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A case of thyrotoxic periodic paralysis as initial manifestation of Graves' disease in a 16-year-old Korean adolescent.

Se Yong JUNG ; Kyung Chul SONG ; Jae Il SHIN ; Hyun Wook CHAE ; Ho Seong KIM ; Ah Reum KWON

Annals of Pediatric Endocrinology & Metabolism.2014;19(3):169-173. doi:10.6065/apem.2014.19.3.169

Thyrotoxic periodic paralysis (TPP) is a rare complication of hyperthyroidism, with recurrent muscle paralysis and hypokalemia that are caused by an intracellular shift of potassium. TPP is relatively common in Asian males, but is extremely rare in children and adolescents, even for those of Asian descent. We describe a 16-year-old Korean adolescent presenting with a two-week history of episodic leg weakness in the morning. He showed sinus tachycardia, lower leg weakness, and hypokalemia. Thyroid function test showed hyperthyroidism, and thyroid ultrasonography revealed a diffuse enlarged thyroid with increased vascularity, consistent with Graves' disease. He was treated with beta-adrenergic blocker and antithyroid drugs. He has been symptom free for one year, as his hyperthyroidism has been controlled well with antithyroid drugs. TPP should be considered in children and adolescents with acute paralysis of the lower extremities and hypokalemia.
Adolescent* ; Antithyroid Agents ; Asian Continental Ancestry Group ; Child ; Graves Disease* ; Humans ; Hyperthyroidism ; Hypokalemia ; Leg ; Lower Extremity ; Male ; Paralysis* ; Potassium ; Tachycardia, Sinus ; Thyroid Function Tests ; Thyroid Gland ; Ultrasonography

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Long-term follow-up on Cushing disease patient after transsphenoidal surgery.

Insook JEONG ; Moonyeon OH ; Ja Hye KIM ; Ja Hyang CHO ; Jin Ho CHOI ; Han Wook YOO

Annals of Pediatric Endocrinology & Metabolism.2014;19(3):164-168. doi:10.6065/apem.2014.19.3.164

Cushing disease is caused by excessive adrenocorticotropic hormone (ACTH) production by the pituitary adenoma. Transsphenoidal surgery is its first-line treatment. The incidence of Cushing disease in children and adolescents is so rare that long-term prognoses have yet to be made in most cases. We followed-up on a 16-year-old male Cushing disease patient who presented with rapid weight gain and growth retardation. The laboratory findings showed increased 24-hour urine free cortisol and lack of overnight cortisol suppression by low-dose dexamethasone test. The serum cortisol and 24-hour urine free cortisol, by high-dose dexamethasone test, also showed a lack of suppression, and a bilateral inferior petrosal sinus sampling suggested lateralization of ACTH secretion from the right-side pituitary gland. However, after a right hemihypophysectomy by the transsphenoidal approach, the 24-hour urine free cortisol levels were persistently high. Thus the patient underwent a total hypophysectomy, since which time he has been treated with hydrocortisone, levothyroxine, recombinant human growth hormone, and testosterone enanthate. Intravenous bisphosphonate for osteoporosis had been administered for three years. At his current age of 26 years, his final height had attained the target level range; his bone mineral density was normal, and his pubic hair was Tanner stage 4. This report describes the long-term treatment course of a Cushing disease patient according to growth profile, pubertal status, and responses to hormone replacement therapy. The clinical results serve to emphasize the importance of growth optimization, puberty, and bone health in the treatment management of Cushing disease patients who have undergone transsphenoidal surgery.
Adolescent ; Adrenocorticotropic Hormone ; Bone Density ; Child ; Dexamethasone ; Follow-Up Studies* ; Hair ; Hormone Replacement Therapy ; Human Growth Hormone ; Humans ; Hydrocortisone ; Hypophysectomy ; Hypopituitarism ; Incidence ; Male ; Osteoporosis ; Petrosal Sinus Sampling ; Pituitary ACTH Hypersecretion* ; Pituitary Gland ; Pituitary Neoplasms ; Prognosis ; Puberty ; Testosterone ; Thyroxine ; Weight Gain

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Prepubertal unilateral gynecomastia in the absence of endocrine abnormalities.

Min KANG ; Chan Jae LEE ; Il Tae HWANG ; Kwanseop LEE ; Min Jae KANG

Annals of Pediatric Endocrinology & Metabolism.2014;19(3):159-163. doi:10.6065/apem.2014.19.3.159

Gynecomastia is defined as the excessive development of breast tissue in males, which can occur as unilateral or bilateral. Although the overall prevalence of gynecomastia is 40%-65%, the prepubertal unilateral gynecomastia in the absence of endocrine abnormalities is unusual, with only a few case reports in literature. Idiopathic gynecomastia in prepuberty is benign but a diagnosis of exclusion. We here report two cases of healthy prepubertal boys aged 8.8 and 9.6 years old, respectively, presented with painful palpable mass in their unilateral breast. Breast tissue with glandular proliferation was confirmed on ultrasonography. Serum levels of estradiol, testosterone, and other laboratory findings were within normal range. They seem to have the prepubertal idiopathic gynecomastia but further follow-up to see their progression is needed.
Breast ; Child ; Diagnosis ; Estradiol ; Gynecomastia* ; Humans ; Male ; Prevalence ; Reference Values ; Testosterone ; Ultrasonography

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A 3-year-old girl with Graves' disease with literature review.

Yo Han HO ; Eun Cho CHUNG ; Sin Ae PARK

Annals of Pediatric Endocrinology & Metabolism.2014;19(3):154-158. doi:10.6065/apem.2014.19.3.154

Graves' disease, the main cause of hyperthyroidism in the pediatric age group, is very rare in children younger than 4 years old but can seriously interfere with growth and development if not recognized and treated. Here we report a case of a 3-year-old girl with Graves' disease who presented with goiter, exophthalmos, heat intolerance, and hyperactivity. At her first visit, her serum concentrations of triiodothyronine (T3) and free thyroxine (free T4) were normal, whereas that of thyroid-stimulating hormone (TSH) was decreased. Antimicrosomal antibody was 7,053.94 IU/mL, and TSH-binding inhibitory immunoglobulin was 31.62%. A thyroid scan showed diffuse enlargement with markedly increased uptake of both thyroid glands. Although T3 and free T4 levels were initially normal, she developed hyperthyroidism 3 months later. She was finally diagnosed with Graves' disease and treated with methimazole for 6 months. This is the first report of Graves' disease in children younger than 4 years old in Korea.
Child ; Child, Preschool* ; Exophthalmos ; Female ; Goiter ; Graves Disease* ; Growth and Development ; Hot Temperature ; Humans ; Hyperthyroidism ; Immunoglobulins ; Korea ; Methimazole ; Thyroid Gland ; Thyrotropin ; Thyroxine ; Triiodothyronine

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New Korean reference for birth weight by gestational age and sex: data from the Korean Statistical Information Service (2008-2012).

Jung Sub LIM ; Se Won LIM ; Ju Hyun AHN ; Bong Sub SONG ; Kye Shik SHIM ; Il Tae HWANG

Annals of Pediatric Endocrinology & Metabolism.2014;19(3):146-153. doi:10.6065/apem.2014.19.3.146

PURPOSE: To construct new Korean reference curves for birth weight by sex and gestational age using contemporary Korean birth weight data and to compare them with the Lubchenco and the 2010 United States (US) intrauterine growth curves. METHODS: Data of 2,336,727 newborns by the Korean Statistical Information Service (2008-2012) were used. Smoothed percentile curves were created by the Lambda Mu Sigma method using subsample of singleton. The new Korean reference curves were compared with the Lubchenco and the 2010 US intrauterine growth curves. RESULTS: Reference of the 3rd, 10th, 25th, 50th, 75th, 90th, and 97th percentiles birth weight by gestational age were made using 2,249,804 (male, 1,159,070) singleton newborns with gestational age 23-43 weeks. Separate birth weight curves were constructed for male and female. The Korean reference curves are similar to the 2010 US intrauterine growth curves. However, the cutoff values for small for gestational age (<10th percentile) of the new Korean curves differed from those of the Lubchenco curves for each gestational age. The Lubchenco curves underestimated the percentage of infants who were born small for gestational age. CONCLUSION: The new Korean reference curves for birth weight show a different pattern from the Lubchenco curves, which were made from white neonates more than 60 years ago. Further research on short-term and long-term health outcomes of small for gestational age babies based on the new Korean reference data is needed.
Asian Continental Ancestry Group ; Birth Weight* ; Female ; Gestational Age* ; Growth Charts ; Humans ; Infant ; Infant, Newborn ; Information Services* ; Male ; United States

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Earlier re-evaluation may be possible in pediatric patients with eutopic congenital hypothyroidism requiring lower L-thyroxine doses.

Min Sun CHO ; Gyung Sun CHO ; So Hyun PARK ; Min Ho JUNG ; Byung Kyu SUH ; Dae Gyun KOH

Annals of Pediatric Endocrinology & Metabolism.2014;19(3):141-145. doi:10.6065/apem.2014.19.3.141

PURPOSE: The incidence of congenital hypothyroidism (CH) has increased in several countries. Lower cut-off in screening programs have led to an increase in the proportion of transient hypothyroidism (TH) cases diagnosed, leading to debate on the associated clinical and economic impact. This study aimed to identify factors that would allow discrimination between TH and permanent CH (PH) in patients with a eutopic thyroid gland. METHODS: Sixty-six patients with CH from 3 different hospitals were studied: 26 cases of TH, and 40 cases of PH. Laboratory findings and clinical parameters were analysed in 56 patients with eutopic thyroid gland. RESULTS: Initial serum thyroid stimulating hormone levels and L-thyroxine dose at 12 and 24 months of age were significantly higher in PH than TH patients with a eutopic thyroid gland. The area under the curve for the 12-month and 24-month dose for the prediction of TH in eutopic CH was 0.799 (95% confidence interval [CI], 0.678-0.919; P<0.001) and 0.925 (95% CI, 0.837-1.000; P<0.001), respectively. The optimum 12-month and 24-month dose in predicting TH is 3.25 microg/kg (12-month: sensitivity, 87.1%; specificity, 68.0%; 24-month: sensitivity 93.5%, specificity 88%). CONCLUSION: Infants with CH requiring lower L-thyroxine doses (<3.25 microg/kg) are likely to have TH, and thus might be re-evaluated at 12 months or 24 months rather than 3 years of age.
Congenital Hypothyroidism* ; Discrimination (Psychology) ; Humans ; Hydrogen-Ion Concentration ; Hypothyroidism ; Incidence ; Infant ; Mass Screening ; Thyroid Gland ; Thyrotropin ; Thyroxine*

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Significant adverse reactions to long-acting gonadotropin-releasing hormone agonists for the treatment of central precocious puberty and early onset puberty.

Ji Woo LEE ; Hyung Jin KIM ; Yun Mee CHOE ; Hee Suk KANG ; Soon Ki KIM ; Yong Hoon JUN ; Ji Eun LEE

Annals of Pediatric Endocrinology & Metabolism.2014;19(3):135-140. doi:10.6065/apem.2014.19.3.135

PURPOSE: Long-acting gonadotropin-releasing hormone agonists (GnRHa) are commonly used to treat central precocious puberty (CPP) in Korea. Although rare, there have been reports on the characteristic of adverse reactions of GnRHa in CPP among the Korean population. This study was intended to report on our clinical experience regarding significant adverse reactions to long-acting GnRHa in CPP and early onset puberty and to evaluate the prevalence rate of serious side effects. METHODS: This retrospective study included children with CPP and early onset puberty, who were administered monthly with long-acting GnRHa (leuprolide acetate, triptorelin acetate) at the outpatient clinic of Department of Pediatrics, at Inha University Hospital, between January 2011 and December 2013. We analyzed the clinical characteristics of patients who experienced significant adverse reactions and evaluated the prevalence rate. RESULTS: Six serious side effects (0.9%) were observed among total of 621 CPP and early onset puberty children with GnRHa therapy. The number of sterile abscess formation was four in three patients (4 events of 621). Anaphylaxis occurred in only one patient, and unilateral slipped capital femoral epiphysis (SCFE) in another one patient. Anaphylaxis occurred after the 6th administration of the monthly depot triptorelin acetate. Unilateral SCFE developed in GnRHa therapy. CONCLUSION: Sterile abscess formation occurred in 0.6% of CPP and early onset puberty patients from the administration of a monthly depot GnRHa therapy. The occurrences of anaphylaxis and SCFE are extremely rare, but can have serious implications on patients. Clinicians should be aware of these potential adverse effects related to GnRHa therapy in CPP.
Abscess ; Adolescent ; Ambulatory Care Facilities ; Anaphylaxis ; Child ; Drug-Related Side Effects and Adverse Reactions ; Gonadotropin-Releasing Hormone* ; Humans ; Korea ; Leuprolide ; Pediatrics ; Prevalence ; Puberty* ; Puberty, Precocious* ; Retrospective Studies ; Slipped Capital Femoral Epiphyses ; Triptorelin Pamoate

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No influence of parental origin of intact X chromosome and/or Y chromosome sequences on three-year height response to growth hormone therapy in Turner syndrome.

Hye Jin LEE ; Hae Woon JUNG ; Gyung Min LEE ; Hwa Young KIM ; Jae Hyun KIM ; Sun Hee LEE ; Ji Hyun KIM ; Young Ah LEE ; Choong Ho SHIN ; Sei Won YANG

Annals of Pediatric Endocrinology & Metabolism.2014;19(3):127-134. doi:10.6065/apem.2014.19.3.127

PURPOSE: Whether parental origin of the intact X chromosome and/or the presence of Y chromosome sequences (Yseq) play a role in three-year height response to growth hormone (GH) were investigated. METHODS: Paternal (Xp) or maternal (Xm) origin of X chromosome was assessed by microsatellite marker analysis and the presence of hidden Yseq was analyzed. The first-, second-, and third-year GH response was measured as a change in height z-score (Z_Ht) in Turner syndrome (TS) patients with 45,Xp (n=10), 45,Xm (n=15), and 45,X/46,X,+mar(Y) (Xm_Yseq) (n=8). RESULTS: The mean baseline Z_Ht did not differ according to Xp or Xm origin, however the mean baseline Z_Ht was higher in the Xm_Yseq group than in Xm group, after adjusting for bone age delay and midparental Z_Ht (P=0.04). There was no difference in the height response to GH between the 3 groups. The height response to GH decreased progressively each year (P<0.001), such that the third-year increase in Z_Ht was not significant. This third-year decrease in treatment response was unaffected by Xp, Xm, and Xm_Yseq groups. Increasing GH dosage from the second to third-year of treatment positively correlated with the increase in Z_Ht (P=0.017). CONCLUSION: There was no evidence of X-linked imprinted genes and/or Yseq affecting height response to 3 years of GH therapy. Increasing GH dosages may help attenuate the decrease in third-year GH response in TS patients with 45,X and/or 46,X/+mar(Y).
Genomic Imprinting ; Growth Hormone* ; Humans ; Microsatellite Repeats ; Parents* ; Turner Syndrome* ; X Chromosome* ; Y Chromosome*

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The treatment of Graves' disease in children and adolescents.

Hae Sang LEE ; Jin Soon HWANG

Annals of Pediatric Endocrinology & Metabolism.2014;19(3):122-126. doi:10.6065/apem.2014.19.3.122

Graves' disease (GD) accounts for 10%-15% of thyroid disorders in children and adolescents. The use of antithyroid drugs as the initial treatment option in GD is well accepted. An average two years remission is achieved in about 30% of children treated with antithyroid drugs. However, the optimal treatment duration and the predictive marker of remission after antithyroid drug therapy are still controversial. Additionally, 131I therapy and surgery are considered the option for treatment in children and adolescents with GD. We review the treatment options for pediatric GD and the possible determinants of remission and relapse on antithyroid drug treatment in children and adolescents.
Adolescent* ; Antithyroid Agents ; Child* ; Drug Therapy ; Graves Disease* ; Humans ; Hyperthyroidism ; Recurrence ; Thyroid Gland

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Neonatal screening and a new cause of congenital central hypothyroidism.

Toshihiro TAJIMA ; Akie NAKAMURA ; Shuntaro MORIKAWA ; Katsura ISHIZU

Annals of Pediatric Endocrinology & Metabolism.2014;19(3):117-121. doi:10.6065/apem.2014.19.3.117

Congenital central hypothyroidism (C-CH) is a rare disease in which thyroid hormone deficiency is caused by insufficient thyrotropin (TSH) stimulation of a normally-located thyroid gland. Most patients with C-CH have low free thyroxine levels and inappropriately low or normal TSH levels, although a few have slightly elevated TSH levels. Autosomal recessive TSH deficiency and thyrotropin-releasing hormone receptor-inactivating mutations are known to be genetic causes of C-CH presenting in the absence of other syndromes. Recently, deficiency of the immunoglobulin superfamily member 1 (IGSF1) has also been demonstrated to cause C-CH. IGSF1 is a plasma membrane glycoprotein highly expressed in the pituitary. Its physiological role in humans remains unknown. IGSF1 deficiency causes TSH deficiency, leading to hypothyroidism. In addition, approximately 60% of patients also suffer a prolactin deficiency. Moreover, macroorchidism and delayed puberty are characteristic features. Thus, although the precise pathophysiology of IGSF1 deficiency is not established, IGSF1 is considered to be a new factor controlling growth and puberty in children.
Adolescent ; Cell Membrane ; Child ; Glycoproteins ; Humans ; Hypothyroidism* ; Immunoglobulins ; Infant, Newborn ; Neonatal Screening* ; Prolactin ; Puberty ; Puberty, Delayed ; Rare Diseases ; Thyroid Gland ; Thyrotropin ; Thyrotropin-Releasing Hormone ; Thyroxine

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