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Article ; Online: Subcutaneous Infusion of rhPTH

Shulman, Dorothy

Journal of the Endocrine Society

2022 Volume 6, Issue 5, Page(s) bvac031

Abstract: We report a successful pregnancy in a young woman with autosomal dominant hypoparathyroidism type 1 (ADH1) due to an activating mutation of the calcium sensing receptor (CASR) (c.2519C>T; p.Ala840Val) who was treated with recombinant human parathyroid ... ...

Abstract	We report a successful pregnancy in a young woman with autosomal dominant hypoparathyroidism type 1 (ADH1) due to an activating mutation of the calcium sensing receptor (CASR) (c.2519C>T; p.Ala840Val) who was treated with recombinant human parathyroid hormone (rhPTH)
Language	English
Publishing date	2022-02-25
Publishing country	United States
Document type	Case Reports
ISSN	2472-1972
ISSN (online)	2472-1972
DOI	10.1210/jendso/bvac031
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Article ; Online: Recombinant human parathyroid hormone (1-84) is effective in CASR-associated hypoparathyroidism.

Hawkes, Colin Patrick / Shulman, Dorothy I / Levine, Michael A

European journal of endocrinology

2020 Volume 183, Issue 6, Page(s) K13–K21

Abstract: Introduction: Gain-of-function mutations in the CASR gene cause Autosomal Dominant Hypocalcemia Type 1 (ADH1), the most common genetic cause of isolated hypoparathyroidism. Subjects have increased calcium sensitivity in the renal tubule, leading to ... ...

Abstract	Introduction: Gain-of-function mutations in the CASR gene cause Autosomal Dominant Hypocalcemia Type 1 (ADH1), the most common genetic cause of isolated hypoparathyroidism. Subjects have increased calcium sensitivity in the renal tubule, leading to increased urinary calcium excretion, nephrocalcinosis and nephrolithiasis when compared with other causes of hypoparathyroidism. The traditional approach to treatment includes activated vitamin D but this further increases urinary calcium excretion. Methods: In this case series, we describe the use of recombinant human parathyroid hormone (rhPTH)1-84 to treat subjects with ADH1, with improved control of serum and urinary calcium levels. Results: We describe two children and one adult with ADH1 due to heterozygous CASR mutations who were treated with rhPTH(1-84). Case 1 was a 9.4-year-old female whose 24-h urinary calcium decreased from 7.5 to 3.9 mg/kg at 1 year. Calcitriol and calcium supplementation were discontinued after titration of rhPTH(1-84). Case 2 was a 9.5-year-old male whose 24-h urinary calcium decreased from 11.7 to 1.7 mg/kg at 1 year, and calcitriol was also discontinued. Case 3 was a 24-year-old female whose treatment was switched from multi-dose teriparatide to daily rhPTH(1-84). All three subjects achieved or maintained target serum levels of calcium and normal or improved urinary calcium levels with daily rhPTH(1-84) monotherapy. Conclusions: We have described three subjects with ADH1 who were treated effectively with rhPTH(1-84). In all cases, hypercalciuria improved by comparison to treatment with conventional therapy consisting of calcium supplementation and calcitriol.
MeSH term(s)	Child ; Female ; Humans ; Hypoparathyroidism/diagnosis ; Hypoparathyroidism/drug therapy ; Hypoparathyroidism/genetics ; Male ; Parathyroid Hormone/administration & dosage ; Receptors, Calcium-Sensing/genetics ; Recombinant Proteins/administration & dosage ; Treatment Outcome ; Young Adult
Chemical Substances	CASR protein, human ; PTH protein, human ; Parathyroid Hormone ; Receptors, Calcium-Sensing ; Recombinant Proteins
Language	English
Publishing date	2020-10-14
Publishing country	England
Document type	Case Reports ; Journal Article
ZDB-ID	1183856-5
ISSN	1479-683X ; 0804-4643
ISSN (online)	1479-683X
ISSN	0804-4643
DOI	10.1530/EJE-20-0710
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Article: Hyperparathyroidism two years after radioactive iodine therapy in an adolescent male.

Gomez, Danielle L / Shulman, Dorothy I

Case reports in pediatrics

2014 Volume 2014, Page(s) 163848

Abstract: Primary hyperparathyroidism is a very rare complication following radioactive iodine therapy. There is typically a latency period of more than a decade following radiation exposure and, therefore, it is observed almost exclusively in adults. Consequently, ...

Abstract	Primary hyperparathyroidism is a very rare complication following radioactive iodine therapy. There is typically a latency period of more than a decade following radiation exposure and, therefore, it is observed almost exclusively in adults. Consequently, pediatricians are not aware of the association. We present a case of primary hyperparathyroidism due to a solitary parathyroid adenoma occurring in an adolescent male two years following radioactive iodine treatment for papillary thyroid carcinoma. Periodic screening of serum calcium following ablative doses of radioactive iodine for thyroid cancer may be justified even in adolescents.
Language	English
Publishing date	2014-01-30
Publishing country	United States
Document type	Journal Article
ZDB-ID	2659094-3
ISSN	2090-6811 ; 2090-6803
ISSN (online)	2090-6811
ISSN	2090-6803
DOI	10.1155/2014/163848
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Article ; Online: Glycemic Outcomes During Early Use of the MiniMed™ 780G Advanced Hybrid Closed-Loop System with Guardian™ 4 Sensor.

Diabetes technology & therapeutics

2023 Volume 25, Issue 9, Page(s) 652–658

Abstract: Background: ...

Abstract	Background:
MeSH term(s)	Adolescent ; Adult ; Child ; Humans ; Blood Glucose ; Blood Glucose Self-Monitoring ; Diabetes Mellitus, Type 1/drug therapy ; Glucose ; Glycated Hemoglobin ; Hypoglycemic Agents/therapeutic use ; Insulin/therapeutic use ; Insulin Infusion Systems
Chemical Substances	Blood Glucose ; Glucose (IY9XDZ35W2) ; Glycated Hemoglobin ; Hypoglycemic Agents ; Insulin
Language	English
Publishing date	2023-06-16
Publishing country	United States
Document type	Clinical Trial ; Journal Article ; Research Support, Non-U.S. Gov't
ZDB-ID	1452816-2
ISSN	1557-8593 ; 1520-9156
ISSN (online)	1557-8593
ISSN	1520-9156
DOI	10.1089/dia.2023.0123
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Article ; Online: Poor head growth as a presenting sign of a cortisol-secreting adrenal adenoma in a 2-year-old boy.

Farooqi, Sonia / Sarangarajan, Sruthi / Shulman, Dorothy I

The Journal of pediatrics

2015 Volume 166, Issue 3, Page(s) 764–766

Abstract: Typical signs of glucocorticoid excess in children are weight gain and poor linear growth. We describe a 2-year-old boy with a cortisol-secreting adenoma who presented with a dramatic decline in head growth. This case underscores concern of adverse ... ...

Abstract	Typical signs of glucocorticoid excess in children are weight gain and poor linear growth. We describe a 2-year-old boy with a cortisol-secreting adenoma who presented with a dramatic decline in head growth. This case underscores concern of adverse effects of excess glucocorticoid on brain growth in very young children.
MeSH term(s)	Adenoma/blood ; Adenoma/diagnosis ; Adrenal Gland Neoplasms/blood ; Adrenal Gland Neoplasms/diagnosis ; Cephalometry ; Child, Preschool ; Diagnosis, Differential ; Head/growth & development ; Humans ; Hydrocortisone/blood ; Hydrocortisone/secretion ; Male
Chemical Substances	Hydrocortisone (WI4X0X7BPJ)
Language	English
Publishing date	2015-03
Publishing country	United States
Document type	Case Reports ; Journal Article
ZDB-ID	3102-1
ISSN	1097-6833 ; 0022-3476
ISSN (online)	1097-6833
ISSN	0022-3476
DOI	10.1016/j.jpeds.2014.11.040
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Article ; Online: Glycemic outcomes of children 2-6 years of age with type 1 diabetes during the pediatric MiniMed™ 670G system trial.

Forlenza, Gregory P / Ekhlaspour, Laya / DiMeglio, Linda A / Fox, Larry A / Rodriguez, Henry / Shulman, Dorothy I / Kaiserman, Kevin B / Liljenquist, David R / Shin, John / Lee, Scott W / Buckingham, Bruce A

Pediatric diabetes

2022 Volume 23, Issue 3, Page(s) 324–329

Abstract: Background: Highly variable insulin sensitivity, susceptibility to hypoglycemia and inability to effectively communicate hypoglycemic symptoms pose significant challenges for young children with type 1 diabetes (T1D). Herein, outcomes during clinical ... ...

Abstract	Background: Highly variable insulin sensitivity, susceptibility to hypoglycemia and inability to effectively communicate hypoglycemic symptoms pose significant challenges for young children with type 1 diabetes (T1D). Herein, outcomes during clinical MiniMed™ 670G system use were evaluated in children aged 2-6 years with T1D. Methods: Participants (N = 46, aged 4.6 ± 1.4 years) at seven investigational centers used the MiniMed™ 670G system in Manual Mode during a two-week run-in period followed by Auto Mode during a three-month study phase. Safety events, mean A1C, sensor glucose (SG), and percentage of time spent in (TIR, 70-180 mg/dl), below (TBR, <70 mg/dl) and above (TAR, >180 mg/dl) range were assessed for the run-in and study phase and compared using a paired t-test or Wilcoxon signed-rank test. Results: From run-in to end of study (median 87.1% time in auto mode), mean A1C and SG changed from 8.0 ± 0.9% to 7.5 ± 0.6% (p < 0.001) and from 173 ± 24 to 161 ± 16 mg/dl (p < 0.001), respectively. Overall TIR increased from 55.7 ± 13.4% to 63.8 ± 9.4% (p < 0.001), while TBR and TAR decreased from 3.3 ± 2.5% to 3.2 ± 1.6% (p = 0.996) and 41.0 ± 14.7% to 33.0 ± 9.9% (p < 0.001), respectively. Overnight TBR remained unchanged and TAR was further improved 12:00 am-6:00 am. Throughout the study phase, there were no episodes of severe hypoglycemia or diabetic ketoacidosis (DKA) and no serious adverse device-related events. Conclusions: At-home MiniMed™ 670G Auto Mode use by young children safely improved glycemic outcomes compared to two-week open-loop Manual Mode use. The improvements are similar to those observed in older children, adolescents and adults with T1D using the same system for the same duration of time.
MeSH term(s)	Blood Glucose ; Blood Glucose Self-Monitoring ; Child ; Child, Preschool ; Diabetes Mellitus, Type 1/drug therapy ; Humans ; Hypoglycemic Agents/administration & dosage ; Insulin/administration & dosage ; Insulin Infusion Systems/adverse effects
Chemical Substances	Blood Glucose ; Hypoglycemic Agents ; Insulin
Language	English
Publishing date	2022-01-31
Publishing country	Denmark
Document type	Clinical Trial ; Journal Article ; Research Support, Non-U.S. Gov't
ZDB-ID	1502504-4
ISSN	1399-5448 ; 1745-1426 ; 1399-543X
ISSN (online)	1399-5448
ISSN	1745-1426 ; 1399-543X
DOI	10.1111/pedi.13312
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Zs.A 5422: Show issues

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Article: Is there "seasonal" variation in height velocity in children treated with growth hormone? Data from the National Cooperative Growth Study.

Shulman, Dorothy I / Frane, James / Lippe, Barbara

International journal of pediatric endocrinology

2013 Volume 2013, Issue 1, Page(s) 2

Language	English
Publishing date	2013-02-02
Publishing country	England
Document type	Journal Article
ZDB-ID	2528691-2
ISSN	1687-9856 ; 1687-9848
ISSN (online)	1687-9856
ISSN	1687-9848
DOI	10.1186/1687-9856-2013-2
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Article ; Online: Progressive central puberty in a toddler with partial androgen insensitivity.

Dougan, Grace C / Uli, Naveen / Shulman, Dorothy I

The Journal of pediatrics

2014 Volume 164, Issue 3, Page(s) 655–657

Abstract: A male infant was diagnosed with partial androgen insensitivity caused by a novel mutation in the androgen receptor. At 3.5 months of age, he received 100 mg of testosterone intramuscularly over the course of 3 months to increase phallic size. He ... ...

Abstract	A male infant was diagnosed with partial androgen insensitivity caused by a novel mutation in the androgen receptor. At 3.5 months of age, he received 100 mg of testosterone intramuscularly over the course of 3 months to increase phallic size. He developed pubic hair after 5 months and signs of progressive central precocious puberty when re-examined at 17.5 months, which subsequently was suppressed with depot leuprolide.
MeSH term(s)	Aggression/drug effects ; Amino Acid Substitution ; Androgen-Insensitivity Syndrome/diagnosis ; Androgen-Insensitivity Syndrome/drug therapy ; Androgen-Insensitivity Syndrome/genetics ; Androgens/administration & dosage ; Androgens/adverse effects ; Delayed-Action Preparations ; Exons ; Gonadotropin-Releasing Hormone/agonists ; Hemizygote ; Humans ; Hypospadias/etiology ; Infant, Newborn ; Leuprolide/therapeutic use ; Luteinizing Hormone/blood ; Male ; Mutation ; Penis/abnormalities ; Puberty, Precocious/chemically induced ; Receptors, Androgen/genetics ; Scrotum/abnormalities ; Testosterone/administration & dosage ; Testosterone/adverse effects ; Testosterone/analogs & derivatives ; Testosterone/blood
Chemical Substances	Androgens ; Delayed-Action Preparations ; Receptors, Androgen ; Gonadotropin-Releasing Hormone (33515-09-2) ; Testosterone (3XMK78S47O) ; testosterone enanthate (7Z6522T8N9) ; Luteinizing Hormone (9002-67-9) ; Leuprolide (EFY6W0M8TG)
Language	English
Publishing date	2014-03
Publishing country	United States
Document type	Case Reports ; Journal Article
ZDB-ID	3102-1
ISSN	1097-6833 ; 0022-3476
ISSN (online)	1097-6833
ISSN	0022-3476
DOI	10.1016/j.jpeds.2013.11.020
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Article: Metabolic effects of growth hormone in the child and adolescent.

Shulman, Dorothy I

Current opinion in pediatrics

2002 Volume 14, Issue 4, Page(s) 432–436

Abstract: During the year 2000, several original studies were published regarding the metabolic effects of growth hormone therapy in pediatric patients. Pharmacologic doses of growth hormone were rarely associated with abnormalities in glucose tolerance in ... ...

Abstract	During the year 2000, several original studies were published regarding the metabolic effects of growth hormone therapy in pediatric patients. Pharmacologic doses of growth hormone were rarely associated with abnormalities in glucose tolerance in children with intrauterine growth retardation and Turner syndrome; however, serum insulin levels were elevated. A report from the Pharmacia International Growth Study database suggested a possible increase in type 2 diabetes in growth hormone-treated patients, indicating the need for continued surveillance for this condition. Growth hormone therapy increased markers of bone turnover and bone mineral density in children with chronic renal failure and Prader-Willi syndrome. In Prader-Willi syndrome, 2 years of growth hormone therapy also induced a sustained decrease in body fat, improvement in strength and physical skills, and increased lean body mass. Serum leptin, a reflection of body fat, declined with growth hormone therapy in a dose-dependent manner in intrauterine growth retardation children; the magnitude of the decline correlated with linear growth response. Skin is a target organ for growth hormone in children; growth hormone increased dermal thickness and reduced skin stiffness in growth hormone-deficient children. Reassuring data were published regarding the risk of tumor recurrence and mortality in children with brain tumors treated with growth hormone. Growth hormone administered to short children prior to kidney transplantation did not have adverse effects on subsequent graft survival or number of rejection episodes.
MeSH term(s)	Adolescent ; Age Factors ; Child ; Child Development/drug effects ; Human Growth Hormone/adverse effects ; Human Growth Hormone/metabolism ; Human Growth Hormone/pharmacology ; Humans
Chemical Substances	Human Growth Hormone (12629-01-5)
Language	English
Publishing date	2002-08
Publishing country	United States
Document type	Journal Article ; Review
ZDB-ID	1049374-8
ISSN	1531-698X ; 1040-8703
ISSN (online)	1531-698X
ISSN	1040-8703
DOI	10.1097/00008480-200208000-00014
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Article ; Online: Safety and Glycemic Outcomes During the MiniMed

Pihoker, Catherine / Shulman, Dorothy I / Forlenza, Gregory P / Kaiserman, Kevin B / Sherr, Jennifer L / Thrasher, James R / Buckingham, Bruce A / Kipnes, Mark S / Bode, Bruce W / Carlson, Anders L / Lee, Scott W / Latif, Kashif / Liljenquist, David R / Slover, Robert H / Dai, Zheng / Niu, Fang / Shin, John / Jonkers, Richard A M / Roy, Anirban /

Grosman, Benyamin / Vella, Melissa / Cordero, Toni L / McVean, Jennifer / Rhinehart, Andrew S / Vigersky, Robert A

Diabetes technology & therapeutics

2023 Volume 25, Issue 11, Page(s) 755–764

Abstract: Background: ...

Abstract	Background:
MeSH term(s)	Adolescent ; Adult ; Child ; Humans ; Blood Glucose ; Blood Glucose Self-Monitoring ; Diabetes Mellitus, Type 1/drug therapy ; Diabetes Mellitus, Type 1/complications ; Diabetic Ketoacidosis/etiology ; Glucose ; Glycated Hemoglobin ; Hypoglycemia/chemically induced ; Hypoglycemia/prevention & control ; Hypoglycemia/complications ; Hypoglycemic Agents/therapeutic use ; Insulin/therapeutic use ; Insulin Infusion Systems ; Treatment Outcome
Chemical Substances	Blood Glucose ; Glucose (IY9XDZ35W2) ; Glycated Hemoglobin ; Hypoglycemic Agents ; Insulin
Language	English
Publishing date	2023-10-25
Publishing country	United States
Document type	Clinical Trial ; Journal Article ; Research Support, Non-U.S. Gov't
ZDB-ID	1452816-2
ISSN	1557-8593 ; 1520-9156
ISSN (online)	1557-8593
ISSN	1520-9156
DOI	10.1089/dia.2023.0255
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