Retraction Watch

Tracking retractions as a window into the scientific process

Patient misdiagnosed with rare neurological side effect in retracted case study

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When two surgeons in Greece learned that a patient had developed a rare side effect following weight loss surgery, they were eager to publish the case.

After extensive testing, the patient was diagnosed with Wernicke’s encephalopathy—a neurological disorder caused by thiamine deficiency—following a sleeve gastrectomy procedure. As the authors note in the paper, they had seen only eight other cases following the procedure in the literature.

It turns out, theirs was not the ninth. After the patient unfortunately died, he was examined by a coroner, who ruled he did not, in fact, have Wernicke’s encephalopathy. So Dimitrios Manatakis and Nikolaos Georgopoulos, both based at Athens Naval and Veterans Hospital in Greece, have retracted their 2014 case study.

When the first learned of the patient, the authors wanted to alert the surgical community to the case, given the rarity of this side effect, Manatakis told us:

We wished to report this case, in order to raise a red flag, because vitamin deficiencies are quite rare following restrictive bariatric procedures. Indeed very few cases of thiamine deficiency after sleeve gastrectomy have been reported in the literature so far.

But the coroner’s report revealed that “typical histopathological lesions of Wernicke’s encephalopathy were absent” and thus could not confirm the diagnosis, Manatakis said:

… the coroner reviewing the case and post-mortem histopathology stated that the typical histopathological lesions of Wernicke’s encephalopathy were absent, therefore not confirming the initial diagnosis, with a final diagnosis of encephalopathy of indeterminate aetiology. Our re-examination of the patient’s file showed that post-operative vitamin B1 levels were at low-normal levels, but still within the institutional reference range.

Manatakis said that the cause of death was later deemed to be an unconfirmed form of encephalopathy, though not Wernicke’s, which can be tricky to diagnose and is often missed.

Manatakis elaborated on how difficult a Wernicke’s encephalopathy diagnosis can be:

There is no specific routine laboratory test for the diagnosis of Wernicke’s encephalopathy. Vitamin B1 levels may be within normal range, as in our patient. Even MRI scans have a sensitivity of around 50%, which means that a normal scan cannot exclude the diagnosis. Improvement with thiamine administration is perhaps the only way to practically confirm the diagnosis. However, even with replacement therapy, half of the patients will still exhibit signs of cognitive impairment.

According to the paper, things began to take a turn about two months post-surgery, when the 51-year-old returned to the hospital suffering from “protracted vomiting and malaise,” which Manatakis said is not uncommon after this type of surgery.

During his stay, the patient developed neurological symptoms, Manatakis said. His symptoms and clinical course ultimately led to a diagnosis of Wernicke’s encephalopathy.

Manatakis noted that the patient’s care required a “multidisciplinary approach, in order to treat the patient in the best possible way.” But the patient’s condition continued to deteriorate, and a week after being admitted, he died in the hospital, Manatakis told us:

The patient died in the ICU as a result of complications of the encephalopathy, and ultimately respiratory failure.

When we asked when did he and his co-author first learned about the coroner’s report, Manatakis explained:

…the coroner’s final report took about 18 months time, with review of the files, histopathology and toxicology tests. The team was notified some time around mid-2016. We contacted the publisher in December in order to request the retraction of the paper.

Here’s another wrinkle. The authors state in the paper that they received the family’s written consent to publish the report (“This report is published with the written consent of the patient’s family”), but it turns out that the consent they received did not explicitly permit them to publish the report. Manatakis told us:

Finally it came to our understanding that the operation consent form, stating that details from the patient file can be anonymously used for scientific reasons, is not adequate and that explicit consent must be given for publication.

Manatakis said that, “For these reasons, we felt inclined to request the retraction of the paper.”

The March 2017 retraction notice for “A Fatal Case of Wernicke’s Encephalopathy after Sleeve Gastrectomy for Morbid Obesity” published in Case Reports in Surgery and not yet indexed by Clarivate Analytics’ Web of Science, formerly part of Thomson Reuters, summarizes the situation:

At the request of the authors, the article titled “A Fatal Case of Wernicke’s Encephalopathy after Sleeve Gastrectomy for Morbid Obesity” [1] has been retracted. The coroner reviewed the case and postmortem histopathology and concluded that the typical histopathological lesions of Wernicke’s encephalopathy were absent, therefore not confirming the initial diagnosis. Reexamination of the patient’s file showed that postoperative vitamin B1 levels were within the institutional reference range; no hyperemesis and no neurological signs were revealed. The article stated that “This report is published with the written consent of the patient’s family,” but there was misunderstanding and the family did not give explicit permission for publication.

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