The association of diabetes insipidus and adipsia after craniopharyngioma surgery has high morbidity. Hypernatremia can be caused by adipsia and be aggravated by diabetes insipidus. Rhabdomyolysis rarely occurs. This is the first report of a diabetic patient with craniopharyngioma who developed diabetes insipidus and adipsia after surgery, evolving with severe hypernatremia that caused considerable rhabdomyolysis. The importance of the evaluation of muscle integrity when under hypernatremic states is pointed out. Although adipsia may have a simple solution through volunteer water ingestion, serious consequences such as repeated severe hypernatremia episodes and intense rhabdomyolysis with high morbidity could occur, if adipsia is not diagnosed.511175-

Baptista, Maria Tereza Matias

Garmes, Heraldo Mendes

Lima, Marcelo de Oliveira

Panzan, Anita Denardo

Zantut-Wittmann, Denise E

Arquivos Brasileiros de Endocrinologia & Metabologia

English

Repositorio da Producao Cientifica e Intelectual da Unicamp

1175Arq Bras Endocrinol Metab 2007;51/7
ABSTRACT
The association of diabetes insipidus and adipsia after craniopharyngioma
surgery has high morbidity. Hypernatremia can be caused by adipsia and
be aggravated by diabetes insipidus. Rhabdomyolysis rarely occurs. Case
report: This is the first report of a diabetic patient with craniopharyngioma
who developed diabetes insipidus and adipsia after surgery, evolving with
severe hypernatremia that caused considerable rhabdomyolysis. Conclu-
sion: The importance of the evaluation of muscle integrity when under
hypernatremic states is pointed out. Although adipsia may have a simple
solution through volunteer water ingestion, serious consequences such as
repeated severe hypernatremia episodes and intense rhabdomyolysis with
high morbidity could occur, if adipsia is not diagnosed. (Arq Bras
Endocrinol Metab 2007;51/7:1175-1179)
Keywords: Rhabdomyolysis; Adipsia; Hypernatremia; Craniopharyn-
gioma; Diabetes insipidus
RESUMO
Rabdomiólise Grave Devido a Hipernatremia Adípsica após Cirurgia
de Craniofaringioma.
A associação de diabetes insipidus e adipsia após cirurgia de craniofarin-
gioma implica em alta morbidade. Hipernatremia pode desenvolver-se
devido a adipsia e ser agravada por diabetes insipidus. Rabdomiólise rara-
mente ocorre. Descrição do caso: Esta é a primeira descrição de paci-
ente diabético com craniofaringioma que desenvolveu diabetes insipidus
e adipsia após a cirurgia, evoluindo com hipernatremia grave e conse-
qüente rabdomiólise maciça. Conclusão: Ressalta-se a necessidade de
avaliar a integridade muscular na vigência de estados hipernatrêmicos.
Apesar de apresentar solução simples, como ingestão voluntária de água,
pode haver sérias conseqüências se o diagnóstico de adipsia não é rea-
lizado, como episódios repetidos de hipernatremia grave com rabdo-
miólise intensa e elevada morbidade. (Arq Bras Endocrinol Metab
2007;51/7:1175-1179)
Descritores: Rabdomiólise; Adipsia; Hipernatremia; Craniofaringioma;
Diabetes insipidus
DIABETES INSIPIDUS CAN BE ASSOCIATED with craniopharyngioma, lead-ing to increased morbidity. Adipsia or hypodipsia rarely occurs as a
hypothalamic lesion. The association of these two osmolarity dysfunctions
causes a significantly higher morbidity (1). Hypernatremia can emerge
from adipsia and be aggravated by diabetes insipidus. Clinical manifesta-
tions include dehydration, muscular weakness, behavioural disturbances,
apresentação de caso
Severe Rhabdomyolysis Due to Adipsic Hypernatremia after
Craniopharyngioma Surgery
DENISE E. ZANTUT-WITTMANN
HERALDO MENDES GARMES
ANITA DENARDO PANZAN
MARCELO DE OLIVEIRA LIMA
MARIA TEREZA MATIAS BAPTISTA
Endocrinology Division, Faculty
of Medical Sciences, State 
University of Campinas 
(Unicamp), Campinas, SP.
Recebido em 08/02/07
Revisado em 18/05/07
Aceito em 06/08/07
delirium, lethargy, and coma. Rhabdomyolysis is a rare
occurrence in hyperosmolarity states, and can progress
with acute renal insufficiency and myoglobinuria.
Muscular weakness and pain are marked symptoms
and the diagnosis is confirmed through the elevation
of creatine phosphokinase (CK) (2).
We report herein a diabetic patient with cranio-
pharyngioma who developed diabetes insipidus and
adipsia, as a complication of surgical resection of a
suprasellar tumour, leading to a hypothalamic lesion,
presenting repeated dehydration episodes with severe
hypernatremia and at least one episode of massive
rhabdomyolysis.
CASE REPORT
Male, 24 years, began his follow up in August 1997,
presenting pubertal and growth delay, excessive
weight gain and headache. Height = 1.63 m; weight =
74 kg; pubertal stage G2P2 (Tanner); bone age = 14
years (chronological age = 17 years, 10 months).
He presented amaurosis of the right eye and
temporal hemianopsia of the left eye. Pituitary insuffi-
ciency was revealed in the functional evaluation,
except for the normal prolactin and thyrotropin
response and free thyroxin levels (table 1). Magnetic
resonance imaging (MRI) of sella turcica showed a
heterogeneous suprasellar expansive lesion, predomi-
nantly T1 hypointense and T2 hyperintense, with cys-
tic areas and calcifications, displacing the 3rd ventricle
and columns of the posterior fornix, pressing the optic
nerve and gyrating laterally the optic tracts, with no
sella turcica invasion (figure 1). Cerebral arteriography
was in accordance to a suprasellar expansive mass. Lab-
oratory findings were normal, except for hyper-
glycemia (113 to 114 mg/dL), leading to the diagno-
sis of diabetes mellitus. On November 1998, he was
submitted to a surgical transfrontal tumour resection.
The anatomopathological diagnosis was craniopharyn-
gioma. He developed diabetes insipidus and hyperna-
tremia (Na = 173 mEq/L; reference values – RV:
133–145). He was normokalemic (K= 3.4 mEq/L,
RV= 3.3–5.1). He started on intranasal Desmo-
pressin–DDAVP (30 µg/day). Pituitary function was
totally insufficient.
MRI (March/2002) showed gyrus rectus atro-
phy, slight hypothalamic irregularity, free optic nerves,
no identification of pituitary stalk, and no signs of
recurrence. Bone age = 15 years (chronological age =
19 years, 01 month); Visual fields: lower lateral quad-
rantopsia of the right eye and lateral hemianopsia of
the left eye.
He was admitted 6 times at the Emergence
Unit (January/1999 through August/2003), present-
ing weakness and muscular pain, dehydration, hyper-
natremia (158 to 173 mEq/L), hyperglycemia (126;
101; 116; 120; 224 mg/dL) and once with hyper-
glycemic ketosis (980 mg/dL, Na = 161 mEq/L, K =
4.3 mEq/L). He was always normokalemic. In Sep-
tember/2003, he referred to intense and generalized
muscular pain weakness for the past two days and
denied any thirst sensation. Laboratory findings: CK =
13,775 IU/L (RV < 170), Na = 165 mEq/L, K = 3.6
mEq/L, glucose = 224 mg/dL, Calcium (Ca) = 8.9
mg/dL (RV = 8.6–10.0), inorganic phosphorus (Pi) =
4.1 mg/dL (RV = 2.7–4.5), urea = 74 mg/dL (RV <
49), creatinine = 1.48 mg/dL (RV < 1.2). A CK peak
was observed 24 hours after admission (CK = 21,261
IU/L, Na = 171 mEq/L, K = 4.2 mEq/L), and an
acute non-oliguric renal insufficiency ensued as a com-
plication. Daily CK (figure 2), natremia, and renal
function were determined. The diagnosis was acute
myopathy secondary to adipsic hypernatremia as a
consequence of cranipharyngioma surgery. The treat-
ment was based on natremia correction with intra-
Rhabdomyolysis Due to Adipsic Hypernatremia
Zantut-Wittmann et al.
1176 Arq Bras Endocrinol Metab 2007;51/7
Table 1. Pituitary functional evaluation before craniopharyngioma surgery.
Time Glucose hGH Cortisol Prolactin TSH LH FSH
0’ 82 1.42 10.0 10.5 1.32 0.4 0.4
15’ 40 < 0.5 27.4 10.48 0.8 1.3
30’ 33 2.8 9.8 27.3 14.55 0.6 1.7
45’ 66 < 0.5 0.9 1.7
60’ 73 0.52 14.0 0.7 2.2
90’ 79 2.5
BRV < 100 mg% < 2.5 ng/mL 5.4–25 µg/dL < 15 ng/mL 0.41–4.15 1.7–8.6 1.5–12.4 
µIU/mL mIU/mL mIU/mL
FT4 = 0.86 ng% (BRV = 0.74–2.1)
BRV = Basal Reference Values; FT4 = Free Thyroxin; hGH = human Growth Hormone; TSH = Thyrotropin;
LH = Luteinizing Hormone; FSH = Follicle Stimulating Hormone.
Rhabdomyolysis Due to Adipsic Hypernatremia
Zantut-Wittmann et al.
1177Arq Bras Endocrinol Metab 2007;51/7
Figure 1. Magnetic resonance imaging of sella turcica showing a
suprasellar expansive lesion in contact with the encephalic trunk, accom-
plishing the 3rd ventricle.
Figure 2. Graph representing serum creatine phosphokinase (CK) and sodium
(Na) during the 6th hospital admission of the patient.
mEq/L IU/L
September/October/2003
180
160
140
120
100
80
60
40
20
0
13 15 17     19    21   23     25 27   29 1      3      5
25000
20000
15000
10000
5000
0
Na
CK
venous hydration and on oriented and assisted water
intake. After the reduction of CK levels and normali-
sation of renal function, the patient was instructed to
maintain continuous DDAVP use and regular and
periodic water ingestion. Glucose control was attained
with NPH and regular insulin and metformin.
Post-surgical visual field alterations remained
unchanged. The muscle integrity follow up was per-
formed through periodic CK, Na, and K serum deter-
minations. His treatment includes daily prednisone, L-
thyroxine, NPH and regular insulin, and monthly
testosterone propionate.
He is now asymptomatic, although he has pre-
sented some mild to moderate episodes of dehydration
and consequences, resolved with outpatient treatment —
as in June/2004: CK = 569 IU/L, Na = 147 mEq/L, K
= 3.2 mEq/L, creatinine clearance = 113 mL/min/1.72
m2; glucose = 102 mg/dL, HbA1c = 5.8% (RV =
3.9–6.1) and in February/2006: CK = 1,258 IU/L, Na
= 146 mEq/L, K = 3.4 mEq/L, creatinine = 0.94
mg/dL; glucose = 84 mg/dL, HbA1c = 6.5%.
DISCUSSION
Hypernatremia secondary to dehydration or to exces-
sive saline administration is rare in healthy young
adults. In hospitalised patients, hypernatremia is usual-
ly secondary to erroneous intravenous administration
of sodium (3). Only rarely do patients with cranio-
pharyngioma develop adipsia or hypodipsia secondary
to a tumour located in the hypothalamic region. How-
ever, it is more common following surgery, occurring
in approximately 20% of the cases (4). These alter-
ations can lead to hypernatremic states that can
unchain severe dehydration episodes, as well as behav-
ioural disturbances, delirium, lethargy, coma, and
muscular weakness due to rhabdomyolysis (1,2).
Rhabdomyolysis is a rare osmolarity disturbance com-
plication, characterised by muscular weakness and
pain. Significant CK levels confirm the diagnosis.
Acute renal failure and myoglobinuria may accompany
the clinical picture. Immediate consequences of mus-
cle destruction include hyperkalemia (increasing the
risk of fatal heart arrhythmia) and hypocalcemia. Acute
renal insufficiency is the result of renal vasoconstric-
tion, intratubular myoglobin deposition, and nephro-
toxicity caused by heme containing proteins (5).
Hypokalemia, hypophosphatemia, and hyponatremia
are uncommon causes of rhabdomyolysis (5). Rhab-
domyolysis can also be associated to the hyperosmo-
larity of diabetic coma (6,7). Hypernatremia causing
myopathy has been reported only a few times and the
association with renal impairment is much more rare
(2,8). One of the explanations for the development of
muscle lesion by hypernatremia could be the inhibi-
tion of Na-K transporter in the cytoplasm membrane
(1). In patients whose myopathy was associated to
hypernatremia (160 to 180 nmol/L), CK levels
ranged from 500 to 60,000 UI/L and the occurrence
of acute renal insufficiency was not rare. In some cases
there was an association with germinoma, pinealoma,
optic glioma or cerebral ischaemia. Diabetes insipidus
was present in only a few cases (2,6,8,9). In this case,
in addition to dehydration, the patient complained of
intense pain and muscular weakness. Although he pre-
sented severe dehydration and hyperglicemia, he did
not mention thirst, information essential in order to
establish the diagnosis of adipsia. Hypernatremia
aggravated by dehydration and hyperglycemia can
explain the aetiology of myopathy and acute renal
insufficiency. The latter is probably due to myoglobin
toxicity resulting from the rhabdomyolisis process
after dehydration. It is relevant to highlight that the
patient was euthyroid, normokalemic, normocalcemic,
and normophosphatemic during rhabdomyolisis,
hence the episodes were not precipitated by such
metabolic disturbances. Muscle lesion was confirmed
by extremely elevated CK levels, with a peak of 21,261
IU/L about 24 h after the admission. In such acute
episodes, the treatment for dehydration corrected
other hydro-electrolytic disturbances. Life long treat-
ment of adipsia is needed to maintain a normal hydro-
electrolytic balance, through systematic stimulated and
regular water ingestion (1). Additionally, a periodic
evaluation of sodium and CK serum levels is obligato-
ry in these patients due to the frequent oligosympto-
matic occurrence of mild or moderate dehydration and
consequent rhabdomyolysis. This is the first report of
a diabetic patient with craniophryngioma who devel-
oped diabetes insipidus and adipsia as a complication
of a hypothalamic tumour surgery, associated with
hyperglycemia, contributing to the hyperosmolarity,
evolving with hypernatremia and hydro-electrolytic
imbalance that provoked severe rhabdomyolysis and
acute renal insufficiency.
We would like to emphasise the need to estab-
lish the diagnosis of adipsia and the evaluation of mus-
cle integrity in hypernatremic states. Although the
solution for adipsia is simple, through volunteer water
intake, if not diagnosed, serious consequences can
occur such as repeated hypernatremia episodes, due to
dehydration, and intense rhabdomyolysis with elevat-
ed morbidity.
Rhabdomyolysis Due to Adipsic Hypernatremia
Zantut-Wittmann et al.
1178 Arq Bras Endocrinol Metab 2007;51/7
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2. Hiromatsu K, Kobayashi T, Fujii N, Itoyama Y, Goto I, Murakami
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salt intake resulting in the highest ever documented sodium
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6. Kung AWC, Pun KK, Lam KSL, Yeung RTT. Rhabdomyolisis
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Determinants of rhabdomyolysis in the diabetic state. Am J
Nephrol 1991;11:447-50.
8. Asahara H, Maruyama S, Motomura S, Tamura K, Mioshi T.
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Endereço para correspondência:
Denise Engelbrecht Zantut-Wittmann
Disciplina de Endocrinologia
Departamento de Clínica Médica
Faculdade de Ciências Médicas
Universidade Estadual de Campinas (Unicamp)
Caixa Postal 6111
Rua Tessália Vieira de Camargo 126
13084-971 Campinas, SP
Fax: (19) 3521-7408
E-mail: zantutw@fcm.unicamp.br
Rhabdomyolysis Due to Adipsic Hypernatremia
Zantut-Wittmann et al.
1179Arq Bras Endocrinol Metab 2007;51/7


Severe Rhabdomyolysis Due To Adipsic Hypernatremia After Craniopharyngioma Surgery.

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Severe Rhabdomyolysis Due To Adipsic Hypernatremia After Craniopharyngioma Surgery.

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