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| Volume 2 | Number 2 | Juli 2018|
Volume 2 ● Number 2 ● Juli 2018 Jurnal Bedah Nasional
Program Studi Ilmu Bedah FK Universitas Udayana dan IKABI cabang Bali ● ISSN: 2548-5962
DAFTAR ISI
Halaman
BOCHDALEK HERNIA
Yuliaji Narendra Putra, Tubagus Odih Rhomdani Wahid, Guntur Surya Alam,
Rohadi
40-43
EVALUASI FAKTOR RISIKO YANG MEMPENGARUHI LUARAN
OPERASI ENDORECTAL PULL-THROUGH SOAVE MODIFIKASI
SOEWARNO PADA PENYAKIT HIRSCHSPRUNG
Yuliaji Narendra Putra
44-50
CASE REPORT OF A NEONATE WITH CONGENITAL LUMBAR HERNIA
Stephanus Haryanto Hokardi, Neil Angelo S. Sael
51-55
BILATERAL GIANT FIBROADENOMA OF THE BREAST: A CASE
REPORT
Jasmine Stephanie Christian, Putu Anda Tusta Adiputra, INW. Steven Christian
56-59
GOITER MULTINODUL DENGAN PELEBARAN KE
RETROSTERNAL: LAPORAN KASUS
Gede Budhi Setiawan
60-63
CASE REPORT
40 | Jurnal Bedah Nasional
BOCHDALEK HERNIA
Yuliaji Narendra Putra1, Tubagus Odih Rhomdani Wahid2, Guntur Surya Alam2, Rohadi3
1General Surgery Training Programme, Faculty of Medicine Gadjah Mada University, Dr. Sardjito Hospital.
Correspondence: yp_narendra@yahoo.com. 2Pediatric Surgery Training Programme, Faculty of Medicine Gadjah Mada University, Dr. Sardjito Hospital.
3Pediatric Surgery Division, Faculty of Medicine Gadjah Mada University, Dr. Sardjito Hospital.
ABSTRACT
Background: Bochdalek hernia is a congenital defect on posterolateral diaphragm with an
abnormal connection between the thoracic cavity and the abdominal cavity. This disease causes
protrusion of abdominal organs into the thoracic cavity. Case: an 8-day-old baby girl admitted to
hospital with shortness of breath 24 hours after delivered. The baby was born spontaneously assisted
by midwife. Upon born, the baby was crying strongly and meconium came out 2 hours after birth. On
physical examination, the abdomen was inspected flat. Darm contour and darm steifung was
observed, and peristaltic sound was heard on left lung. Radiological examination demonstrated a
diaphragmatic hernia with ileus obstruction. The patient underwent laparatomy and stomach, ileum,
transverse colon, and spleen, was found on foramen Bochdalek. Post-surgery chest X Ray showed
favourable result. Ten days after treatment, the patient was discharged in a good condition with no
respiratory or digestive problems. After 1 months the patient’s condition remained good and there
were no respiratory or digestive complaints. Conclusion: In a rare case like Bochadalek hernia,
laparotomy performed as a promising attempt to return the anatomic position of organ.
Keywords: Bochdalek hernia, laparotomy, surgery.
HERNIA BOCHDALEK
Yuliaji Narendra Putra1, Tubagus Odih Rhomdani Wahid2, Guntur Surya Alam2, Rohadi3
1Program Studi Ilmu Bedah, Fakultas Kedokteran Universitas Gadjah Mada, RSUP Dr. Sardjito. Korespondensi:
yp_narendra@yahoo.com.
2Program Studi Ilmu Bedah Anak, Fakultas Kedokteran Universitas Gadjah Mada, RSUP Dr. Sardjito.
3Divisi Bedah Anak, Fakultas Kedokteran Universitas Gadjah Mada, RSUP Dr. Sardjito.
ABSTRAK
Latar belakang: hernia Bochdalek adalah cacat bawaan pada diafragma posterolateral yang
terdapat hubungan antara rongga thoraks dan rongga perut, sehingga terjadi penonjolan organ perut ke
dalam rongga thoraks. Kasus: seorang bayi perempuan umur 8 hari dibawa ke rumah sakit swasta
dengan keluhan sesak nafas 24 jam setelah dilahirkan. Satu minggu yang lalu, pasien lahir spontan
ditolong bidan,menangis kuat, mekonium keluar 2 jam setelah lahir. Pada pemeriksaan fisik
didapatkan abdomen flat,didapatkan darm contour dan darm steifung, Suara peristaltik terdapat pada
auskultasi di hemithoraks kiri. Pada pemeriksaan radiologis terdapat gambaran udara usus pada
hemithoraks kiri. Dari pemeriksaan diatas ditegakkan diagnosis hernia diafragmatika dengan ileus
obstruksi. Dilakukan laparotomi, durante operasi didapatkan gaster, ileum, kolon transversum, dan
lien masuk ke dalam rongga thoraks sinistra, defek ukuran 2x8 cm pada posterior (foramen
Bochdalek). Rontgen thoraks post laparotomy paru-paru mengembang kedua sisi dan diafragma kiri
terlihat intak. Sepuluh hari setelah menjalani perawatan, pasien pulang dengan keadaan baik tidak ada
gangguan pernafasan maupun pencernaan. Evaluasi selama 1 bulan terkhir keadaan pasien tetap baik
Volume 2 ● Number 2 ● Juli 2018 Bochdalek Hernia
41
dan tidak ada keluhan pernafasan maupun pencernaan. Simpulan: telah dilakukan laparotomi untuk
mengembalikan ke posisi anatomi pada pasien hernia Bochdalek dengan hasil baik.
Kata kunci: hernia Bochdalek, laparotomi, operasi.
INTRODUCTION
Bochdalek hernia is a congenital defect
on posterolateral diaphragm with an
abnormal connection between the thoracic
cavity and the abdominal cavity, resulting
in the protrusion of the abdominal organs
into the thoracic cavity.1 The incidence in
neonates is 1:2000-50002-3 with pulmonary
hypoplasia as the most common
complication.4 Despite advances in the
diagnosis and treatment of congenital
defects, pulmonary hypoplasia still caused
a fairly high mortality rate.5,6
Surgical treatment can be performed
either through the abdominal or thoracic
approach. The abdominal approach has the
advantage of correcting malrotation at the
same time.7 However, closure of the
abdominal wall can result in the difficulty
of organs replacement to abdominal cavity.
For the solution, Charles et al has
recommended a delayed abdominal muscle
closure. In this case, it will be presented
regarding the management of Bochdalek
hernia in children.
CASE REPORT
An 8-day-old baby girl was admitted
taken to a private hospital with shortness
of breath. The patient was born
spontaneously assisted by midwife, crying
strongly, and meconium came out 2 hours
after birth. At 24 hours later, the patient
appeared to have difficulty of breathing,
and referred to a primary hospital. After 6
days of treatment, no improvement was
observed, thus the patient was referred to a
tertiary hospital.
In physical examination, the baby
showed high body temperature, shortness
of breath with respiratory rates 39 times
per minute, but the baby could cry
strongly. Chest x-ray examination showed
multiple cystic shadow at the left
hemithorax accompanied by right
deviation of mediastinum and trachea with
suspicion of diaphragmatic hernia. On the
abdominal x-ray, the distal end of the
nasogastric tube only reached 5th-6th
thoracic vertebra with intestinal air
portrayed on the left chest. Babygram
examination showed that left intestine on
hemithorax was unfilled with contrasts, the
air intestinal image on the left hemithorax
is not filled with contrasts, with left
diaphragmatic hernia image showing
intestines and colon (Figure 1).
Figure 1. Babygram pre-surgery.
Preoperative diagnosis is left
diaphragmatic hernia and laparotomy
surgery via abdominal approach was
decided. A transverse incision of the upper
umbilical (3 cm above of umbilicus),
deepened layer by layer to the peritoneum.
After the peritoneum was opened,
Yuliaji Narendra Putra Jurnal Bedah Nasional
42
intestinal, gastric, and part of transverse
colon system were seen inside of the
abdomen. The inlet of foramen Bochdalek
was seen released and re-inserted the
content of the hernia consisting ileum and
part of colon to the inside of the abdomen
cavity (Figure 2). The diameter of
foramen Bochdalek was 2x8 cm, then
incision on septum tendinous was made
then 2 layers was sutured. Aspiration of
pleura cavity was performed with
nasogastric tube. Exploration showing
intestinal systems is intact. The surgical
wound was closed layer by layer. The
surgery was completed.
Figure 2. Clinical picture during surgery.
Chest X-ray after surgery showed
inflation of left lung. The patient was
treated for 10 days and discharged on the
11th day after the surgery. Follow up for 1
months shows patient’s good condition
and neither disturbance was observed in
the digestive system nor respiratory
system.
DISCUSSION
Bochdalek hernia (Figure 3) is a
congenital defect on posterolateral
diaphragm developing an abnormal
connection between the thoracic cavity and
the abdominal cavity. This pleuroparietal
canal normally closed by the pleuroparietal
membrane at the 8th to 10th week of
pregnancy. Failure of canal closure might
result in Bochdalek hernia. Approximately
80-90% cases of Bochdalek hernia occur
on the left side and are very rare in adults.
In this case, a large defect in the left
posterolateral diaphragm (Bochdalek
foramina) was encountered with the
content of ileum and part of colon.4,5
Bochdalek hernia diagnosed from
anamnesis, physical examination,
investigation especially chest x-ray and
OMD.6
Figure 3. Location of Bochdalek Foramen.8
The diagnosis of this patient was made
after physical and radiological examination
because rarity of this disease and the non-
specific diagnosis could lead to delay of
diagnosis. Surgical repair of the defect is
the recommended therapy for Bochdalek
Hernia.7 For this patient, laparotomy
surgery was performed with abdominal
approach.8 A transverse incision of the
upper umbilical (3 cm above of
umbilicus), deepened layer by layer to the
peritoneum. After the peritoneum was
opened, intestinal system, gastric, and part
of transverse colon was seen inside of the
abdomen. Post-surgery showed inflation of
left lung. Long term follows up was also
favourable. This showed that laparotomy
for management of Bochdalek Hernia led
to either short term and long term results.
Volume 2 ● Number 2 ● Juli 2018 Bochdalek Hernia
43
CONCLUSION
Laparotomy performed as an attempt to
return the anatomic position of patient with
Bochdalek hernia showing good result.
REFERENCES
1. Brunicardi FC, Billiar TR, Dunn DL,
et al. Congenital Diaphragmatic
Hernia. In: Schwartz SI, Fischer JE,
Daly JM, et al., editors. Schwartz’s
Principles of Surgery. 7th Edition.
Asian Student Edition. Singapore:
McGraw-Hill; 1998. p.1691-3.
2. Way LW. Esophagus & Diaphragm.
In: Way LW, Doherty GM, editors.
Current Surgical Diagnosis and
Treatment. 11th Edition. Connecticut:
McGraw-Hill/Appleton & Lange;
2002. p.438-40.
3. Townsend CM, Beauchamp RD,
Mattox KL, et al. Congenital
Diaphragmatic Hernia In: Townsend
CM, Beauchamp RD, Evers BM, et al.,
editors. Sabiston Textbook of Surgery:
The Biological Basis of Modern
Surgical Practice. 16th Edition.
Philadelphia: WB Saunders; 2001.
p.1480-3.
4. Eisenberg RL. Esophagus and
Diaphragmatic Hernia In: Eisenberg
RL, editor. Diagnostic Imaging in
Surgery. New York: McGraw-Hill;
1987. p.275-6.
5. Liechty RD. Respiratory
Diaphragmatic Hernia. In: Niederhuber
JE, editor. Fundamentals of Surgery.
Stamford: Appleton & Lange; 1998.
p.192-3.
6. Reynolds M. Diaphragmatic
Anomalies. In: Raffensperger JG,
editor. Swenson’s Textbook of
Paediatric Surgery. 5th edition. New
York: Appleton & Lange; 1990. p.
721-30.
7. Burki T, Amanullah A, Rehman AU, et
al. Late Presentation of Bochdalek
Hernia with Intestinal Symptoms. J
Ayub Med Coll Abbottabad. 2002;14:
27-8.
8. Nason LK, Walker CM, McNeeley
MF, et al. Imaging of the Diaphragm:
Anatomy and Function.
RadioGraphics. 2012;32:E51-70.
ORIGINAL ARTICLE
44 | Jurnal Bedah Nasional
EVALUASI FAKTOR RISIKO YANG MEMPENGARUHI LUARAN
OPERASI ENDORECTAL PULL-THROUGH SOAVE MODIFIKASI
SOEWARNO PADA PENYAKIT HIRSCHSPRUNG
Yuliaji Narendra Putra
Program Studi Ilmu Bedah, Fakultas Kedokteran Universitas Gadjah Mada, RSUP Dr. Sardjito. Korespondensi:
yp_narendra@yahoo.com.
ABSTRAK
Tujuan: untuk mengevaluasi luaran operasi endorectal pull-through Soave modifikasi Soewarna
pada pasien Hirschsprung. Metode: penelitian ini adalah penelitian deskriptif pada 48 penderita
Hirschsprung, dimana data dikumpulkan dengan wawancara langsung ke orang tua penderita. Setelah
didapatkan skoring, kemudian dinilai angka keberhasilan penatalaksanaan operasi Soave modifikasi
Soewarno, kemudian dilakukan analisis dengan menggunakan uji statistik Chi-kuadrat. Analisis
antara skor Klotz dengan faktor prognostik, seperti berat badan lahir, status gizi, berat badan saat
operasi, kadar albumin, kadar hemoglobin, kadar kalium, lama perawatan, dan jenis kelamin di
analisis dengan uji Chi-Kuadrat. Hasil: pada penelitian ini, didapatkan pada faktor-faktor risiko yang
mempengaruhi luaran operasi endorectal pull-through Soave modifikasi Soewarno adalah status gizi
p<0,001 dengan RR 28,0 dan albumin p=0,047 dengan RR 1,23. Faktor risiko hemoglobin (p=0,372),
kalium (p=0,256), berat badan lahir (p=0,66), berat badan saat operasi (p=0,0605), lama operasi
(p=0,941), dan lama perawatan (p=0,683) tidak berpengaruh secara signifikan. Simpulan: Status gizi
dan kadar albumin menjadi faktor prognosis signifikan terhadap luaran pasien dengan penyakit
Hirchsprung yang dilakukan tindakan operasi Soave modifikasi Soewarno.
Kata kunci: penyakit Hirschsprung, faktor prognostik, skor Klotz.
EVALUATION OF RISK FACTORS AFFECTING OUTCOME OF SOAVE
ENDORECTAL PULL-THROUGH OPERATION SOEWARNO
MODIFICATION IN HIRSCHSPRUNG’S DISEASE
Yuliaji Narendra Putra
General Surgery Training Programme, Faculty of Medicine Gadjah Mada University, Dr. Sardjito Hospital.
Correspondence: yp_narendra@yahoo.com.
ABSTRACT
Objective: to evaluate the outcome of Soave endorectal pull-through operation with Soewarno
modification in Hirschsprung patients. Methods: this study was a descriptive study of 48
Hirschsprung patients, where data were collected by direct interviews to the patient's parents. After
scoring then assessed the success rate of Soave operation Soewarno modification, then analyzed by
using statistic Chi-square test. The analysis between Klotz score with prognostic factors, such as birth
weight, age of Soave, nutritional status, body weight during surgery, albumin level, hemoglobin level,
potassium level, length of treatment, and sex in analysis with Chi-Square test. Results: the significant
prognostic factors affected the outcome of Soave endorectal pull-through operation with Soewarno
modification in Hirschsprung patients were nutritional status (RR 28; p=0.047) and hemoglobin (RR
1.14; p<0.001). Others factors like hemoglobin (p=0.372), kalium (p=0.256), birth weight (p=0.66),
weight before surgery (p=0.0605), duration of surgery (p=0.941), dan length of hospital stay
(p=0.683). Conclusion: nutritional status and hemoglobin level were the most significant factors that
Volume 2 ● Number 2 ● Juli 2018 Evaluasi Faktor Risiko yang Mempengaruhi
45
affected the outcome of Hirchsprung’s disease performed Soave operation with Soewarno
modification.
Keywords: Hirschsprung's disease, prognostic factor, Klotz score.
PENDAHULUAN
Penyakit Hirschsprung disebut juga
megakolon kongenital merupakan kelainan
tersering dijumpai sebagai penyebab
obstruksi usus pada neonatus. Pada
penyakit ini, tidak dijumpai pleksus
mienterikus sehingga bagian usus tersebut
tidak dapat mengembang.1,2 Angka
insidensi Hirschprung adalah 1 diantara
5000 kelahiran, maka dengan penduduk
220 juta dan tingkat kelahiran 35 per mil,
diperkirakan akan lahir 1400 bayi setiap
tahunnya dengan penyakit Hirschsprung di
Indonesia.3-6
Pasien dengan penyakit Hirschsprung
harus dikelola segera setelah diagnosis
ditegakkan.3 Prosedur Soave disebut juga
prosedur pull-through ekstramukosa
endorektal dari Soave. Di Subbagian
Bedah Anak FK UGM/RSUP Dr.Sardjito
Yogjakarta, Soave dikerjakan mulai awal
tahun 1990, dalam pelaksanaannya
didapatkan kesulitan dalam pengupasan
mukosa, sehingga diciptakan teknik
prosedur modifikasi Soewarno dan hingga
saat ini dijadikan prosedur tetap untuk
penanganan penderita Hirschsprung di Sub
Bagian Bedah Anak UGM/RS Dr. Sardjito
Yogyakarta. Walaupun begitu, proses
penyembuhan atau keberhasilan setelah
operasi sangat tergantung pada sistem
imun dan kemampuan adaptasi pasien.
Dalam hal ini yang berpengaruh adalah
usia, kadar hemoglobin, kadar kalium
serum, kadar albumin serum, status gizi,
berat badan lahir, berat badan saat operasi,
dan lama perawatan. Tujuan penelitian ini
mengevaluasi luaran operasi endorectal
pull-through Soave modifikasi Soewarno
pada pasien Hirschsprung.
METODE
Penelitian ini dilakukan dengan
rancangan analitik deskriptif cross
sectional pada penderita Hirshsprung yang
ditatalaksana dengan metode Soave
modifikasi Soewarno di RS. Dr. Sardjito
Yogyakarta antara bulan Januari 2005
sampai Februari 2008. Tujuan penelitian
adalah untuk mencari faktor risiko yang
berpengaruh terhadap luaran pasien
Hirschsprung yang ditatalaksana dengan
prosedur Soave modifikasi Soewarno.
Parameter yang dinilai untuk variabel
independen (risiko) meliputi usia, kadar
hemoglobin, kadar kalium serum, kadar
albumin serum, status gizi, berat badan
lahir, berat badan saat operasi, dan lama
perawatan. Variabel dependen atau luaran
hasil operasi dinilai dengan skor Klotz.
Semua parameter didapatkan datanya dari
rekam medis dan wawancara atau
pengukuran langsung. Skor Klotz ≤13
dinyatakan baik/cukup dan dinyatakan
jelek/kurang jika skor Klotz >13.
Pada seluruh subjek penelitian, harus
menyetujui informed consent dari orang
tuanya. Data usia, status gizi, dan berat
badan diperoleh dengan melakukan
alloanamnesis dan pemeriksaan fisik.
Pemeriksaan barium enema dilakukan
untuk mengetahui penyakit Hirschsprung,
seperti adanya penyempitan rektosigmoid,
daerah transisi, daerah dilatasi dan
ketidakteraturan mukosa. Tindakan bedah
definitif dilakukan pada semua pasien
Yuliaji Narendra Putra Jurnal Bedah Nasional
46
yang menderita penyakit Hirschsprung
yang sudah dikerjakan kolostomi. Jenis
prosedur bedah yang dikerjakan adalah
prosedur pull-through ekstramukosa
endorektal prosedur Soave modifikasi
Soewarno.
Prosedur operasi modifikasi Soewarno
dilakukan dengan irisan transversal pada
dinding depan abdomen mulai 4 cm
sebelah medial SIAS (spina iliaka anterior
superior) kanan melalui garis langer
sampai mencapai lobang kolostomi. Irisan
delanjutkan melengkung ke kraniolateral
secukupnya. Arteri hemorrhoidalis
superior dan arteri sigmoidalis di
identifikasi selanjutnya diikat dan
dipotong. Dilakukan reseksi kolon 3-4 cm
di proksimal kolostomi dan 1-2 cm di
proksimal refleksi peritoneum. Pungtum
proksimal kemudian ditutup, dilakukan
pengupasan mukosa rektum dari lapisan
seromuskuler, dengan cara memegang
mukosa dengan 4 buah klem Allis. Irisan
pertama dilakukan secara tajam
selanjutnya seromuskuler dipegang dengan
4 buah klem Allis, selanjutnya dilakukan
pengupasan secara tumpul. Pengupasan ke
anal sejauh mungkin sehingga mencapai
linea dentata. Selanjutnya dilakukan
pembebasan kolon proksimal yang sehat,
sampai cukup untuk diteroboskan keluar
anus. Pembebasan ini harus hati- hati
sehingga jalinan pembuluh darah tetap
terjamin. Bila sudah dinilai cukup, maka
operasi dilanjutkan lewat peritoneum.
Anus disiapkan, kemudian cerobong
mukosa ditarik dengan jalan memasukkan
sonde khusus dengan ujung berbentuk
kepala yang lebih besar. Mukosa diikat
pada leher sonde tersebut dan ditarik
keluar secara melipat terbalik. Kolon yang
sehat kemudian diteroboskan di dalam
cerobong mukosa. Lapisan mukosa
difiksasi dengan kolon dengan benang
plain catgut, dan dipasang rectal tube di
dalam kolon yang diteroboskan tersebut
sampai melewati sfingter ani. Operasi
dilanjutkan lewat abdominal, vesika
urinaria, dan organ abdomen lain ditata
kembali, cerobong seromuskuler difiksasi
dengan serosa kolon yang diteroboskan
dengan chromic catgut. Dilakukan
appendektomi insidental, rongga abdomen
dicuci dan ditutup lapis demi lapis.
Sepuluh hari setelah dioperasi endorectal
pull-through, telah terjadi perlekatan
antara cerobong seromuskuler dengan
serosa kolon. Dilakukan pemotongan
pungtum kolon yang diteroboskan 1 cm
proksimal linea dentata, dilanjutkan
dengan penjahitan mukosa dengan
mukosa. Selama 3 hari rectal tube terus
dipasang pada rektum yang baru sehingga
gangguan obstruksi akibat edema di daerah
anorektal dapat dihindari.8
Setelah operasi, peneliti mewawancara
orang tua pasien dengan menggunakan
kuesioner berisi tabel skor Klotz (Tabel 1)
terhadap hasil pembedahan operasi
definitif metode Soave modifikasi
Soewarno kemudian dibuat skoring Klotz
tiap pasien berdasarkan kuisioner hasil
wawancara peneliti dengan orang tua
pasien. Hasil skoring Klotz dicari
korelasinya dengan faktor prognostik
disajikan secara tekstular dan tabular dan
diuji statistik dengan Chi-kuadrat.
HASIL
Karakteristik subjek penelitian dari 48
kasus didapatkan jenis kelamin laki-laki
terdapat 35 orang (72,92%) dan
perempuan ada 13 orang (27,08%). Rata-
rata usia 627,32±525,36 hari dengan usia
terendah 1 hari dan maksimum 1980 hari
(5,42 tahun). Berat badan lahir rata-rata
2,93±0,27 kg dengan berat minimal 1,90
kg dan berat maksimal 3,60 kg. Status gizi
Volume 2 ● Number 2 ● Juli 2018 Evaluasi Faktor Risiko yang Mempengaruhi
47
yang baik ada 41 orang (85,4%),
sedangkan status gizi jelek ada 7 orang
(14,6%).
Tabel 1. Tabel Skor Klotz
No Variabel Kondisi Skor
1 Defekasi 1-2 kali sehari 1
2 hari sekali 1
3 – 5 kali sehari 2
3 hari sekali 2
> 4 hari sekali 3
> 5 hari sekali 3
2 Kembung Tidak Pernah 1
Kadang – kadang 2
Terus Menerus 3
3 Konsistensi Normal 1
Lembek 2
Encer 3
4 Perasaan ingin BAB Terasa 1
Tidak terasa 3
5 Soiling / Kecepirit Tidak pernah 1
Bersama Flatus 2
Terus menerus 3
6 Kemampuan menahan
feses yang akan keluar
Lebih dari satu menit 1
Kurang dari satu
menit
2
Tidak bisa 3
7 Komplikasi Lain Tidak ada 1
Minor 2
Mayor 3
Penilaian hasil skoring:
a. Skor 7: sangat baik
b. skor 8-9: baik
c. Skor 10-13: cukup
d. Skor 14-lebih: kurang/ jelek
Hasil analisis Klotz terhadap jenis
kelamin, hemoglobin, dan kalium, secara
statistik tidak bermakna dimana p>0,05.
Skor Klotz bermakna secra statistik pada
status gizi dan albumin, yang mana status
gizi dan albumin yang baik memiliki skor
Klotz yang baik (Tabel 2). Pada Tabel 3,
hasil analisis Klotz terhadap rata-rata
umur, berat badan lahir, berat badan saat
operasi, lama operasi, dan lama perawatan
secara statistik tidak bermakna p>0,05.
DISKUSI
Pasien dengan penyakit Hirschsprung
harus dikelola segera setelah diagnosis
ditegakkan. Pengobatan definitif
aganglionosis kolon adalah pembedahan
dengan membuang semua bagian yang
aganglionik, kemudian membawa usus
(kolon) yang normal persarafannya
(ganglionik) ke anus dengan
memperhatikan kontinensi. Tanpa
penanganan, tingkat mortalitas penyakit ini
80%, di mana pasien penyakit
Hirschsprung akan meninggal pada bulan-
bulan pertama kehidupannya, sebagian
besar pada masa neonatus. Keterlambatan
dan kegagalan tindakan bedah, baik
tindakan bedah sementara maupun bedah
definitif dapat mengakibatkan cacat
bahkan kematian.3
Prosedur Soave merupakan prosedur
pembedahan yang paling sering dilakukan.
Prosedur Soave konvensional dilakukan
dengan pemisahan mukosa dan
seromuskuler dengan prokain hidroklorida,
tepat di proksimal masuknya arteri
hemorrhoidalis superior ke lapisan otot
rektum. Pengupasan lapisan seromuskuler
dari lapisan mukosa dengan kasa bulat dan
kecil. Setelah lapisan seromuskuler
terpisah dengan mukosa dilakukan
pemotongan mukosa secara melintang
dilanjutkan dengan pembebasan kolon
proksimal sepanjang 30-50 cm di
proksimal daerah transisi. Di perineum,
mukosa kolon ditarik keluar lubang anus
dan dijahit.7
Untuk lebih memudahkan dengan hasil
luaran yang lebih baik, dilakukan prosedur
operasi modifikasi Soewarno yang
dilakukan dengan irisan transversal pada
dinding depan abdomen mulai 4 cm
sebelah medial SIAS kanan melalui garis
langer sampai mencapai lobang kolostomi.
Irisan dilanjutkan melengkung ke
kraniolateral secukupnya. Arteri
hemorrhoidalis superior dan arteri
Yuliaji Narendra Putra Jurnal Bedah Nasional
48
sigmoidalis di identifikasi selanjutnya
diikat dan dipotong.
Tabel 2. Analisis uji Chi-kuadrat antar faktor risiko dengan skor Klotz
Variabel Skor Klotz RR 95% CI p
Jelek Baik
Jenis kelamin (%)
Laki-laki 1 (20,0) 12 (27,9) 0,67 0,08-5,48 0,706
Perempuan 4 (80,9) 31 (72,1)
Status gizi (%)
Kurang baik 4 (80) 2 (4,7) 28,0 3,72-210,5 0,000
Baik 1 (20) 41 (95,3)
Albumin (%)
Jelek (<3,5 g/dL) 0 (0) 21 (48,8) 1,23 1,02-1,47 0,047
Baik 5 (100) 22 (51,2)
Hemoglobin (%)
Jelek (<11 g/dL) 0 (0) 6 (14) 1,14 1,02-1,27 0,372
Baik 5 (100) 37 (86)
Kalium (%)
Jelek (<3,5 mmol/L) 0 (0) 9 (20,9) 1,15 1,02-1,29 0,256
Baik 5 (100) 34 (79,1)
RR: risiko relatif, CI: confidence interval
Tabel 3. Hasil skor Klotz terhadap rata-rata umur, berat badan lahir, berat badan saat operasi,
dan lama operasi, dan lama perawatan
Variabel Skor Klotz (rata-rata ± standar deviasi) p
Jelek Baik
Umur (hari) 439,60 ± 406,11 649,1 ± 537,1 0,405
Berat badan lahir (kg) 2,98 ± 0,265 2,93 ± 281,47 0,660
Berat badan saat operasi (kg) 11,40 ± 1,67 8,1 ± 3,84 0,065
Lama operasi (jam) 2,45 ± 0,447 2,47 ±0,60 0,941
Lama perawatan (hari) 12,6 ± 1,34 13,39 ± 4,26 0,683
Dilakukan reseksi kolon 3-4 cm di
proksimal kolostomi dan 1-2 cm di
proksimal refleksi peritoneum. Pungtum
proksimal kemudian ditutup, dilakukan
pengupasan mukosa rektum dari lapisan
seromuskuler, dengan cara memegang
mukosa dengan 4 buah klem Allis. Irisan
pertama dilakukan secara tajam
selanjutnya seromuskuler dipegang dengan
4 buah klem Allis, selanjutnya dilakukan
pengupasan secara tumpul. Pengupasan ke
anal sejauh mungkin sehingga mencapai
linea dentata. Selanjutnya dilakukan
pembebasan kolon proksimal yang sehat,
sampai cukup untuk diteroboskan keluar
anus. Pembebasan ini harus hati- hati
sehingga arkade pembuluh darah tetap
terjamin. Bila sudah dinilai cukup, maka
operasi dilanjutkan lewat peritoneum.
Anus disiapkan, kemudian cerobong
mukosa ditarik dengan jalan memasukkan
sonde khusus dengan ujung berbentuk
kepala yang lebih besar. Mukosa diikat
pada leher sonde tersebut dan ditarik
keluar secara melipat terbalik. Kolon yang
sehat kemudian diteroboskan di dalam
cerobong mukosa. Lapisan mukosa
difiksasi dengan kolon dengan benang
plain catgut, dan dipasang rectal tube di
dalam kolon yang diteroboskan tersebut
sampai melewati sfingter ani. Operasi
dilanjutkan lewat abdominal, vesika
urinaria, dan organ abdomen lain ditata
kembali, cerobong seromuskuler difiksasi
Volume 2 ● Number 2 ● Juli 2018 Evaluasi Faktor Risiko yang Mempengaruhi
49
dengan serosa kolon yang diteroboskan
dengan chromic catgut. Dilakukan
appendektomi insidental, rongga abdomen
dicuci dan ditutp lapis demi lapis.
Sepululuh hari setelah dioperasi endorectal
pull-through, telah terjadi perlekatan
antara cerobong seromuskuler dengan
serosa kolon. Dilakukan pemotongan
pungtum kolon yang diteroboskan 1 cm
proksimal linea dentata, dilanjutkan
dengan penjahitan mukosa dengan
mukosa. Selama 3 hari rektal tube terus
dipasang pada rektum yang baru sehingga
gangguan obstruksi akibat udema di
daerah anorektal dapat dihindari.8
Di Subbagian Bedah Anak FK
UGM/RSUP Dr.Sardjito Yogjakarta,
Soave dikerjakan mulai awal tahun 1990,
dalam melaksanakan tersebut didapatkan
kesulitan dalam pengupasan mukosa,
sehingga diciptakan teknik prosedur
modifikasi Soewarno dan hingga saat ini
dijadikan prosedur tetap untuk penanganan
penderita Hirschsprung di Sub Bagian
Bedah Anak UGM/RS Dr. Sardjito
Yogyakarta. Walaupun begitu, proses
penyembuhan atau keberhasilan setelah
operasi sangat tergantung pada sistem
imun dan kemampuan adaptasi pasien.
Dalam hal ini yang berpengaruh adalah
Usia,kadar hemoglobin, kadar kalium
serum, kadar albumin serum, status gizi,
berat badan lahir, berat badan saat operasi,
dan lama perawatan.
Hasil analisis skor Klotz pada pasien
dengan penyakit Hirchsprung yang
dilakukan tindakan operasi Soave
modifikasi Soewarno dipengaruhi faktor
prognosis berupa status gizi (p=0,000
dengan RR 28,0) dan albumin (p=0,047
dengan RR 1,23) yang secara statistik
signifikan. Sedangkan faktor lain seperti
jenis kelamin, hemoglobin, kalium, umur,
berat badan lahir, berat badan saat operasi,
lama operasi, dan lama perawatan tidak
berpengaruh pada skor Klotz.
SIMPULAN
Teknik operasi endorectal pull-through
Soave modifikasi Soewarno merupakan
pilihan terapi pada penyakit Hirschsprung.
Penyakit Hirchsprung yang dilakukan
tindakan operasi Soave modifikasi
Soewarno dipengaruhi faktor prognosis
berupa status gizi dan albumin, yang dapat
mempengaruhi penyembuhan paska
tindakan.
DAFTAR PUSTAKA
1. Soewarno. Tatalaksana penderita
penyakit Megakolon kongenital pada
bayi dan anak dengan prosedur
Duhamel di RSUP Dr. Sardjito.
Naskah Pertemuan Ilmiah alumnus FK
UGM, HUT FK UGM XI dan HUT
RSUP Dr. Sardjito IV. Yogyakarta: FK
UGM; 1986.
2. Swenson O, Sherman JO. Diagnosis of
Congenital Megacolon: an analysis of
501 patient. J Pediatric Surgery.
1973;8:587-94.
3. Kartono D. Penyakit Hirschsprung,
Perbandingan prosedur Swenson dan
Duhamel modifikasi [disertasi].
Jakarta: Universitas Indonesia; 1993.
4. Yoshida Jr C. Hirschsprung Disease.
Department of Diagnosis Imaging
federal of University of Sao Paulo
(UNIFEST); 2004.
5. Kartono D. Penyakit Hirschsprung.
Jakarta: Sagung Seto; 2004.
6. Holschneider A, Ure BM.
Hirschsprung's disease. Dalam:
Ashcraft KW, Murphy JP, Sharp RJ,
dkk, penyunting. Pediatric Surgery.
Edisi ke-3. Philadelphia: Saunders;
2000. p.453-72.
7. Sieber WK. Hirschsprung’s Disease.
Yuliaji Narendra Putra Jurnal Bedah Nasional
50
Dalam: Welch KJ, Randolph JG,
Ravitch MM, dkk, penyunting.
Pediatric Surgery. Edisi ke-4.
Chicago: Year Book Medical Publisher
Inc; 1986. p.995-1019.
8. Santos MC, Giacomantonio JM, Lau
HYC. Primary Swenson pull-through
compared with multiple-stage pull-
through in the neonate. Journal of
Pediatric Surgery. 1999;34:1079-81.
CASE REPORT
Jurnal Bedah Nasional | 51
CASE REPORT OF A NEONATE WITH CONGENITAL SUPERIOR
LUMBAR HERNIA
Stephanus Haryanto Hokardi*, Neil Angelo S. Sael
Department of Surgery, De Los Santos Medical Center, 201 E. Rodriguez Sr. Ave., Quezon City, NCR,
Philippines. *Correspondence: yanto1886@yahoo.com.
ABSTRACT
Background: congenital lumbar hernias are rare. It constitutes to 20% of all lumbar hernias which
is less than 1.5% of all the abdominal wall hernias. There are no more than 50 cases reported in
literature till date. We report a case of congenital lumbar hernia in a preterm female neonate located
on the superior lumbar triangle. Case: a preterm female neonate was born, presented with a mass at
the right lumbar area with a size of 8x8 cm, round, movable with bluish discoloration, well delineated
border, no visible veins, increases in size when the patient cries, and reduces easily. Ultrasonography
revealed a right posterolateral abdominal mass measuring 4.2x2.88x1.59 cm. CT scan revealed right
posterolateral mid-abdominal wall hernia with protrusion and no intestinal obstruction. The patient
underwent exploratory laparotomy, where hernia defect was about 2 cm in diameter in the right
posterior abdominal wall, pararenal area, and just below the 12th rib. The ascending colon and parts of
the ileum were adherent inside the hernia defect at the right lumbar area. Primary closure of the hernia
defect was done by suturing the psoas major and the transversus abdominis and internal oblique
muscles. The postoperative, patient had good bowel movement, no abdominal distention or vomiting.
Feeding was then started and well tolerated. After two weeks follow-up, there were no signs or
symptoms of intestinal obstruction such as nausea and vomiting. Patient is being fed regularly and
passes bowel movement almost 2-3 times a day. Conclusion: appropriate diagnosis of the extent of
the defect through the advent of CT scan and early detection of other congenital anomalies should be
routine in these cases. Open surgery with primary repair is almost always done but we can consider
laparoscopic approach in the future with uncomplicated lumbar hernias.
Keywords: congenital hernia, superior lumbar hernia, surgery.
LAPORAN KASUS NEONATUS DENGAN HERNIA LUMBAR SUPERIOR
KONGENITAL
Stephanus Haryanto Hokardi*, Neil Angelo S. Sael
Departmen Bedah, De Los Santos Medical Center, 201 E. Rodriguez Sr. Ave., Kota Quezon, NCR, Filipina.
*Korespondensi: yanto1886@yahoo.com.
ABSTRAK
Latar belakang: hernia lumbal kongenital jarang terjadi. Ini merupakan 20% dari semua hernia
lumbar yang mana kurang dari 1,5% dari semua hernia dinding abdomen. Tidak ada lebih dari 50
kasus yang dilaporkan dalam literatur hingga saat ini. Kami melaporkan kasus hernia lumbar
kongenital pada neonatus perempuan prematur yang terletak di segitiga lumbar superior. Kasus:
neonatus perempuan prematur lahir, terdapat massa di daerah pinggang kanan dengan ukuran 8x8cm,
bulat, dapat digerakkan dengan warna kebiru-biruan, batas baik, tidak ada vena yang terlihat,
peningkatan ukuran ketika pasien menangis, dan mengecil dengan mudah. Ultrasonografi
menunjukkan massa perut posterolateral kanan berukuran 4,2x2,88x1,59 cm. CT scan menunjukkan
hernia dinding abdomen tengah posterolateral kanan dengan protrusi dan tidak ada obstruksi usus.
Stephanus Haryanto Hokardi Jurnal Bedah Nasional
52
Pasien menjalani laparotomi eksplorasi. Lubang hernia sekitar 2 cm dan lubang berada di dinding
perut posterior kanan, daerah pararenal, dan tepat di bawah tulang rusuk ke-12. Kolon asendens dan
bagian ileum melekat di dalam defek hernia di area lumbar kanan. Penutupan primer defek hernia
dilakukan dengan menjahitkan psoas major dan transversus abdominis dan otot oblikus internal. Pada
pasca operasi, pasien mengalami gerakan usus yang baik, tidak ada distensi abdomen atau muntah.
Makan dimulai dan ditoleransi baik. Setelah dua minggu evaluasi, tidak ada tanda-tanda atau gejala
obstruksi usus seperti mual dan muntah. Pasien diberi makan secara teratur dan buang air besar 2-3
kali sehari. Simpulan: diagnosis yang tepat mengenai derajat defek melalui CT scan dan deteksi dini
anomali kongenital lainnya harus rutin dalam kasus ini. Operasi terbuka dengan perbaikan primer
hampir selalu dilakukan tetapi kita dapat mempertimbangkan pendekatan laparoskopi di masa depan
dengan hernia lumbar tanpa komplikasi.
Kata kunci: hernia kongenital, hernia lumbar superior, operasi.
INTRODUCTION
Congenital lumbar hernias are rare
abdominal wall hernias in infants and in
children. Approximately 10% are
congenital and the majority are unilateral.1
They are divided into three types
depending on the site. There are superior,
which occur through the superior lumbar
triangle (Grynfelt-Lesshaft triangle),
inferior, which occur through the inferior
lumbar triangle (Petit), and combined.
Most common location of a congenital
lumbar hernia is in the inferior triangle.1,2
Less than 50 cases of congenital lumbar
hernias are reported in literature till
date.2,3,4 These hernias may contain colon,
small intestine, spleen, or liver and all
have been reported.4,5 CT Scan has
become the imaging modality of choice in
confirming the diagnosis.6 Lumbar hernias
have a 25% risk of incarceration and an
8% risk of strangulation, making surgery a
reasonable option for management.5
Congenital lumbar hernias when
diagnosed, are frequently associated with
other anomalies. Lumbocostovertebral
syndrome is one example wherein there is
a presence of hemivertebra, congenital
absence of ribs, anterior mylomeningocele,
and hypoplasia of anterior abdominal wall
presenting as congenital lumbar hernia.
Other anomalies associated are anorectal
malformations, hydrocephalus, congenital
diaphragmatic hernia, caudal regression
syndrome, pelvoureteric junction
obstruction, cloacal exstrophy, an absent
kidney, or meningomyelocele. Diagnosis
of a congenital lumbar hernia must prompt
one to investigate further for these
anomalies.1,2 We report a case of
congenital lumbar hernia in a preterm
female neonate located on the superior
lumbar triangle.
CASE REPORT
A preterm female neonate was born to a
20-year-old G2P2 (1102) via Cesarean
Section at 36 weeks gestation. She
presented with a mass at the right lumbar
area with a size of 8x8 cm, round, movable
with bluish discoloration, well delineated
border, no visible veins and increases in
size when the patient cries and reduces
easily.
Ultrasonography revealed a right
posterolateral abdominal mass measuring
4.2x2.88x1.59 cm. A dorsal hernia is
considered. There were no signs of bowel
obstruction. Mild pelvocaliectasia was
noted on the right. The liver, gallbladder,
Volume 2 ● Number 2 ● Juli 2018 Case Report of Neonate with Congenital Superior
53
pancreas, spleen, left kidney and urinary
bladder were unremarkable. There was no
hepatobiliary duct ectasia nor
demonstrable suprarenal or pelvic mass.
The scout film of the abdomen was also
done which showed displaced bowel loops
in the right lower quadrant.
CT scan (Figure 1a, 1b) revealed
minimal to moderate hepatomegaly,
normal spleen, multiple surface
calcification, sequel of an intrauterine
granulomatous infection, right
posterolateral mid-abdominal wall hernia
with protrusion, no intestinal obstruction,
small umbilical hernia. There were no
signs of obstruction, no vomiting nor
abdominal distention until on the 5th day of
life, the patient underwent exploratory
laparotomy. Patient was placed in supine
position and a transverse incision was
made on the abdominal right upper
quadrant and carried down to the
peritoneum. There was multiple mucoid
meconium noted on bowels with minute
calcifications. Meconium peritonitis was
considered due to the prolonged standing
of the congenital lumbar hernia. Bowel run
was done and noted no signs of
perforation. The bowels were noted to be
inflamed in different segments.
Adhesiolysis was done with sharp and
blunt dissection. Hernia defect was about 2
cm in diameter and is noted to be in the
right posterior abdominal wall, pararenal
area, and just below the 12th rib. This
marks the superior lumbar triangle. The
ascending colon and parts of the ileum
were adherent inside the hernia defect at
the right lumbar area. Mobilization of the
ascending colon and cecum was done then
the bowels were delivered from the hernia
defect. Primary closure of the hernia defect
was done by suturing the psoas major and
the transversus abdominis and internal
oblique muscles with 6 stitches of Prolene
3-0 simple interrupted suturing. Primary
closure was considered due to the presence
of meconium peritonitis. Interval
appendectomy was done. The rest of the
abdominal wall was closed in layers.
Patient tolerated the procedure well.
Figure 1. (a,b) CT scan imaging of the right
posterolateral abdominal wall hernia. Note only
small bowel loops are located in the hernia defect;
(c,d) pre-operative images of the congenital lumbar
hernia; (e) intraoperative image of the hernia
defect; (f) post-operative image.
The immediate postoperative period
was uneventful. On the second
postoperative day, patient had 3 episodes
of bowel movement. No abdominal
distention or vomiting was noted. Repeat
scout film of the abdomen was done which
revealed non-dilated bowel loops in the
abdominal cavity. No free air noted.
Feeding was then started and well
tolerated. The rest of the hospital stay was
unremarkable. Antibiotics were completed
and on the 16th day of life, baby was sent
home with stable vital signs, good suck,
and active.
After two weeks follow-up, there were
no signs or symptoms of intestinal
Stephanus Haryanto Hokardi Jurnal Bedah Nasional
54
obstruction such as nausea and vomiting.
Patient is being fed regularly and passes
bowel movement almost 2-3 times a day.
DISCUSSION
The most common site of congenital
lumbar hernias is found in the inferior
lumbar triangle or Petit triangle.7 Acquired
or traumatic lumbar hernias are more
frequently seen in the superior lumbar
triangle because it is the thinnest area in
the lateral and posterior abdominal wall.
Hernias from this location result from
direct trauma, flank incision, or an
abscess.5 In our case, the patient presented
with a congenital lumbar hernia at the
superior lumbar triangle which is rare.8
During embryologic development,
weakening of the area of the aponeuroses
of the layered abdominal muscles that
derive from somatic mesoderm, which
invades the somatopleure, may potentially
lead to lumbar hernias.5
The aim of the surgery is to reduce the
hernia sac, repair the defect and to
strengthen the weakened posterior
abdominal wall. It could be done by
simple anatomical closure, overlapping of
the aponeurosis, or use of prosthetic
meshes or laparoscopic mesh repair in
cases of uncomplicated lumbar hernias.3,6
Elective surgical repair is suggested at
any early age to prevent incarceration and
strangulation.9 Intervention should be done
before 12 months because the hernia defect
may enlarge with growth making primary
direct closure with surrounding tissue
difficult.9 Because of the rarity of
congenital lumbar hernia, appropriate
surgical procedures are still controversial.
Open repair has been performed in most
patients.1,2,5,10 and laparoscopic repair is
preserved with small uncomplicated
hernias. In the present case, the hernial
defect was large, and surrounding
abdominal wall muscles were hypoplastic.
Primary closure was also intended due to
the presence of meconium peritonitis and
mesh repair was not considered.
Aside from primary closure, muscle
grafts could be done by using a fascia lata
graft.9 Currently, laparoscopy is also being
performed in patients with congenital
lumbar hernia. It could be approached
either transabdominally or
extraperitoneally and minimal reports of
recurrence is seen.2,5,6,9,11
CONCLUSION
Congenital lumbar hernia is one of the
rare types of hernias reported in neonates;
and occasionally in older children.
Appropriate diagnosis of the extent of the
defect through the advent of CT scan and
early detection of other congenital
anomalies should be routine in these cases.
Since there is almost to none regarding
Philippine literature on the practice of
congenital lumbar hernia, we can
recommend further studies for the surgical
approaches in this anomaly. Open surgery
with primary repair is almost always done
but we can consider laparoscopic approach
in the future with uncomplicated lumbar
hernias.
REFERENCES
1. Al-Salem AH. Abdominal Wall
Hernias and Hydroceles. In: Al-Salem
AH, editor. An Illustrated Guide to
Pediatric Surgery. Switzerland:
Springer; 2014. p.15-27.
2. Esposito C, Settimi A, De Marco M, et
al. Congenital Lumbar Hernia: Two
Case Reports and a Review of the
Literature. Journal of Pediatric
Surgical Specialties. 2009;3:40-2.
Volume 2 ● Number 2 ● Juli 2018 Case Report of Neonate with Congenital Superior
55
3. Wakhlu A, Wakhlu AK. Congenital
lumbar hernia. Pediatr Surg Int.
2000;16:146-8.
4. Peláez Mata DJ, Alvarez Munoz V,
Fernandez Jimenez I, et al. Congenital
lumbar hernia. Cir Pediatr.
1998;11:126-8.
5. Stamatiou D, Skandalakis JE,
Skandalakis LJ, et al. Lumbar hernia:
surgical anatomy, embryology, and
technique of repair. Am
Surg. 2009;75:202-7.
6. Dakin GF, Kendrick ML. Challenging
Hernia Locations: Flank Hernias. In:
Jacob BP, Ramshaw B, editors. The
SAGES Manual of Hernia Repair. New
York: Springer; 2013. p. 531-40.
7. Pachani AB, et al. A Primary
Idiopathic Superior Lumbar Triangle
Hernia with Congenital Right
Scoliosis: A Rare Clinical Presentation
and Management. Int J Appl Basic
Med Res. 2011;1:60-2.
8. Tavares-de la Paz LA, Martínez-Ordaz
JL. Lumbar hernia. Case report and
literature review. Cir Cir. 2007;75:381-
4.
9. Morita K, Miyano G, Nouso H, et al.
Laparoscopic repair for a congenital
lumbar hernia with free fascia lata graft
reinforcement. J Pediatr Surg Case
Rep. 2014;2:101-3.
10. Sharma A, Pandey A, Rawat J, et al.
Congenital lumbar hernia: 20 years'
single centre experience. J Paediatr
Child Health. 2012;48:1001-3.
11. Moreno-Egea A, Baena EG, Calle MC,
et al. Controversies in the Current
Management of Lumbar Hernias. Arch
Surg. 2007:142:82-8.
CASE REPORT
56 | Jurnal Bedah Nasional
BILATERAL GIANT FIBROADENOMA OF THE BREAST: A CASE
REPORT
Jasmine Stephanie Christian1, Putu Anda Tusta Adiputra2*, INW Steven Christian2
1General Surgery Training Programme, Faculty of Medicine Udayana University, Sanglah General Hospital,
Denpasar, Bali.
2Surgical Oncology Division, Department of Surgery, Faculty of Medicine Udayana University, Sanglah
General Hospital, Denpasar, Bali. *Correspondence: andatusta@unud.ac.id.
ABSTRACT
Background: fibroadenoma of the breast is one of the ANDI (Aberration of the Normal
Development and Involution of the Breast) groups. Benign breast disorders are common in females
younger than 30 years, but such masses are not common in juvenile or pre-menarche age groups. The
exact pathological diagnosis can be investigated after surgery. For treatment, surgical procedures are
needed to remove the lump. Case: a 30-year-old girl visited the outpatient clinic with psychological
distress due to the presence of lumps in her both breasts. The lumps have been growing bigger and
bigger in the last 3 years. On the physical examination both breasts are huge. FNAB showed the
interpretation of atypical ductal hyperplasia and excisional biopsy examination confirmed the
presence of bilateral giant fibroadenoma of breast. In this patient, the treatment was carried out in
two-staged surgery due to her initial rejection to undergo total breast lumps and tissue removal.
Several lumps still remained in the right breast which was indicated for the second surgery. The
following surgery was done to remove all of the lumps and breast tissue with reduction breast surgery.
Conclusion: conducting breast surgery on the young girl or un-married is not easy as many physical
and psychology problems should be considered. Therefore, conducting breast surgery, in this case,
should be performed wisely as the recurrence of the lumps was still possible.
Keywords: multiple giant fibroadenoma, surgery, psychology.
FIBROADENOMA RAKSASA BILATERAL PADA PAYUDARA: SEBUAH
LAPORAN KASUS
Jasmine Stephanie Christian1, Putu Anda Tusta Adiputra2*, INW Steven Christian2
1Program Studi Ilmu Bedah, Fakultas Kedokteran Universitas Udayana, Rumah Sakit Umum Pusat Sanglah,
Denpasar, Bali.
2Divisi Bedah Onkologi, Departemen Ilmu Bedah, Fakultas Kedokteran Universitas Udayana, Rumah Sakit
Umum Pusat Sanglah, Denpasar, Bali. *Korespondensi: andatusta@unud.ac.id.
ABSTRAK
Latar Belakang: fibroadenoma payudara adalah salah satu kelompok ANDI (Aberration of the
Normal Development and Involution of the Breast), yang mencakup spektrum luas dari penyakit
payudara benigna. Gangguan payudara jinak umumnya terjadi pada wanita yang kurang dari 30 tahun,
tetapi massa tersebut tidak umum ditemui pada kelompok usia remaja atau pra-menarche. Giant
fibroadenoma didefinisikan sebagai benjolan berukuran lebih dari 5 cm. Fibroadenoma dapat terjadi
pada satu atau kedua payudara dan tumbuh perlahan. Prosedur bedah diperlukan untuk mengangkat
massa. Diagnosis patologis dapat ditegakkan setelah operasi. Kasus: seorang wanita 30 tahun
mengunjungi klinik rawat jalan dengan tekanan psikologis karena adanya benjolan di kedua
payudaranya. Benjolan tumbuh semakin besar dalam 3 tahun terakhir. Pada pemeriksaan fisik,
ditemukan massa di seluruh kuadran payudara kanan dan kiri dengan berbagai ukuran. Pemeriksaan
Volume 2 ● Number 2 ● Juli 2018 Bilateral Giant Fibroadenoma of the Breast
57
FNAB menunjukkan gambaran hiperplasia duktus atipikal, kecurigaan ke arah keganasan.
Pemeriksaan biopsi eksisi mengkonfirmasi adanya fibroadenoma raksasa bilateral pada payudara.
Pada pasien ini, dilakukan operasi dua tahap karena pasien menolak dilakukan operasi total kedua
payudara. Beberapa benjolan masih tersisa di payudara kanan dan direncanakan untuk operasi kedua.
Operasi selanjutnya dilakukan untuk mengangkat seluruh masa dan jaringan payudara. Simpulan:
tindakan operasi payudara pada wanita muda yang belum menikah bukanlah hal yang mudah karena
banyak masalah fisik dan psikologis yang harus dipertimbangkan. Oleh karena itu, melakukan operasi
payudara, dalam hal ini, harus dilakukan dengan bijaksana karena kekambuhan masih dapat terjadi.
Kata kunci: fibroadenoma raksasa multipel, operasi, psikologi.
INTRODUCTION
Giant fibroadenoma of the breast is
defined as a fibroadenoma larger than 5
cm1 which is most common find in young
women.2 Fibroadenoma mamma is the one
of the groups of ANDI (Aberration of the
Normal Development and Involution of
the Breast).3 ANDI is an all-encompassing
term that is used to describe a wide
spectrum of Benign Breast Disease.
Fibroadenomas have previously been
regarded as benign neoplasms, but should
now be considered as an aberration of
normal development.4
It may occur during the time of the
normal development of the breast. Most
normal development of both breasts are
range up to 25 years of age. The breast has
reached its major development by 20 years
of age and will usually begin to undergo
atrophic changes in the fifth decade of
life.5 Benign breast disorders are common
in females younger than 30 years, but such
masses are not common in juvenile or
premenarchal age groups. Giant
fibroadenoma may occur in one or both
breast and growing rapidly. Although most
fibroadenomas are benign, the presence of
a mass can cause considerable anxiety
because of the concern for cancer. These
benign tumors arise from the epithelium
and stroma of the terminal duct-lobular
unit. In one consecutive series of patients,
44% of fibroadenomas occurred in
postmenopausal women.6 For treatment,
surgical procedures are needed to remove
the lump. The exact pathological diagnosis
can be investigated after surgery.
CASE REPORT
A 30-year-old un-married girl came
with the psychological distress due to her
both breast growing bigger and bigger in
last 3 years as well as shown on the
picture. Family history was not relevant.
On the physical examination, both breasts
are huge and swelling with multiple lumps
varies in size and involving of the whole
quadrant of the breast. Her right breast
consists of ten lumps involving the whole
quadrant of the breast with the size of the
lumps are varies more than 5 cm. The left
breast consists of nine lumps involving of
the whole quadrant. The lumps were solid,
painless, and moveable. The skin over the
both breasts were hyperpigmented and
showed dilatation of peripheral vascular.
Normal of breast nipple and wide
pigmented areolar of the breast are also
noted. In general, patient is wellbeing. The
blood laboratory test is in normal limit.
Chest x-ray shown normal, no evidence of
lesion. Breast ultrasonography
examination is not provided as well as
mammography. There was no evidence of
both axillary lymphadenopathies.
Jasmine Stephanie Christian Jurnal Bedah Nasional
58
Fine needle aspiration biopsy (FNAB)
examination showed an interpretation for
atypical ductal hyperplasia suspicious for
malignancy. Incisional biopsy showed the
result of bilateral fibroadenoma of breast.
There was no evidence of malignancy.
Patient underwent two-staged surgery due
to the rejection of taking the whole lumps
for may all breast tissue should be
removed. So, several lumps of the right
breast are still remained (Figure 1).
Majority of normal breast tissue were
removed during excision of the lumps.
Normal breast tissue also being removed
and the breast became flat. The surgery
was stopped because patient dan family
need to be informed. Patient was fully
distressed and rejected any kinds of
surgery later.
Figure 1. Clinical feature in the first presentation.
The patient presented with bilateral giant
fibroadenoma of breast (left) and already get an
operation excisional biopsy on her right breast
(right).
Three months later, she came to the
hospital to make another surgery because
her breast became bigger (Figure 2). The
next surgery is to remove the all the lumps
and breast tissue as well (reduction breast
surgery) in the left breast. But the several
lumps on the right breast still remain.
Figure 2. The lumps are getting bigger and worse
after 3 months and next surgery is removed the all
the lumps and breast tissue as well (reduction
breast surgery) in the left breast.
DISCUSSION
The diagnosis and treatment of giant
fibroadenoma can be a challenge for
physicians. As fibroadenoma is considered
a benign lesion without tendency to
malignant degeneration, surgical treatment
is generally not considered necessary to
treat fibroadenoma. However, because of
their size, giant fibroadenoma could result
in local problems including breast
asymmetry or deformity. It can become
cosmetic problem for the affected patient
with serious psychological effects. In fact,
patient is often extremely concerned about
potential cosmetic changes to the
appearance of the breast after surgery. In
the serious case, while some authors
recommend reduction mammaplasty with
inverted-incision technique to resect large
fibroadenomas, others recommend using a
Volume 2 ● Number 2 ● Juli 2018 Bilateral Giant Fibroadenoma of the Breast
59
more cosmetically appealing incision.7,8
The surgical management of giant
fibroadenoma is still a matter of
controversial debate in the literature. The
necessity for excision is not disputed in
patients presenting with breast deformity
or suffering from local problems such as
venous congestion, pressure necrosis and
even occasionally ulcer.
CONCLUSION
Many problems should be considered
before performing breast surgery in un-
married young girl. Although radical
mastectomy will reduce the recurrence, the
patient’s satisfaction and psychological
problems can be surfaced.
REFERENCES
1. Hille-Betz H, Klapdor R, Henseler H,
et al. Treatment of Giant
Fibroadenoma in Young Women:
Results after Tumor Excision without
Recontructive Surgery. Geburtshilfe
Frauenheilkd. 2015;75:929-34.
2. Chang DS, McGrath MH.
Management of benign tumors of the
adolescent breast. Plast Reconstr Surg.
2007;120:13e-19e.
3. Robert E. Mansel. Management of
Breast Pain. In: Harris JR, Lippman
ME, Morrow M, et al. editor. Disease
of The Breast. 5th Edition.
Philadelphia: Wolters Kluwer Health;
2014. p.51-7.
4. Purushotham AD, Britton P, Bobrow
L. In: Borgen PI, Hill ADK, editors.
Benign Breast Disease. Texas: Landes
Bioscience; 2000. p.35-40.
5. Osborne MP, Boolbol SK. Breast
Anatomy and Development. In: Harris
JR, Lippman ME, Morrow M, et al.
editor. Disease of The Breast. 5th
Edition. Philadelphia: Wolters Kluwer
Health; 2014. p.3-14.
6. Bleicher RJ. Management of the
Palpable Breast Mass. In: Harris JR,
Lippman ME, Morrow M, et al. editor.
Disease of The Breast. 5th Edition.
Philadelphia: Wolters Kluwer Health;
2014. p.29-37.
7. Biggers BD, Lamont JP, Etufugh CN,
et al. Inframammary approach for
removal of giant juvenile
fibroadenomas. J Am Coll Surg.
2009:208:e1-4.
8. Jacob MM. Application of reduction
mammaplasty in treatment of giant
breast tumour. Br J Plast Surg.
2000;53:265-6.
CASE REPORT
60 | Jurnal Bedah Nasional
GOITER MULTINODUL DENGAN PELEBARAN KE RETROSTERNAL:
LAPORAN KASUS
Gede Budhi Setiawan
Divisi Bedah Onkologi, Departemen Ilmu Bedah, Fakultas Kedokteran Universitas Udayana, Rumah Sakit
Umum Pusat Sanglah, Denpasar, Bali. Korespondensi: dhiwans@hotmail.com.
ABSTRAK
Latar belakang: goiter multinodul retrosternal merupakan massa tiroid yang meluas sampai
sternum hingga mengisi ruang inlet torakal. Dapat disebut retrosternal multinodul tiroid apabila massa
tiroid lebih dari separuhnya berada di dalam inlet torakal. Pembedahan merupakan pilihan utama.
Pilihan pembedahan yaitu dengan insisi collar dan sternotomi, clamshell insisi, bahkan sampai
thorakotomi. Kasus: dilaporkan kasus pasien wanita 62 tahun dengan goiter multinodul hingga
retrosternal. Hasil USG menunjukkan struma diffusa kiri dengan kista multipel kompleks pada tiroid
kanan. CT Scan didapatkan massa tiroid multipel kiri dengan gambaran nekrosis sentral dan
kalsifikasi egg shell, dan infiltrasi toraks. Dari foto toraks didapatkan massa di leher kiri yang meluas
hingga rongga toraks yang tampak mendesak trakea ke kanan. Telah dilakukan total tiroidektomi,
sternotomi, dan trakeostomi. Simpulan: Operasi pengangkatan goiter substernal dapat dilakukan
dengan pendekatan leher di sebagian besar pasien tetapi dalam beberapa kondisi, pasien melakukan
pendekatan leher dan sternotomi.
Kata kunci: goiter multinodul retrosternal, tiroidektomi, sternotomi.
MULTINODULAR GOITER WITH RETROSTERNAL EXTENSION: CASE
REPORT
Gede Budhi Setiawan
Surgical Oncology Division, Department of Surgery, Faculty of Medicine Udayana University, Sanglah General
Hospital, Denpasar, Bali. Correspondence: dhiwans@hotmail.com.
ABSTRACT
Background: retrosternal goiter refers to the thyroid mass grows along dermal sternum from the
neck to the substernal portion, descending below the thoracic inlet. The current accepted definition of
retrosternal goiter is thyroid gland with more than 50% of its mass located below the thoracic inlet.
Surgery is the treatment choice of retrosternal goiter. Nowadays, several surgical options have been
used for the surgical resection of this type of goiters, which is related to the size of the gland and the
location, such as collar neck incision or/and sternotomy, clamshell incision or even thoracotomy.
Case: we reported female 62-year-old with multinodular goiter with retrosternal extension.
Ultrasonography showed diffuse mass of left thyroid and multiple complex cyst of right thyroid. CT
Scan examination revealed multiple mass of left thyroid with central necrotic and egg shell
calcification, and infiltrated thorax. Thorax x-ray showed mass derived from left neck to intra thoracic
cavity and there is deviation of trachea to the right. Fine needle aspiration biopsy shows colloid
nodule and follicular neoplasm. We perform total thyroidectomy, sternotomy, and tracheostomy.
Conclusion: Surgical removal of substernal goiters can be performed by a cervical approach in the
majority of patients but in some condition, patient performed cervical and sternotomy approach.
Keywords: retrosternal multinodular goiter, total thyroidectomy, sternotomy.
Volume 2 ● Number 2 ● Juli 2018 Multinodular Goiter with Retrosternal Extension
61
PENDAHULUAN
Goiter retrosternal mengacu pada massa
tiroid yang tumbuh sepanjang sternum dari
leher ke bagian substernal, turun di bawah
inlet torakal. Tiroid retrosternal mencakup
kelenjar tiroid dengan lebih dari 50% dari
massa yang terletak di bawah inlet torakal.
Pembedahan adalah pengobatan pilihan
untuk tiroid retrosternal. Saat ini, pilihan
operasi yang berbeda telah digunakan
untuk reseksi jenis goiter ini, yang
berkaitan dengan ukuran kelenjar dan
lokasinya, seperti sayatan leher collar atau
dengan sternotomi, sayatan clamshell, atau
bahkan torakotomi.1
Meskipun sebagian besar goiter
retrosternal adalah ekstensi dari leher,
goiter intra torakal murni, mungkin terjadi.
Goiter retrosternal lebih cenderung di sisi
kiri dan sangat jarang meninggalkan sisi
kiri kemudian turun ke sisi kanan toraks
yang disebut "crossed substernal goiter".
Dengan beberapa pengecualian, goiter
yang besar mungkin dioperasi dengan
pendekatan servikal tetapi sekitar kurang
dari 2% pasien membutuhkan pendekatan
servikal dan sternotomi.1 Dalam artikel ini
kami melaporkan kasus langka wanita
berusia 62 tahun yang memiliki goiter
multinodular dengan ekstensi retrosternal
yang sangat besar. Tumor diangkat melalui
pembedahan dengan pendekatan servikal
dan sternotomi.
LAPORAN KASUS
Seorang wanita 62 tahun datang ke
pusat kanker kami dengan keluhan utama
adanya massa leher yang besar sejak 20
tahun yang lalu. Pemeriksaan lokal
mengungkapkan pembengkakan kelenjar
tiroid yang difusa (Gambar 1).
Pemeriksaan radiologis dilakukan
(Gambar 2) dan ultrasonografi
menunjukkan adanya massa difusa tiroid
kiri dan kista kompleks multipel tiroid
kanan. Computed tomography (CT) scan
leher menunjukkan adanya lesi multipel
padat pada tiroid kiri berukuran 5,6 x 5,6
cm dengan peningkatan kontras dan
nekrosis sentral. Lesi lain berukuran 6,0 x
4,6 cm dengan kalsifikasi egg shell dan
peningkatan kontras. Nodul multipel kanan
dengan peningkatan kontras, diameter 1,7
cm. Massa memanjang ke inlet torakal dan
menggeser trakea ke kanan. Tidak ada
limfadenopati servikal.
Gambar 1. Gambaran klinis
Gambar 2. Pemeriksaan ultrasonografi dan CT
scan leher.
X-ray toraks (Gambar 3) menunjukkan
massa yang berasal dari leher kiri ke intra
toraks dan ada pergeseran trakea ke kanan.
Hasil biopsi aspirasi jarum halus
menunjukkan nodul koloid dan neoplasma
folikel. Uji fungsi tiroid (FT4 dan TSH)
Gede Budhi Setiawan Jurnal Bedah Nasional
62
dilakukan, yang mana FT4 2,00 ng/dL dan
TSH 0,03 µIU/mL.
Gambar 3. X-ray toraks.
Gambar 4. Posisi kelenjar tiroid.
Pembedahan dilakukan dengan anestesi
umum dan sayatan collar dilakukan 2 cm
di atas sternum. Otot strap kemudian
dipisahkan sampai tampak kelenjar tiroid.
Kelenjar tiroid kiri dimobilisasi lebih
dahulu kemudian diikuti dengan kelenjar
tiroid kanan. Kedua saraf laringeal rekuren
diidentifikasi dan diamankan (Gambar 4).
Setelah tiroid kiri dibebaskan, dilanjutkan
dengan tiroid kanan. Sternotomi dilakukan
untuk enukleasi massa retrosternal.
Kelenjar tiroid bersama dengan massa
retrosternal dipotong (Gambar 5).
Trakeostomi dilakukan untuk menjaga
patensi saluran napas. Pada kasus ini
pasien keluar rumah sakit setelah
menjalani perawatan 21 hari.
Gambar 5. Kelenjar tiroid dan massa retrosternal
sudah dieksisi.
DISKUSI
Pembedahan adalah pengobatan pilihan
untuk goiter retrosternal dengan atau tanpa
gejala klinis. Definisi gioter retrosternal
tidak seragam dan sering bervariasi di
antara penulis. Menurut Katlic dan rekan-
rekan goiter retrosternal adalah ketika
lebih dari 50% dari massa terletak jauh ke
outlet toraks. Pasien sering mengeluh
pertumbuhan massa yang lambat dan
pertumbuhan massa progresif biasa terjadi
pada dekade kehidupan ke-5 atau ke-6.2
Goiter retrosternal dapat menyebabkan
gangguan pernapasan, disfagia, kompresi
vaskular, dan bahkan menimbulkan
kematian mendadak.3-5
Kondisi tersebut tidak jarang dan risiko
keganasan antara 3-21%. Operasi
pengangkatan goiter substernal dapat
dilakukan dengan pendekatan servikal
pada sebagian besar pasien. Telah
dilaporkan bahwa ahli bedah yang ahli,
dengan pengalaman operasi tiroid yang
baik, perlu melakukan pendekatan ekstra
servikal pada 2-5% tiroidektomi untuk
goiter substernal, tetapi beberapa penulis
telah melaporkan kejadian sternotomi
terjadi pada 29% pasien. CT scan telah
menjadi standar emas investigasi radiologi
pra operasi untuk penilaian massa tiroid
dengan ekstensi substernal dan
hubungannya dengan struktur yang
berdekatan, dan juga dapat digunakan
dalam menentukan pasien yang
kemungkinan akan memerlukan
pendekatan toraks. Beberapa orang telah
Volume 2 ● Number 2 ● Juli 2018 Multinodular Goiter with Retrosternal Extension
63
meneliti faktor-faktor yang meningkatkan
kemungkinan sternotomi.4
Flati dkk menyatakan bahwa sternotomi
tidak dapat dihindari ketika gondok lebih
dari 70% berada di dalam mediastinum.
Beberapa penulis telah menyatakan operasi
revisi sebagai indikasi yang mungkin
untuk sternotomi.5 Sternotomi hanya boleh
digunakan ketika goiter tidak dapat
diekstraksi dari toraks dengan "manuver
lembut", juga dalam kasus semua gondok
yang berulang dan menyimpang.4 Di sisi
lain sternotomi diperlukan ketika traksi
yang berlebihan diperlukan selama
operasi, ketika perluasan yang paling
inferior dari nodul dapat dipalpasi, dalam
kasus operasi revisi, adanya kompresi
trakea akut, obstruksi vena yang parah,
keganasan, dan diagnosis preoperatif yang
tidak jelas. Banyak rekomendasi
sternotomi untuk nodul substernal ganas,
goiter mediastinum posterior dengan
ekstensi kontralateral, goiter mediastinum
dengan suplai darah dari mediastinum,
gondok yang menyebabkan sindrom vena
cava superior, kasus operasi revisi, jika
eksisi massa toraks yang sulit, perdarahan
yang signifikan, dan ketika diameter nodul
mediastinum secara signifikan melebihi
diameter inlet torakal.4
SIMPULAN
Goiter retrosternal cukup umum dan
dapat diagnosis secara klinis dan
radiologis yang jelas. Pembedahan
merupakan pilihan utama. Operasi
pengangkatan goiter substernal dapat
dilakukan dengan pendekatan servikal di
sebagian besar pasien tetapi dalam
beberapa kondisi pasien melakukan
pendekatan servikal dan sternotomi. CT
scan dapat digunakan dalam menentukan
pasien yang kemungkinan akan
membutuhkan pendekatan toraks.
DAFTAR PUSTAKA
1. Mohammad A, Rajnish N, Shirsendu
R, et al. Huge Nodular Goitre with
Retrosternal Extension-A Rare Case
Report. J Otolaryngol ENT Res.
2017;7:00215.
2. Wang L-S. Surgical management of a
substernal goiter. Formosan Journal of
Surgery. 2012;45:41-4.
3. Sheng YR, Xi RC. Surgical approach
and technique in retrosternal goiter:
Case report and review of the
literature. Ann Med Surg (Lond).
2016;5:90-2.
4. Coskun A, Yilridim M, Erkan N.
Substernal Goiter: When is Sternotomy
Required? Int Surg. 2014;99;419-25.
5. Rodrigues J, Furtado R, Anant R, et al.
A rare instance of retrosternal goitre
presenting with obstructive sleep apnea
in a middle-aged person. Int J Surg
Case Rep. 2013;4:1064-6.
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