Ramakrishnapillai Padmakumar1, Aravind Balakrishnan2, Madhukara Pai3, Kevin J Chiramel4, Farish Shams5, Premna Subin6

Abstract

Aim and objective: To show the advantage of endoscopic approach for lateral ectopic thyroid removal.

Background: Ectopic thyroid tissue lateral to midline is very rare. Because of its unusual location, lateral ectopic thyroid gland can cause diagnostic difficulties when diseased.

Case description: Here we are presenting a case of a male patient with submandibular ectopic thyroid tissue with multinodular goiter and absent thyroid tissue in normal anatomic site. He underwent endoscopic-assisted total thyroidectomy (endoscopic thyroidectomy). This technique for ectopic thyroid removal has not been reported in the literature so far.

Conclusion: Endoscopic approach for removal of the diseased gland will allow for a magnified view of the adjoining structures and better cosmesis for the patient.

Clinical significance: Lateral ectopic thyroid should be in differential diagnosis of lateral neck swelling.

Keywords: Ectopic thyroid, Endoscopic, Endoscopic thyroidectomy, Minimal access surgery, Submandibular region, Endoscopic Ectopic Thyroidectomy.

Background

Ectopic thyroid tissue usually occurs in the midline and that too most commonly in cervical region (lingual 90%).1–4 Its prevalence is approximately 1/100,000 to 1/300,000.5 The remaining ectopic thyroid glands (10%) can be found in infrahyoid, submandibular, prelaryngeal, mediastinum, esophagus, heart, diaphragm, and parapharyngeal regions. In most of the cases, the ectopic thyroid gland will be the only functioning gland. Ectopic thyroid tissue lateral to midline is very rare. These lateral ectopic thyroid tissues when diseased may lead to difficulty in diagnosis due to its unexpected location. Here we are going to present a case of a male patient with submandibular ectopic thyroid tissue with multinodular goiter and absent normal tissue in normal anatomic site. He underwent endoscopic-assisted total thyroidectomy. This technique for ectopic thyroid removal has not been reported in literature so far.

Case Description

A 58-year-old male patient presented to our department with complaints of a swelling in the upper neck on left submandibular region for about 6 months.

It was associated with rapid increase in size. There were no other associated symptoms. On examination, a 5 × 3 cm painless swelling was noted in the left submandibular region. The lump was soft in consistency. Thyroid tissue was not palpable in the normal anatomical location (Fig. 1).

Ultrasonography revealed a well-circumscribed heterogeneously hyperechoic nodule in the left submandibular region with absent thyroid gland in the thyroid bed—possibly ectopic thyroid nodule. Fine-needle aspiration biopsy of the swelling was taken which revealed colloid goiter. Thyroid scintigraphy revealed an area of increased radionuclide uptake in the submandibular region and no radionuclide uptake was seen in the neck in the thyroid bed.

Preoperative thyroid hormones and biochemical tests were normal. As gland was enlarging rapidly in size, endoscopic-assisted complete removal of ectopic thyroid tissues was done under general anesthesia. Initially, the gland was approached from left axillary breast ports. We dissected the gland from the surrounding tissues by endoscopic method. For retrieval of specimen, we put a small skin incision in the submandibular region and the specimen was removed in toto (Fig. 2). This helped in significantly minimizing the scar in cervical region.

Postoperative period was uneventful, and he was discharged on third postoperative day. Thyroxine 100 μg was started, as there was no other functioning thyroid gland. Histopathology report showed
features consistent with multinodular colloid goiter.

Fig. 1: Extended neck showing submandibular ectopic thyroid gland

Fig. 2 – Ectopic thyroid in relation with surrounding structures

Discussion

Ectopic thyroid in the submandibular region was first described by Helidonis et al.6 They speculated that the ectopic thyroid has a parahyoid location and because of its dimension resembled a submandibular gland anomaly. Abnormalities of thyroid gland during embryologic development and migration may result in ectopic thyroid gland. Normally, migration of the thyroid gland is from the foramen cecum to the pretracheal position.7 In addition to normal migration pathway of the thyroid gland, ectopic thyroid tissue can be seen even in mediastinal, intracardiac, gastrointestinal, and intraperitoneal locations.4,8,9 Ectopic thyroid tissue is mostly (90%) localized in sublingual position.

Asymptomatic ectopic thyroid tissue may become symptomatic, particularly in the adolescence and pregnancy period due to increase in thyroid-stimulating hormone level and due to thyroid tissue hyperplasia.10,11 All diseases that involve thyroid tissue in its normal location can also involve ectopic thyroid tissue. The differential diagnosis should include thyroglossal duct cyst, hyperplastic lymphoid tissue, lymphangioma, fibroma, lipoma, dermoid cyst, squamous cell carcinoma, minor salivary gland tumor, lymphoma, and vascular tumors.12,13

Ultrasonography (USG), scintigraphy, computerized tomographic scan (CT), and magnetic resonance imaging (MRI) are the methods that can be used in the diagnosis. Thyroid scintigraphy is a sensitive and specific method in determining that thyroid gland is not in its normal location.13 USG and CT are beneficial in the diagnosis but have low sensitivity and specificity. In MRI, ectopic tissue is observed to be iso- or hyperintense compared to muscles. 13 In addition to imaging of the normal thyroid tissue, thyroid
scintigraphy is also important to show the functions of the lingual thyroid tissue. In our case, we performed USG followed by FNAC and then thyroid scintigraphy was performed for confirmation of our diagnosis. All the surgeries performed for lateral ectopic thyroid so far has been by open method. We performed an endoscopic assisted total thyroidectomy which has not been reported in literature so far. Endoscopic approach for removal of the diseased thyroid gland will give a magnified view of the adjoining structures and better cosmesis for the patient.

Conclusion

Lateral ectopic thyroid tissue is a very rare condition of which most common site is in submandibular location. Endoscopic approach for removal of the diseased ectopic gland has not been reported earlier. The procedure is very safe and gives a much better cosmetic outcome.

References

1. Zieren J, Paul M, Scharfenberg M, et al. Submandibular ectopic thyroid gland. J Craniofac Surg 2006;17(6):1194–1198. DOI: 10.1097/01. scs.0000246502.69688.60.
2. Choi JY, Kim JH. A case of an ectopic thyroid gland at the lateral neck masquerading as a metastatic papillary thyroid carcinoma. J Korean Med Sci 2008;23(3):548–550. DOI: 10.3346/jkms.2008.23.3.548.
3. Huang TS, Chen HY. Dual thyroid ectopia with a normally located pretracheal thyroid gland: case report and literature review. Head Neck 2007;29(9):885–888. DOI: 10.1002/hed.20604.
4. Aköz T, Erdogan B, Ayhan M, et al. Ectopic submandibular thyroid tissue. Rev Laryngol Otol Rhinol (Bord) 1998;119(5):323–325.
5. Akanmu IN, Adewale OM. Lateral cervical ectopic thyroid masses with eutopic multinodular goiter: an unusual presentation. Hormones (Athens) 2009;8(2):150–153. DOI: 10.14310/horm.2002.1232.
6. Helidonis E, Dokianakis G, Papazoglou G, et al. Ectopic thyroid gland in the submandibular region. J Laryngol Otol 1980;94(2):219–224. DOI: 10.1017/S0022215100088708.
7. Yılmaz MS, Aytürk S, Güven M, et al. Submandibular ectopic thyroid with normally located thyroid gland. Kulak Burun Bogaz Ihtisas Derg 2014;24(1):50–53. DOI: 10.5606/kbbihtisas.2014.41713.
8. Feller KU, Mavros A, Gaertner HJ. Ectopic submandibular thyroid tissue with a coexisting active and normally located thyroid gland: case report and review of literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2000;90(5):618–623. DOI: 10.1067/moe.2000.108804.
9. Kanaya H, Tanigaito Y, Shyono N, et al. A rare case of ectopic, normally functioning thyroid tissue presenting as a left submandibular mass. Nippon Jibiinkoka Gakkai Kaiho 2005;108(9):850–853. DOI: 10.3950/jibiinkoka.108.850.
10. Kurukahveciŏglu S, Arslan H, Kocatürk S, et al. Ectopic thyroid gland at infrahyoid localization: case report. Kulak Burun Bogaz Ihtis Derg 2007;15(2):87–90.
11. Bersaneti JA, Silva RDP, Ramos RRN, et al. Ectopic thyroid presenting as a submandibular mass. Head and Neck Pathology 2011;5(1):63–66. DOI: 10.1007/s12105-010-0209-z.
12. Prado H, Prado A, Castillo B. Lateral ectopic thyroid: a case diagnosed preoperatively. Ear, Nose Throat J 2012;91(4):E14–E18. DOI: 10.1177/014556131209100417.
13. Çeliker M, Çeliker FB, Turan A, et al. Submandibular lateral ectopic thyroid tissue: ultrasonography, computed tomography, and scintigraphic findings. Case Rep Otolaryngol 2015;2015:769604. DOI:
    10.1155/2015/769604.

1–4Department of General and Laparoscopic Surgery, VPS Lakeshore Hospital and Keyhole Clinic, Kochi, Kerala, India

5,6Department of Laparoscopic Surgery, Verwandeln Institute, Kochi, Kerala, India

Corresponding Author:

Ramakrishnapillai Padmakumar,
Department of General and Laparoscopic Surgery,
VPS Lakeshore Hospital and Keyhole Clinic, Kochi, Kerala, India,
Phone: +91 9447230370, e-mail: drrpadmakumar@gmail.com

How to cite this article:
Padmakumar R, Balakrishnan A, Pai M, et al. Endoscopic Ectopic Thyroidectomy. World J Lap Surg 2020;13(3): 136–137.

Source of support: Nil
Conflict of interest: None

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Abstract

Laparoscopic inguinal anatomy detail is different from that is necessary for open hernia surgery. ‘PK Band’ is a condensation of areolar tissue lateral to inferior epigastric vessels on either side. It extends fromarcuate line to apex of triangle of doom. It is more condensed and prominent in the upper part. ‘PK Band’ is more prominent in males and tall structured individuals. It is an important land mark during laparoscopic inguinal dissection in both TEP (total extra peritoneal) and TAPP (trans abdominal preperitoneal) methods. Lateral blunt dissection in the preperitoneal plane will definitely be restricted by this band. Forcing the scope laterally will cause tear of the peritoneum at that level. Muscle injury may also be caused by blunt dissection. Division of this band during laparoscopic hernia repair connects the space of Bogros with space of Retzius. This provides sufficient space necessary for proper placement of mesh and hence significantly reduce recurrence.

Keywords : ‘PKBand’ . Laparoscopy . Inguinal Hernia . TAPP . TEP . Facia Condensation

Watch Video Presentation in YouTube

Conflict of Interest

None

Introduction

This is an observational information where we attempt to explain the anatomical importance and clinical significance of “PK Band” which is a fibrous band encountered while performing cases of laparoscopic inguinal hernia repair. During our 20 years of experience in performing over 6000 cases of laparoscopic hernia repairs, we made this observation. Identification of this structure is necessary for proper dissection and comfortable placement of mesh during laparoscopic inguinal hernia surgery.

Background

Nyhus et al. (in 1991) [1] have mentioned about transversalis fascia analogues [2, 3]. In the endoabdominal fascial sac, there are several locations of condensation of the fascia, which are continuous with and integrated to the sac itself.

These condensations, termed as transversalis fascia analogues, are found at points of insertion of various muscles or at points where aponeurotic structures are attached to the fascial sac. The five important fascial analogues are transversalis fascial sling, transversus abdominis aponeurotic arch, the iliopubic tract, iliopectineal ligament, and the interfoveolar ligament.

There is mention of medial boundary of Bogros space as a condensed area lateral to inferior epigastric vessel [4]. Also, there is mention of lateral boundary of Retzius space as condensation of tissue lateral to inferior epigastric vessel. But the surgical importance of this thickened area was not evaluated or described probably because it may not be important in open hernia surgery.

Location of the ‘PK Band’

The “PK band” is a condensation of the loose areolar tissue between the space of Bogros and the space of Retzius. It lies lateral to the inferior epigastric artery bilaterally. It extends from the arcuate line to the apex of the triangle of doom. It is more condensed and prominent in the upper part.

Prevalence of PK Band

It is seen in all patients. More prominent in males, tall statured and in well-built individuals.

Significance in Laparoscopic Hernia Repair

1. In totally extraperitoneal (TEP) repair

For surgeons, the lateral blunt dissection in the preperitoneal plane will definitely be restricted by this band. Forcing the scope laterally will cause tear of the peritoneum at that level. Extraperitoneal space is lost and the operating surgeon will not be able to complete a good lateral dissection. The procedure may have to be converted to intraperitoneal approach or open surgery due to air leak into the general peritoneal cavity. Many surgeons limit the lateral dissection to this band and place the mesh improperly. The principle behind mesh placement is that it should be placed equidistant laterally and medially to the inferior epigastric artery so that it adequately covers the indirect and direct hernial defects. Mesh placed without lateral dissection will not be covering the indirect hernia defect. Mesh folding can occur and it may lead to recurrence of hernia [5]. To prevent all these lapses during surgery and to avoid unnecessary delay and difficulty in dissection, the surgeon should divide “PK Band.”

2. In transabdominal preperitoneal (TAPP) repair

While dissecting in the loose areolar plane for placement of mesh, “PK band” is the landmark which is encountered lateral to the inferior epigastric artery. There is loose areolar tissue on either side of this band. In order to complete lateral dissection for smooth placement of mesh, it is imperative that just as in TEP the surgeon divides “PK Band.” If not identified, there is strong possibility for either peritoneal tear or injury to the muscles, and mesh placement will be improper leading to recurrence of hernia.

Conclusion

“PK Band” acts as an important anatomical landmark in both TAPP and TEP. Its division is necessary in proper completion of lateral dissection and proper placement of mesh.

References

1. Nyhus LM, KleinMS, Rogers FB, et al. (1991) Current problems in surgery. 28(6):407–450. doi: https://doi.org/10.1016/0011-38
2. MemonMA, Quinn THet al (1999) J Laparoendosc Adv Surg Techn 9(3):267–272. https://doi.org/10.1089/lap.1999.9.267
3. Annibali R., Fitzgibbons R.J. (1995) Laparoscopic Anatomy of the Abdominal Wall. In: Phillips E.H., Rosenthal R.J. (eds) Operative Strategies in Laparoscopic Surgery. Springer, Berlin, Heidelberg
4. Ansari MM, et al. (2017) Retzius and Bogros spaces: a prospective laparoscopic study and current perspectives.Ann IntMedDental Res 3(5)
5. Choy C, Shapiro K, Patel S et al (2004) Surg Endosc 18:523. https://doi.org/10.1007/s00464-003-8183-0

Author information

Affiliations

1. Department of General & Laparoscopic Surgery, VPS Lakeshore Hospital, Kochi, India
   • Padmakumar Ramakrishnapillai
   • , Sandeep Gupta
   • , Madhukara Pai
   • , Aravind Balakrishnan
   • , Kevin J Chiramel
   •  & Premna Subin
2. VSM Hospital, Mavelikkara, India
   • Farish Shams
3. Verwandeln Institute, Kochi, Kerala, 682306, India
   • Subin Thomas

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• Parathyroid surgery
• Bariatric surgery
• Gallbladder surgery
• Primary hyperthyroidism

Author: Dr R Padmakumar et al.

Contents

1. Introduction
2. Functions
3. Deficiency Causes
4. Correlation of calcium level in vitamin D deficiency
5. Primary hyperparathyroidism
6. Obesity & After Bariatric Surgery
7. Gallbladder Disease & Vitamin D Deficiency
8. Treatment

Introduction:

Vitamin D is a fat soluble vitamin and produced by skin (from cholesterol) when exposed to ultraviolet B radiation (“sunshine vitamin”) and also obtained from dietary sources, including supplements. Most important components, vitamin D3 (cholecalciferol) comes from fortified foods, animal foods (fish, eggs and liver) and can be made internally when skin is exposed to ultraviolet (uv) radiation from the sun and vitamin D2 (ergocalciferol)

• Deficiency: 25 (OH) D levels below 12 ng/ml
• Inadequate: 25 (OH) D levels between 12-20ng/ml
• An adequate: 25 (OH) D levels between 20-50ng/ml
• Excessive: 25 (OH) D levels over 50nglml

Excessive intakes of vitamin D can lead to high levels of calcium (hypercalcemia). The symptoms of this are weakness, confusion, constipation, loss of appetite and development of painful calcium deposits. To avoid this, advice to keep the supplement intake below the tolerable upper limits.

Functions:

The need for vitamin D goes way beyond preventing and treating rickets. Various researchers have claimed that vitamin D benefits are associated with the following:

1. Enhancing intestinal absorption of calcium and phosphorus
2. Prevention of osteoporosis and osteopenia
3. Allowing proper functioning of parathyroid hormone
4. Lowering blood pressure in people with hypertension
5. Lowering incidence and severity of cardiovascular disorders
6. Decreasing the incidence of type 2 diabetes: research has shown that those with blood vitamin D level over 25ng/ml had a 43% reduced risk of developing type 2 diabetes compared with those with levels under 14ng/ml
7. Decreasing inflammation: research has shown a decrease in levels of C-reactive protein , a marker of inflammation , with increased levels of vitamin D to just below 21ng/ml
8. Reducing risk of allergies in children and adolescents
9. Prevention of dental caries
10. Prevention and treatment of depression
11. Possibly helping with erectile dysfunction (ED)
12. Regulating cholesterol levels in the blood: it has been shown that without adequate sun exposure, vitamin D precursors turn to cholesterol instead of vitamin D
13. Decreasing mortality rate of certain cancers
14. Increase immunity
15. Deficiency in pregnancy may lead to gestational diabetes, pre eclampsia and small infants

Deficiency – Causes:

1. Inadequate sun exposure
2. Limited oral intake
3. Impaired intestinal absorption
4. Primary hyperparathyroidism
5. After bariatric surgery
6. After Gallbladder surgery
7. Parathyroid removal
   • Accidental during thyroid surgery
   • For parathyroid adenoma or hyperplasia

Correlation of calcium level in vitamin D deficiency

• If parathyroid function is normal
• When primary hyperparathyroidism

In vitamin D deficiency due to reduced oral intake or reduced sun exposure ,Vitamin D level and calcium level will be low with normal parathyroid function. Calcium level will be high and vitamin D will be low when primary hyperparathyroidism exists.

Primary hyperparathyroidism

• There is high calcium level in the blood
• May present with parathyroid tumor
• High PTH takes calcium from the bone and shut down calcium absorption
• There will be low vitamin D

Obesity and After Bariatric Surgery

Research has begun to show a relationship between BMI and vitamin D deficiency. A study done on 2,187 overweight and obese subjects, found that those with a BMI above 40 had 18% lower serum vitamin D levels than those with a BMI under 40. Some possible reasons for this are lower intakes of vitamin D , less exposure to sunlight (uv) radiation and a higher distribution volume of vitamin D . Even with exposure to sunlight, there remains a risk for deficiency.

Morbid obese patients have a higher incidence of high PTH and low vitamin D even before surgery compared with normal individuals.

In gastric bypass: low calcium absorption due to bypassed duodenum and jejunum may be the cause.

In sleeve gastrectomy the vitamin D and calcium level are maintained within normal limits compared to bypass surgery.

Bariatric Surgery

Bariatric Surgery deals with the study of obesity, the causes, prevention and treatment of obesity. Different weight loss procedures are Laparoscopic Sleeve Gastrectomy, Roux-en-Y Gastric Bypass, Mini Gastric Bypass, Laparoscopic Gastroplasty. Intragastric Balloon Placement is a non-surgical weight loss procedure.

Bariatric Surgery Sleeve Gastrectomy Roux-en-Y Gastric Bypass Gastroplasty Intragastric Balloon Placement

Gallbladder Disease and Vitamin D Deficiency

Low vitamin D may cause gallbladder stone formation

Gall bladder removal may reduce vitamin D and magnesium levels. This may be due to reduced absorption of fat soluble vitamins (D, E, A and K).

People with compromised liver or digestive function are often vitamin D deficient. Exposure of skin to the sun’s UVB rays enables body to manufacture vitamin D. However, this process occurs in the liver and kidneys. Here people with a sluggish liver often do not manufacture vitamin D adequately.

Gallbladder Surgery

The gallbladder is an organ that helps in the digestion process. Gallstones is a common cause of Gallbladder disease. Laparoscopic Cholecystectomy is a procedure performed to remove the gallbladder from the body

Laparoscopic Cholecystectomy

Treatment

1. Sun rays exposure
2. Increased oral supplements
3. When the blood level of vitamin D is below 30 ng/ml, a minimum of 1,000 IU/day will be needed for children and 1,500 to 2,000 IU/day of vitamin D3 for adults.
4. Another rule of thumb is for every 1 ng/ml increase in blood level one need is an additional 100IU/day.
5. In obese patients , with malabsorbtion syndromes and patients on medications affecting vitamin D metabolism, it is advised to have a higher dose (two to three times higher , atleast 6,000-10,000IU/day) of vitamin D to treat deficiency to maintain a 25(OH)D level above 30ng/ml, followed by maintenance therapy of 3,000-6,000IU/day.
6. After parathyroid removal , may be wise to have a concomitant replacement of vitamin D with calcitriol (0.25-1mcg/day). Dependence on calcium supplementation for more than 6 months shows permanent hypoparathyroidism; which will necessitate continued use of such medications.

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• Branchial cyst excision endoscopy.
• Axillary approach-no neck incision.
• First ever in a child.

Journal of Pediatric Surgery Case Reports

Abstract

This is the case report of a three-year-old female Arab child who presented with a neck swelling since one year of age. Physical examination was suggestive of a 6 × 4 cm swelling in the right lateral neck, which was dumbbell shaped, deep to sternocleidomastoid muscle on either side of the muscle. Ultrasound scan revealed a large cyst with internal echoes in the right carotid space anteriorly. The cyst was dissected endoscopically by an axillary approach and complete excision was achieved, thus avoiding the need for a large incision in the neck. No complications noted after treatment. The lowest age at which branchial cyst excision was carried out endoscopically was 18 years as per the available literature. Hence this case seems to be the first ever endoscopic branchial cyst excision in the pediatric age group.

1. Introduction

A branchial cyst is a congenital epithelial cyst on the lateral aspect of the neck, due to failure of obliteration of branchial cleft during embryonic development. It is the most common congenital cause of a neck mass. Though congenital, it may present at a later date due to secondary infection. The definitive treatment is surgical excision of the entire cyst wall.

Presently, the principles of remote access and endoscopy are combined together and are used regularly for removal of thyroid masses[1]. In literature there is a case report of endoscopic branchial cyst excision in a patient aged 18 years [2]. We hereby report a case of endoscopic branchial cyst excision in a 3-year-old child via axillary approach without a neck incision. This case seems to be the first ever endoscopic branchial cyst excision in the pediatric age group.

Read More on the Article in

Journal of Pediatric Surgery Case Reports

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Abstract

We were unable to find reports in the published medical literature of any cases of bowel surgery being successfully performed at such a low hemoglobin level, without blood transfusion or blood products pre or post-surgery, with the patient’s uncomplicated recovery. This study is about such a case. A patient presenting with severe gastrointestinal bleeding was diagnosed with enteric fever and multiple ileal ulcers. He had an extremely low hemoglobin level (2 g/dL) and mild renal and hepatic impairment. He was immediately admitted for right hemicolectomy under general anesthesia though he refused transfusion of blood or blood products prior to, during, or after surgery on religious grounds (Jehovah’ s Witnesses). After the surgery and having survived these potentially life-threatening circumstances, he left the hospital without major complications. In such circumstances, lives may be saved by prompt clinical decision-making, collaboration and swift surgical intervention coupled with the immediate consultation and input of the patient and family.

Keywords

Gastrointestinal bleed; Low hemoglobin level; Jehovah’s witnesses; Multiple terminal ileal ulceration; Enterotomy; Right hemicolectomy, Minimum hemoglobin for surgery.

Core tip:

It is unheard of in the medical history to take up a patient with hemoglobin of 2 g/dL for anesthesia and major bowel surgery, without transfusing blood or blood products prior to, during or after surgery; and saving the life without complications. We would like to report regarding such a patient who was treated at our hospital.

INTRODUCTION

Few, if any case studies in the published medical literature report on the successful, uncomplicated surgical outcome of a patient with hemoglobin of 2 g/dL for anesthesia and major bowel surgery, without blood transfusion or blood products pre or post-surgery. Thus, we would like to report our recent experience treating such a patient at our hospital.

CASE REPORT

A 39-year-old male patient was admitted to Sunrise Hospital, Cochin, Kerala, India; after being referred from a nearby hospital [from where he left against medical advice] with a history of severe lower gastrointestinal bleeding. The diagnosis was enteric fever with bleeding from the gastrointestinal tract. Salmonella typhi “O” and “H” titer was 1/640, which was confirmed by tube agglutination test (stained febrile antigen set, Manufacturer- Span diagnostics ltd, Sachin, Surat, Gujarat, India). His hemoglobin was extremely low (4 g%) at admission. On examination, he was very pale and icteric, with a pulse rate of 120/beats per minute and blood pressure of 110/70 mmHg and mild distension of the abdomen was present. Table 1 shows the blood investigations of the patient at admission. An immediately performed colonoscopy showed multiple terminal ileal ulcerations with diffuse active bleeding that was not amenable to either colonoscopic or radiological intervention at that stage. Hence, immediate surgical intervention was planned.

It was decided as absolutely necessary to transfuse blood and blood products to correct the hemoglobin level, which had dropped to 2 g/dL within 12 h of admission. We discussed with the patient’s relatives the urgent need for blood transfusion, before proceeding with surgery to arrest the ongoing bleeding. However, the patient, his wife and other relatives were firm in their decision not to receive blood or blood products, due to their religious beliefs. Giving erythropoietin alone at that time was not an option in this patient with active bleeding. Thus, we were faced with the option of not performing the surgery at all, but at the almost certain cost of the patient’s life. Wanting to give the patient the chance to survive, it was decided to offer the patient surgery, despite his clinical condition and the treatment constraints mentioned above that make for an extremely risky surgery. The patient was prepared for laparotomy with a high-risk consent that also listed the distinct possibility of death during surgery and the continued unwillingness to receive blood or blood products.

The patient was given general anesthesia, during which minimal intravenous fluids were administered to maintain the blood pressure at 100/60 mmHg, as overhydration could lead to a further drop of in hemoglobin levels. The bowel was found to be thickened at the ileocecal region. Enterotomy was performed and linear ulcers with bleeding base were observed at the terminal ileum. Limited right hemicolectomy was performed, excising the distal 21 cm of ileum including the ulcer, cecum, appendix and a portion of ascending colon. The entire bowel was edematous,and the blood that oozed during resection appeared thin and watery. To reduce the duration of surgery, reconstruction was attempted by side-to-side ileocolic stapler anastomosis, but staplers did not hold due to bowel wall edema. Revision of anastomosis with hand-sewn, end-to-end, ileocolic two-layer suturing was performed with 3-0 Vicryl (polyglycolic acid)-continuous all coat and 3-0 silk, intermittent seromuscular.

Postoperatively, the patient was kept in the intensive care unit for 10 d with nasal oxygen to enrich the available hemoglobin with oxygen. He had features of renal and hepatic impairment, which gradually improved (Table 2). Improvement in hemoglobin level is presented in Figure 1. Total parenteral nutrition and albumin infusion were initiated. He was given intravenous ferric carboxymaltose and recombinant erythropoietin. Small doses of frusemide were given intravenously for the benefit of hemoconcentration.

The patient was put on oral fluids on the fifth day of surgery, which was gradually switched over to soft diet by the seventh day. A high protein, high calorie diet was given together with oral iron supplementation. Gradually, his hemoglobin level improved, as shown in Table 2. The biopsy report showed linear ulcers in the distal ileum, with the largest measuring 1.5 cm. Microscopy showed a mucosal ulcer infiltrated by histiocytes, lymphocytes, plasma cells and occasional neutrophils consistent with typhoid ulcer ileum. No granulomas were present.

DISCUSSION

We were unable to find reports in the published medical literature of any similar cases of bowel surgery being successfully performed at such a low hemoglobin level with the patient’s uncomplicated recovery. Various reports of surgeries performed without blood transfusion in severely anemic patients who does not have the minimum hemoglobin for surgery (hemoglobin level less than 5 g/dL) showed mortality of approximately 50%^[4], which further increased to 91% in the 2 to 3 g/dL group^[5].

Hemoglobin is the major oxygen-carrying protein in blood. Low hemoglobin level is generally defined as less than 13.5 g/dL for men and less than 12 g/dL for women. Unless it is very severe, anemia, perse, does not cause any problems with anesthesia or surgery. Severe anemia can precipitate cardiac arrest due to (1) increased cardiac output; (2) vasodilatation associated with anesthesia; and (3) hemodilution during resuscitation of hemorrhagic shock using saline. The degree of hemodilution is directly associated with acid base imbalance and is proportional to the level of compromise of metabolic recovery, which in turn is projected on to mortality^[6]. In addition, tissue hypoxia due to reduced oxygen-carrying capacity of the blood leads to end-organ damage and systemic immune response syndrome. There also is a risk of surgical site infection, delayed wound healing and bowel leak at the site of the anastomosis.

Jehovah’s Witnesses are a Christian denomination with non trinitarian beliefs distinct from mainstream Christianity and have a worldwide membership of over 7.78 million people. Their beliefs are based on interpretations of the Bible and they prefer to use their own translation. They refuse blood transfusions, which they consider to be a violation of God’s law based on their interpretation of Acts 15:28, 29. Since 1961, the willing acceptance of blood transfusion by an unrepentant member has been grounds for expulsion from the religion. Watch Tower Society literature directs Witnesses to refuse blood transfusions, even in “a life-or-death situation”^[7]. They refuse transfusions of whole blood or of any of its four primary components – red cells, white cells, platelets and plasma (serum)^[7,8].

It is always a dilemma for the surgeon to decide whether to urgently operate on a bleeding patient with hemorrhagic shock without blood transfusion. We believe that life-saving interventions should not be delayed for patients with active bleeding, even though blood transfusion may be refused. Atabek and colleagues have reported a case of active bleeding in a Jehovah’s Witness patient, where early surgery led to rescue of the patient^[9]. Initial conservative treatment with delayed surgery led to a 75% mortality rate, compared with a 20% mortality rate in patients who underwent emergency surgical intervention within 24 h of admission to the hospital^[10,11].

Our patient was at high-risk for cardiac arrest due to oxygen depletion, but eventually survived without complications. Anastomotic site healing was also potentially in jeopardy, but he recovered without leak.

In conclusion, even in the rarest of situations, such as the one described, a successful attempt to save the patient’s life can be made, even though the general condition of the patient is clinically unstable. Prompt decisionmaking and effective communication among the treating doctors and with the patient and his relatives were important factors that helped the patient’s positive outcome. Had the clinical team refused to perform surgery on this patient, or even delayed the operation because of his extremely low hemoglobin, death would likely have been assured.

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^*Author contributions: Padmakumar R performed surgical operation, designed the report; Pai M and Farish S contributed to the surgical operation, review of literature, data collection; Rajeev J contributed to the attending doctor for the patients (colonoscopy), editing assistance; Sanjeev T, anesthesiologist for the procedure, was responsible for the literature review; Sreevalsan TV was critical care specialist; Sheetal B and Sooraj YS were attending doctors for the patient; Rowther SS organized the report, statistics, and manuscript preparation.

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