Arch Dis Child: first published as 10.1136/adc.60.8.763 on 1 August 1985. Downloaded from

Alco(hOI intoxication, an lunderdiagnosed problem? 763 was 7t) mmol/l. The following morning she admitted greater than 85 mmol/l may be fatal.4 It is uncertain, to having drunk a cocktail of alcoholic beveratges however, whether these adult blood concentrations before the episode. have the same clinical importance in children, where age related variations in alcohol susceptibility and Case 4. A 12 year old girl was admitted with a three metabolic clearance may occur.?5 hour history of vomiting followed by loss of con- In our patients alcoholic beverages were the most sciousness. The possibility of alcohol or drug inges- commonly identified source of ethanol. The Amer- tion was denied. She was drowsy but responded to ican Academy of Pediatrics Committee on Drugs, verbal commands. No alcohol related smell was however, has recently emphasised the potentially detected on her breath. Her axillary temperature toxic concentrations of ethanol in some paediatric was 35-1°C. A toxicology screen was negative. Her pharmaceutical preparations.6 In the United Kingdom serum alcohol concentration was 44-5 mmol/l aind alcohol is used similarly in many paediatric prepara- plasma glucose was 5-7 mmol/l. After the laboratory tions-for example paracetamol elixir BP contains results were available she admitted to having drunk 10% v/v and chlorpheniramine elixir BP 6-3% v/v. a 'small gin and tonic' with friends. Although the harmful effect of ethanol in these concentrations remains to be established it could Discussion have an additive effect in fasting related hypo- glycaemia or cause problems in accidental ingestion Our experience shows the importance of considering if its presence went unrecognised. alcohol intoxication in childreni with unexplained drowsiness, hypoglycaemia. or hypothermia. The References Le Conte M. Saint-Jacques 1. Cagnat R, Vellicux F, Bois- mode of presentation may be confused with other siere 11. Le coma alcoolique par intoxication accidentelle chez conditions such as head in'ury or post-ictal state. It l'enfaint. Sewtl Hop Paris 1978X54:315-24. was characteristic that parents were often reluctatnt 2 Curry AS. Walker GW, Simpson GS. Determination of ethanol in blood by gas chromatography. Anaiyst 1966;91:742-3. to admit even the possibility of alcohol ingestioni by 3 who in MacLaircn NK, Valman HB, Levin B. Alcohol-induced hypo- copyright. their children, turn rarely volunteered the glycaemial in childhood. Br Med J 1970;1:278-80. information. Detection of an alcohol related smell 4 Johnson RA, Noll EC, Rodney WM. Survival after a serum on the breath was an unreliable sign and emphasises ethanol concentration of 1½/2%. Lancet 1982;ii:1394. the value of blood alcohol measurements. The 5 Ragan FA. Samuels MS, Hite SA. Ethanol ingestion in children. A five year review. JAMA 1979;242:2787-8. importance of diagnosis arises from the potentially Pruitt AW. Anyan WR. Hill RM, et al. American Academy of serious complication of alcohol induced hypo- Pediatrics Committee on Drugs. Ethanol in liquid preparations glycaemia' and the necessity to monitor blood intended for children. Pediatrics 1984;73:405-7.

glucose concentrations. In adults the blood alcohol http://adc.bmj.com/ Correspondcnce to Dr I A Auchterlonie, Raeden Centre, Mid- concentration is aLlso a guide to the severity of the stocket Roaid. Aberdeen AB2 4PE. intoxication, values over 30) mmol/l being associated with increasing confusion while concenitrations Received 13 March 1985

Hypertrichosis due to primary hypothyroidism on September 30, 2021 by guest. Protected

S R STERN AND C J H KELNAR Royal Hospital for Sick Children, Edinburgh

Hypertrichosis and hirsutism are common reasons SUMMARY A 10 year old girl with hvpertrichosis for seeking medical advice, particularly in young associated with primary hypothyroidism that re- women. Only a small proportion of subjects have a solved after 6 months' replacement treatment with diagnosable condition, and treatment is often un- thyroxine is reported. It is important to consider the satisfactory, especially in childhood.' Hypothyroid- diagnosis of hypothyroidism in children with ism is usually associated with loss of hair.2 We abnormal hairiness or distribution of body hair. report a 10 year old girl whose hypertrichosis was associated with primary hypothyroidism and re- Arch Dis Child: first published as 10.1136/adc.60.8.763 on 1 August 1985. Downloaded from

764 Archives of Disease in Childhood, 1985, 60 solved after replacement treatment with thyroxine been 108 cm (50th centile). Blood pressure was for six months. 90/25 mm Hg. Tendon reflexes were normal. Skin- fold thickness at triceps and subscapular sites were Case report 25th and 50th centile, respectively. A 10 year old girl was referred for assessment with a Results history of poor growth for about three years, with increasingly severe asthma and eczema, lethargy, Investigations showed normal diurnal cortisol, and growth of body hair over the previous six oestradiol, testosterone, prolactin, 17 a hydroxy months. Serum thyroxine concentrations found at progesterone, androstenedione, dehydroepiandro- the referring hospital were normal (110 nmol/l). sterone, and sex hormone binding globulin con- Thyroid stimulating hormone and thyroid antibodies centrations. The responses of growth hormone to were not measured. On examination she had dry insulin hypoglycaemia and gonadotrophins to lutei- skin with patchy excoriated eczema and cold ex- nising hormone releasing hormone were both tremities. Hypertrichosis was particularly noticeable normal. Basal thyroxine and thyroid stimulating over the lateral aspects of the limbs, the back, and hormone were <55 nmoUl and 511 mU/l respect- the forehead (Fig. 1) and was cosmetically disturb- ively. After treatment with thyrotrophin (200 ig ing to her and her family. She had Tanner stage 2 intravenously) thyroid stimulating hormone con- pubic hair with stage 1 breasts and axillary hair3 and centration rose further (>640 mU/l). Karyotype normal female external genitalia. Standing height was 46,XX. Bone age was 8-3 years at a chrono- was 130-5 cm (third to 10th centile; two standard logical age of 10-4 years. An x-ray film of the skull deviations below mean parental height) with normal suggested rounding of the sella turcica but a com- body proportions. Height at entry to school had puted tomogram of the head was normal. Full copyright. http://adc.bmj.com/ on September 30, 2021 by guest. Protected

Fig. 1 Distribution of hair at diagnosis. Arch Dis Child: first published as 10.1136/adc.60.8.763 on 1 August 1985. Downloaded from

Hypertrichosis due to primary hypothyroidism 765 copyright.

_ | J * _ _ __ | _ __

*tt i.'J_ | 1|1 W 1|_ __ ...... | | __A4|l Flg. 2 #Dlstrlbutlon e ESof .halr agSejzjaogzs;after six months' treatment with lthyroxine. l X blood count_ lshowed an eosinophilia (806x and of what he called http://adc.bmj.com/ l || 10'/I) hirsutism in dSP children, with _l l_ body radioallergosorbent_ elX#RIil |fitests__XMJyielded #1|_|strongly_I|_|1|positive __S%fi_ distributioneB of hair similar to ours, who responded results for | Abdominal __EN§ -Fo...... grass pollen. |s gultrasoundI| _to replacementS treatment (thyroid extract),4 con- showed...... 0%,rno .adrenal...... abnormality _ _...... but # l§AfcysticRRM | Benlarge- ___ _ l | __|-trasting A4EAASIA with the case reported by Maekawa et al in ment*.. :;.:of'.''..:.the left ..''.ovary. ::._: ;: |Y_ | !| ! ,g lNI 0II-_'°o.'0. | _ which an underlying abnormality of keratinisation After receiving appropriate replacement with was thought to lead to hair retention.5 The patho- thyroxine (0.1 mg/m2/24 hours) the girl grew at a physiology of the relation is unknown. The in- normal rate with appropriate advance in bone age. creased body hair in our patient was not generally in on September 30, 2021 by guest. Protected Thyroxine and thyroid stimulating hormone con- areas sensitive to androgen (hirsutism) despite some centrations became normal. She lost 4 kg in weight, pubic hair growth. Clearly, stimulation of the skinfold thicknesses reached the 10th centile, and growth of hair by hyperprolactinaemia mediated by performance at school and wellbeing improved thyroid releasing hormone, either directly or via dramatically. During six months her hypertrichosis adrenal androgens, was not the mechanism in our resolved progressively (Fig. 2). patient. Cystic ovarian enlargement in untreated hypo- Discussion thyroidism is well recognised.6 In most cases, as in ours, ovarian size returns to normal with treatment. It is unusual to find a cause for hypertrichosis or It is difficult to explain the normal plasma hirsutism and treatment is often unsatisfactory. 1 The thyroxine concentration obtained at the referring causal association between hypothyroidism and hospital, particularly in the absence of a simulta- hypertrichosis has not been reported in paediatric neous measurement of thyroid stimulating hor- studies, though congenitally hypothyroid infants mone. In view of the length of the history of often have a low hairline. Perloff reported four cases impairment of growth, compensated hypothyroid- Arch Dis Child: first published as 10.1136/adc.60.8.763 on 1 August 1985. Downloaded from

766 Archives of Disease in Childhood, 1985, 60 ism seems unlikely, and an error in handling or assay 2 Church RE. Br J Dermatol 1965;77:661. is more probable. Measurement of thryroid stimu- 3 Tanner JM. Growth at adolescence. 2nd ed. Oxford: Blackwell lating hormone might have enabled the Science Publications. 1962. diagnosis 4 Perloff WH. Hirsutism-a manifestation of juvenile hypo- considerably earlier. thyroidism. JAMA 1955;157:651-2. We suggest that full assessment of thyroid func- 5 Maekawa Y. Rolled hair and hypertrichosis-a manifestation of tion be carried out in all cases of hypertrichosis or juvenile hypothyroidism. J Dermatology (Tokyo) 1983;10: abnormal If 157-60. distribution of body hair. hypothyroid- 6 Lindsay AN, Voorhess ML, MacGillivray MH. Multicystic ism is noted and treated the loss of body hair may be ovaries in primary hypothyroidism. Obstet Gynecol 1983;61: dramatic and, to the patient, extremely rewarding. 433-7. Coirespondence and requests for reprints to Dr C J H Kelnar, References Royal Hospital for Sick Children, Edinburgh EH9 ILF. Ginsburg J, White MC. Hirsutism and virilization. Br Med J 1980;280:369-7 1. Received 5 March 1985

Congenital hypothyroidism with hereditary, raised thyroxine binding globulin

L N J ARCHER, B P O'MALLEY, AND P G F SWIFT Departments of Child Health and Pharmacology, University of Leicester and Departnment of Paediatrics, Leicester Royal Infirmary

umbilical hernia, and this was confirmed by a copyright. SUMMARY A boy with congenital hypothyroidism protein bound iodine value of 3 [tg/100 ml (normal and hereditary raised thyroxine binding globulin is range 4-0 to 8-4 Ftg/100 ml). Treatment with oral L- described. This hitherto unreported combination thyroxine (150 'tg daily) was begun at 14 weeks of resulted in under treatment of the thyroid deficiency age. He smiled at 3 months, sat unsupported at 8 until serum thyroid stimulating hormone measure- months, and walked at 20 months. At 22 months, ment became routinely available. Inadequate L- the family moved to a new area, at which time his

thyroxine replacement treatment between 2 and 7 height was at the 50th centile, three ossification http://adc.bmj.com/ years of age caused retarded bone maturation, poor centres had been reported on a wrist radiograph growth velocity, and probably added to his educa- (now unavailable), and the protein bound iodine tional difficulties. concentration was 12 ,tg/100 ml. Apart from his delay in walking, development was considered normal. In view of the high protein bound iodine Abnormalities in serum thyroxine binding globulin concentration, thyroxine treatment was gradually may be inherited or acquired,' and result in raised reduced. At 2½/2 years it was stopped completely for or depressed total thyroid hormone concentrations five weeks, whereupon the protein bound iodine fell on September 30, 2021 by guest. Protected even in euthyroid subjects. If true thyroid dysfunc- to 1-7 [tg/100 ml. L-thyroxine treatment (50 [ig orally tion occurs in the presence of thyroxine binding daily) was begun again, after which the protein globulin derangement, diagnosis and management bound iodine rose to 15-2 [tg/100 ml. Again his will be unsatisfactory unless the existence of both thyroxine treatment was reduced to 25 [tg daily and abnormalities is recognised. We report a patient he remained on this dose from age 2-8 years until with congenital hypothyroidism in association with 7-25 years. Over this period his height fell from the hereditary, raised thyroxine binding globulin, a 50th centile to the 3rd centile with a correspondingly combination not previously described. slow rate of increase. Radiological bone age was reported as 'within normal limits' at chronological Case history age 2 years, and was 6 years (Greulich and Pyle) at a chronological age of 8 years. He entered a normal A boy (birthweight 3180 g) was born at term in 1970. primary school, but required remedial teaching Perinatal and family medical histories were normal. because of slow progress. Although poor growth A clinical diagnosis of hypothyroidism was made at caused concern, no alteration was made to the 3 months of age when he presented with an thyroxine dose (25 [tg daily) as protein bound iodine