Adrenal Hemorrhage as a Rare Cause of Neonatal Anemia Associated with Hydrocephalus Secondary to Intraventricular Hemorrhage—A Case Report

doi:10.4236/ijcm.2013.410081

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International Journal of Clinical Medicine, 2013, 4, 455-458

http://dx.doi.org/10.4236/ijcm.2013.410081 Published Online October 2013 (http://www.scirp.org/journal/ijcm)

Adrenal Hemorrhage as a Rare Cause of Neonatal Anemia

Associated with Hydrocephalus Secondary to

Intraventricular Hemorrhage—A Case Report*

Krushnakumar Kesan#, Rahul Kumar Gupta, Paras Kothari, Abhaya Gupta, Ritesh Ranjan,

Kedar Mudkhedkar, Parag Karkera

MCh Pediatric Surgery, Department of Pediatric Surgery, LTMMC & LTMGH Sion, Mumbai, India.

Email: #[email protected]

Received May 8th, 2013; revised June 10th, 2013; accepted July 13th, 2013

License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

ABSTRACT

Neonatal adrenal hemorrhage is frequently associated with birth trauma, perinatal asphyxia, intrauterine infection, co-

agulation defects and thromboembolism. It has varied clinical presentation depending on degree of hemorrhage and

amount of adrenal cortex compromised by hemorrhage. The most common clinical presentations are persistent jaundice

and flank mass. We report a case of left sided adrenal hemorrhage in a breech delivered male neonate with perinatal

asphyxia presented with anemia and fever. On further evaluation, he was also having moderate communicating hydro-

cephalus secondary to intraventricular hemorrhage. The adrenal hemorrhage was managed conservatively. Subsequent

abdominal ultrasound showed resolving adrenal hemorrhage. Right ventriculoperitoneal shunt was done for hydro-

cephalus. Postoperative course was uneventful. The patient is asymptomatic at follow-up.

Keywords: Adrenal Hemorrhage; Neonatal Anemia; Hydrocephalus; Intraventricular Hemorrhage

1. Introduction

The adrenal gland in neonatal age is vulnerable to hem-

orrhage because of its large size and high vascularity [1].

The incidence of adrenal hemorrhage in detected cases

ranges from 1.7 to 2.1 per 1000 births. Because adrenal

bleeding may remain asymptomatic, the real occurrence

is probably higher [2]. Neonatal adrenal hemorrhage is

frequently associated with birth trauma, perinatal as-

phyxia, intrauterine infection, coagulation defects and

thromboembolism [1,3]. Adrenal hemorrhage usually

presents with neonatal jaundice, paleness and/or flank

mass, discoloration of the scrotum, anemia and adrenal

insufficiency [4-6]. In this report, we present a rare case

of neonatal adrenal hemorrhage having anemia and hy-

drocephalus.

2. Case Report

A 25-day-old, 2.8 kg, male neonate was referred to us

with complaint of fever and severe anaemia. Records

revealed that he was born of nonconsanguineous mar-

riage, fullterm, breech delivered with history of delayed

cry. APGAR scores were 4 and 5 at 1 and 5 minutes,

respectively. Patient was admitted to intensive care unit

for perinatal asphyxia.

On general examination gross pallor was evident and

there was no icterus. An abdominal mass was palpated in

the left flank. In addition, the baby was having wide open

anterior fontanels.

Haematological investigations were suggestive mi-

crocytic hypochromic anaemia (Hb 6.2 gm%, Platelet

count 7.1 lac/cu mm). Renal function and liver function

tests including coagulation profile were normal. Blood

culture and urine culture were sterile. TORCHS’ test

[toxoplasmosis, other infections, rubella, cytomegalovi-

rus (CMV), and herpes simplex virus (HSV)] was also

negative.

Abdominal ultrasound showed a well defined hypoe-

choic lesion in left suprarenal area, of size 4 × 3 cm, at

the upper pole of the left kidney suggestive of adrenal

mass which could be either neuroblastoma or adrenal

hemorrhage. The margins between the mass and the left

kidney were indistinct. Contrast enhanced computerised

tomography (CECT) abdomen revealed a 3 × 3.5 × 4 cm

*Conflict of interest: NIL.

#Corresponding author.

Adrenal Hemorrhage as a Rare Cause of Neonatal Anemia Associated with Hydrocephalus Secondary

to Intraventricular Hemorrhage—A Case Report

456

sized well defined cystic density lesion with enhancing

walls in left paravertebral region suggestive of adrenal

hemorrhage with superadded infection (Figures 1(a) and

(b)). USG and CT skull revealed moderate dilatation of

all four ventricles. CT also revealed intraventricular

hemorrhage in right temporoparital region (Figure 2).

Serum cortisol level and 24 - hour urinary vanillyl

mandelic acid (VMA) levels were normal, which ruled

(a)

(b)

Figure 1. CECT abdome n showing we ll defined cystic de nsity le sion with enhancing walls in left paravertebral region sugges-

tive of adrenal hemorrhage.

Adrenal Hemorrhage as a Rare Cause of Neonatal Anemia Associated with Hydrocephalus Secondary

to Intraventricular Hemorrhage—A Case Report

457

Figure 2. CECT abdome n showing we ll defined cystic de nsity le sion with enhancing walls in left paravertebral region sugges-

tive of adrenal hemorrhage.

out neuroblatoma as a differential diagnosis.

The patient was initially managed with blood transfu-

sions and intravenous antibiotic. Adrenal haemorrhage

was managed conservatively. Subsequent serial abdomi-

nal ultrasound done at 1 week interval for 2 weeks

showed gradual reduction in size of adrenal hemorrhage.

Ventricular CSF was tapped showing plenty of RBC.

Patient was kept on oral acetazolamide and CSF tap re-

peated. Right ventriculoperitoneal shunt was done for

hydrocephalus when CSF was clear. Postoperative course

was uneventful. The patient is asymptomatic at 6 months

of follow up and USG at 6 months showed complete

resolution of hemorrhage.

3. Discussion

Adrenal hemorrhage is a relatively uncommon condition

frequently seen in term male infants delivered vaginally

[7,8]. The incidence of detected cases ranges from 1.7 to

2.1 per 1000 births [4]. Adrenal hemorrhage is more

common in neonates than in children or adults because of

large size (relative to body weight gland is 10 to 20 times

larger than adults) and unique blood supply. It is supplied

by subcapsular plexus formed by fifty to sixty arterial

branches from three suprarenal arteries. This plexus is

drained into medullary sinusoids via a very few venous

channels. In case of hypoxia due to any cause there oc-

curs shunting of blood from vital organ to adrenal gland

which resulting into hemorrhage. Various predisposing

factors are birth trauma, perinatal asphyxia, intrauterine

infection, coagulation defects and thromboembolism, or

it can be spontaneous.

There is a male predominance for this condition. Right

side is more commonly affected than left and 5% - 10%

cases may be bilateral [9]. Right adrenal gland is more

susceptible, as it is more likely to be compressed be-

tween the liver and spine and, the right adrenal vein usu-

ally drains directly into the inferior vena cava, so it is

prone to changes in venous pressure.

Adrenal hemorrhage may be asymptomatic or may

presents with neonatal jaundice, paleness and/or flank

mass, discoloration of the scrotum, anemia and adrenal

insufficiency [4]. Clinical presentations depend on de-

gree of hemorrhage and amount of adrenal cortex com-

promised by hemorrhage. Our patient was having anemia

with flank mass, in addition moderate communicating

hydrocephalus and intraventricular hemorrhage.

In case of neonatal adrenal hemorrhage it is very im-

portant to rule out neuroblastoma particularly the cystic

form. 24-hour urinary excretion of VMA should be done,

since an increase in VMA is virtually diagnostic of

neuroblastoma [10]. Other differential diagnosis of AH

are neonatal adrenal abscess, cortical renal cyst, ob-

structed upper cortical renal cyst and an obstructed upper

excretory tract in the duplicated kidney [11].

Diagnosis is established with USG and CECT abdo-

men. Serial abdominal USG are more helpful as they

Adrenal Hemorrhage as a Rare Cause of Neonatal Anemia Associated with Hydrocephalus Secondary

to Intraventricular Hemorrhage—A Case Report

458

show resolving adrenal hemorrhage. The pattern of echo-

genicity of an adrenal hematoma depends on its age. An

early-stage hematoma appears solid with diffuse or in-

homogeneous echogenicity. As liquefaction occurs, the

mass demonstrates mixed echogenicity with a central

hypoechoic region and eventually becomes completely

anechoic and cyst like. Calcification can appear as early

as 1 - 2 weeks after hemorrhage.

Presentation of adrenal hemorrhage with neonatal

anemia, flank mass has been reported by number of au-

thors [2,12]. But association between adrenal hemor-

rhage and hydrocephalus has not been reported in litera-

ture. Both conditions can be attributed with common

etiological factor of perinatal asphyxia.

4. Conclusion

In case of neonatal anaemia with perinatal asphyxia, ad-

renal haemorrhage should be suspected. Most of adrenal

haemorrhage resolves with conservative. Our patient was

having adrenal hemorrhage and hydrocephalus secondary

to intraventricular hemorrhage.

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