A Family Case of Nephrogenic Diabetes Insipidus

OKAYASU, T., SHIGIHARA, K., KOBAYASHI, N., ISHIKAWA, A., FUKUSHIMA, N., TAKASE, A., HATTORI, S., NAKAJIMA, T., SHISHIDO, T. and AGATSUMA, Y. A Family Case of Nephroqenic Diabetes Insipidus. Tohoku J. Exp. Med., 1990, 162 (2), 137-145 - Two brothers, patient 1 with fever and vomiting, and patient 2 w...

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Veröffentlicht in:The Tohoku Journal of Experimental Medicine 1990, Vol.162(2), pp.137-145
Hauptverfasser: OKAYASU, TAKAKO, SHIGIHARA, KAZUE, KOBAYASHI, NORIO, ISHIKAWA, AKASHI, FUKUSHIMA, NAOKI, TAKASE, AIKO, HATTORI, SATOSHI, NAKAJIMA, TAKEO, SHISHIDO, TETSUO, AGATSUMA, YOSHINORI
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container_end_page 145
container_issue 2
container_start_page 137
container_title The Tohoku Journal of Experimental Medicine
container_volume 162
creator OKAYASU, TAKAKO
SHIGIHARA, KAZUE
KOBAYASHI, NORIO
ISHIKAWA, AKASHI
FUKUSHIMA, NAOKI
TAKASE, AIKO
HATTORI, SATOSHI
NAKAJIMA, TAKEO
SHISHIDO, TETSUO
AGATSUMA, YOSHINORI
description OKAYASU, T., SHIGIHARA, K., KOBAYASHI, N., ISHIKAWA, A., FUKUSHIMA, N., TAKASE, A., HATTORI, S., NAKAJIMA, T., SHISHIDO, T. and AGATSUMA, Y. A Family Case of Nephroqenic Diabetes Insipidus. Tohoku J. Exp. Med., 1990, 162 (2), 137-145 - Two brothers, patient 1 with fever and vomiting, and patient 2 with failure to gain weight were studied. After 4hr of water deprivation test. the urinary osmolality of the patient 1 was only 105 mOsm/liter and his body weight showed a 4.6% reduction. In response to desamino-8-D arginine vasopressin intranasal administration, no significant elevation of urinary osmolality of patient 1 occurred. After low dose vasopressin tests, the maximal urinary osmolality of their father was in the normal range, but that of their mother was below the normal range. Moreover, the patients showed no significant increase of urinary osmolality after the same tests. The brothers were diagnosed as nephrogenic diabetes insipidus (NDI) and their mother was diagnosed as a carrier. An early diagnosis of NDI is important, since adequate managements such as low-solute diet with restricted protein and salt intake or such as water intake at frequent intervals can prevent the hyperosmolality which would develop the delayed mental and physical developments. The usefulness of the combination of indomethacin with thiazide diuretics is described.
doi_str_mv 10.1620/tjem.162.137
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A Family Case of Nephroqenic Diabetes Insipidus. Tohoku J. Exp. Med., 1990, 162 (2), 137-145 - Two brothers, patient 1 with fever and vomiting, and patient 2 with failure to gain weight were studied. After 4hr of water deprivation test. the urinary osmolality of the patient 1 was only 105 mOsm/liter and his body weight showed a 4.6% reduction. In response to desamino-8-D arginine vasopressin intranasal administration, no significant elevation of urinary osmolality of patient 1 occurred. After low dose vasopressin tests, the maximal urinary osmolality of their father was in the normal range, but that of their mother was below the normal range. Moreover, the patients showed no significant increase of urinary osmolality after the same tests. The brothers were diagnosed as nephrogenic diabetes insipidus (NDI) and their mother was diagnosed as a carrier. An early diagnosis of NDI is important, since adequate managements such as low-solute diet with restricted protein and salt intake or such as water intake at frequent intervals can prevent the hyperosmolality which would develop the delayed mental and physical developments. 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subjects Adult
antidiuretic hormone (ADH)
chlorothiazide
cyclic AMP
Deamino Arginine Vasopressin
diabetes insipidus (DI)
Diabetes Insipidus - genetics
Diabetes Insipidus - physiopathology
Female
Follow-Up Studies
Humans
Infant
Infant, Newborn
Kidney Diseases - genetics
Kidney Diseases - physiopathology
Kidney Diseases - therapy
Male
Osmolar Concentration
Pedigree
Urine - chemistry
vasopressin (AVP)
Water Deprivation
title A Family Case of Nephrogenic Diabetes Insipidus
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