitis (LPH). The term LPH was first proposed by us in 1995, although its entity and pathogenesis still remain controversial. Here we report five cases of LPH, who visited our clinics during 1994 to 2009. All cases were female of 20 to 77 years of age, and one case was associated with pregnancy. They presented with polyuria (n = 4), headache (n = 3), general malaise, polydipsia (n = 2), blunted vision, diplopia, amenorrhea or appetite loss (n = 1). Magnetic resonance imaging showed the pituitary swelling, the thickened stalk, the loss of the T1 hyperintense neurohypophysis (n = 4), or the atrophic pituitary (n = 1). Endocrinological examinations revealed deficiencies of TSH, ADH in all cases, GH, ACTH in three cases, LH, PRL in two cases, and FSH in one case, respectively. The severity of ADH deficiency varied among the cases. Anti-pituitary antibody was not detected in the cases examined. The biopsy of the pituitary lesions was performed except for one case, all of which revealed the diffuse lymphocytic infiltration. These results suggest that LPH is characterized by the female predominance, the atypical patterns of anterior pituitary hormone deficiencies and the variable degrees of diabetes insipidus in Japanese.

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Section: Methodsmentioning

confidence: 99%

“…Here, in order to clarify the characteristics of LPH in Japanese, we report five cases of LPH, who visited our clinics during 1994 to 2009, including three cases previously reported by us. 5,9,10…”

Section: Introductionmentioning

confidence: 99%

Lymphocytic Panhypophysitis: Its Clinical Features in Japanese Cases

Wada

Hamamoto²,

Nakamura

et al. 2011

Jpn Clin Med

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“…If the urine osmolality is between 300 -800 mosmol/kg at the end of dehydration, either partial cranial DI or primary polydipsia (PP) is present. 8,9 In partial cranial DI the increase in urine osmolality after DDAVP is >9%. The increase in urine osmolality after DDAVP only <9% generally indicate PP.10 Based on these findings, our patient was diagnosed as having partial cranial DI.…”

Section: Discussionmentioning

confidence: 99%

Partial Empty Sella Syndrome Presenting with Panhypopituitarism and Masked Central Diabetes Insipidus

Wathurapatha

Dissanayeke

2022

J. Ruhunu Clin. Soc.

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Empty sella syndrome (ESS) is an uncommon cause of hypopituitarism. It is usually detected incidentally or with neurological symptoms whereas it is rare to present with symptomatic endocrine dysfunction. A 48-year old previously healthy man presented with generalized body weakness, lethargy, fatigue and postural dizziness of one month with no headache or visual disturbance. He also had features suggestive of hypogonadism and hypothyroidism. Panhypopitutarism secondary to partial ESS was diagnosed based on pituitary hormone testing and MRI brain. After starting glucocorticoid (GC) replacement patient developed polyuria and polydipsia and was diagnosed to have partial central diabetes insipidus (DI). He was successfully treated with hydrocortisone, thyroxin, intranasal vasopressin and testosterone. ESS causes significant pituitary dysfunction in considerable proportion of patients.

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“…Although the authors could find no reports of IgG4-related hypophysitis associated with pregnancy or the postpartum period in the literature, we noted 8 patients with pregnancy-associated hypophysitis and DI. Clinical features of these cases of pregnancyassociated DI related to lymphocytic hypophysitis are shown in Table 2 [59][60][61][62][63][64][65][66]. Among them, 4 cases developed lymphocytic hypophysitis in the postpartum period.…”

Section: Pregnancy-associated DI Due To Pituitary Injurymentioning

confidence: 99%

Pregnancy-associated diabetes insipidus in Japan—a review based on quoting from the literatures reported during the period from 1982 to 2019

et al. 2021

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This Review Article overviews the literature on diabetes insipidus (DI) associated with pregnancy and labor in Japan published from 1982 to 2019. The total number of patients collected was 361, however, only one-third of these cases had detailed pathophysiologic information enabling us to identify the respective etiology and subtype. Pregnancyassociated DI can be divided into 3 etiologies, central (neurogenic) DI, nephrogenic DI, and excess vasopressinaseassociated DI. Neurogenic DI has various causes: for example, DI associated with tumoral lesions in the pituitary and neighboring area, DI associated with Sheehan's syndrome and/or pituitary apoplexy, and DI associated with lymphocytic infundibuloneurohypophysitis (LINH, stalkitis). Nephrogenic DI results from defective response of the kidney to normal levels of vasopressin. However, the most interesting causal factor of pregnancy-associated DI is excess vasopressinase, caused either by excess production of vasopressinase by the placenta or defective clearance of vasopressinase by the liver. Hepatic complications resulting in pregnancy-associated DI include acute fatty liver of pregnancy (AFLP) and HELLP syndrome (syndrome of hemolysis, elevated liver enzymes, low platelets), as well as pre-existing or co-incidental hepatic diseases. A possible role of glucose uptake in putative stress-induced DI and the importance of correct diagnosis and treatment of pregnancy-associated DI, including use of 1-deamino 8-D arginine vasopressin, are also discussed.

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A case of lymphocytic panhypophysitis (LPH) during pregnancy

Cited by 12 publications

References 21 publications

Lymphocytic Panhypophysitis: Its Clinical Features in Japanese Cases

Lymphocytic Panhypophysitis: Its Clinical Features in Japanese Cases

Partial Empty Sella Syndrome Presenting with Panhypopituitarism and Masked Central Diabetes Insipidus

Pregnancy-associated diabetes insipidus in Japan—a review based on quoting from the literatures reported during the period from 1982 to 2019

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